[CLINICAL AND EPIDEMIOLOGICAL PECULIARITIES OF CYSTIC ECHINOCOCCOSIS IN CHILDREN].

PubMed

Melia, Kh; Kokaia, N; Manjgaladze, M; Kelbakiani-Kvinikhidze, T; Sulaberidze, G

2017-04-01

The postoperative period of cystic echinococcosis was studied in 13 children. Demographic, epidemiological, clinical diagnosis, treatment, number location, and development of cysts and serologic data were analyzed. Age of children at diagnosis range 5 to 17 years. All patients with cystic echinococcosis had abdominal cysts. The liver was the main organ involved in ten patients (76,9%) - they had cysts located in the liver, two patients (15,4%) had lung cyst, one patient had concomitant lung and liver cysts. Twelve patients had single cysts and one had more than one abdominal cysts. Surgical treatment was performed in 23,1% cases. Ultrasound studies (US) were performed during the monitoring period. Evaluation of cysts was assessed by monitoring US changes. Positive dynamics was revealed in all patients; relapse of the disease was not noticed. Proceeding from the fact that in all patients echoarchitectonics of the hepatic tissue was lumped with a non-uniform structure and uneven ultrasound distribution, it is assumed that these changes are indicative of the development of connective tissue in the liver.

Hepatic and pulmonary cystic echinococcosis in a patient from the Central African Republic.

PubMed

Develoux, Michel; Enache-Angoulvant, Adela; Gounant, Valerie; Brian, Emmanuel; Khalil, Antoine; Bazelly, Bernard; Hennequin, Christophe

2011-03-01

Apical lung opacity was diagnosed in an asymptomatic 30 year-old woman native of Central African Republic by routine chest X-ray. CT scan demonstrated an excavated pulmonary mass and revealed a simple hepatic cyst. Tuberculosis was suspected but mycobacterial cultures remained negative. Three months later, ultrasonography showed septations within the hepatic lesion suggestive of cystic echinococcosis. The detection of seric anti-Echinococcus antibodies was positive. Hepatic and pulmonary cysts were removed surgically and association with three-month course of albendazole resulted in a favorable outcome. Cystic echinococcosis is exceptional in Central Africa and to our knowledge never reported from the Central African Republic. Copyright © 2011 Elsevier Ltd. All rights reserved.

An autochthonous case of cystic echinococcosis in Finland, 2015.

PubMed

Hämäläinen, Sari; Kantele, Anu; Arvonen, Miika; Hakala, Tapio; Karhukorpi, Jari; Heikkinen, Jukka; Berg, Ensio; Vanamo, Kari; Tyrväinen, Erja; Heiskanen-Kosma, Tarja; Oksanen, Antti; Lavikainen, Antti

2015-01-01

We report a case of pulmonary cystic echinococcosis in a child from eastern Finland with no history of travelling abroad. The cyst was surgically removed and the organism molecularly identified as Echinococcus canadensis genotype G10. This parasite is maintained in eastern Finland in a sylvatic life cycle involving wolves and moose; in the present case, the infection was presumably transmitted by hunting dogs.

Trends and correlates of cystic echinococcosis in Chile: 2001–2012

PubMed Central

Gelaw, Fkadu; Antillon, Marina; Fuentes, Rodrigo; Weinberger, Daniel Martin

2017-01-01

Echinococcosis is a neglected zoonotic disease affecting over 1 million people worldwide at any given time. It is the leading cause of hospital admissions for parasitic diseases in Chile. We conducted a retrospective investigation of hospitalized cases to describe the epidemiological trends of echinococcosis in Chile. We also examined the potential environmental risk factors for echinococcosis hospitalization rates. Through nation-wide hospital discharge data, a total of 11,516 hospitalized patients with cystic echinococcosis were identified between January 2001 and December 2012. The mean age of hospitalization was 40 years, with notable gender difference in pediatric patients. The hospitalization rate was found to be overall steadily decreasing from 2001 (7.02 per 100,000) to 2012 (4.53 per 100,000) with a 5% decrease per year (rate ratio = 0.95 [95% CI: 0.94, 0.96]). The hospitalization rate was higher in the south of Chile compared to the north. Goat density and intermediate precipitation were found to be significantly positively associated with the hospitalization rate while annual average temperature was found to be significantly negatively associated with the hospitalization rate. Findings of this study indicate that echinococcosis is still an important public health burden in Chile related to interaction with livestock and climate. Efforts should be placed on targeted prevention measures for farmers and raising awareness of echinococcosis among health care workers. PMID:28915247

Retrospective study of human cystic echinococcosis over the past decade in France, using a nationwide hospital medical information database.

PubMed

van Cauteren, Dieter; Millon, Laurence; de Valk, Henriette; Grenouillet, Frederic

2016-11-01

No specific surveillance system of cystic echinococcosis infections in humans exists in France. The incidence and trends over time have not been documented since the last decade of the twentieth century. We performed the current analysis to assess human cystic echinococcosis epidemiology over a 10-year period in France (including the island of Corsica but excluding the overseas territories) using a nationwide hospital medical information database. A total number of 2629 patients were identified in this database between 2005 and 2014. The average annual incidence rate was 0.42 hospitalized cases/100,000 inhabitants. It was highest in the Island of Corsica (1.76 cases/100,000) and in the region Provence-Alpes-Côte d'Azur (0.85 cases/100,000). This retrospective analysis of hospital records provides a population-based estimate of cystic echinococcosis incidence and trends over a 10-year period at a national level. It indicates a significant decrease of the incidence between 2005 and 2014 at the national level and in the area that reported the highest incidence. It stresses the fact that in France, cystic echinococcosis is not re-emerging and that the incidence remains low in comparison with similar studies in other Mediterranean countries.

Echinococcus canadensis G7 (pig strain): an underestimated cause of cystic echinococcosis in Austria.

PubMed

Schneider, Renate; Gollackner, Bernd; Schindl, Martin; Tucek, Gerhard; Auer, Herbert

2010-05-01

Anamnesis data of 104 patients with Cystic Echinococcosis were correlated retrospectively with the detected species/strain of Echinococcus. Ninety-two percent (N = 23) of autochthonous Austrian and 33% (N = 9) of patients with former Yugoslavian (YU) origin were infected with E. canadensis G7, the pig strain. All patients originating from Turkey harbored E. granulosus G1, the sheep strain. All E. canadensis G7-infected patients showed small liver cysts (ø 5.9 cm), only one of them an additional lung cyst. The median age at the time of operation of the Austrian patients was 55 years, of the Turkish patients 30 years, and of the former YU patients 23 years in the E. canadensis and 42 years in the E. granulosus-infected patients, respectively. The unexpected high number of E. canadensis G7-infected patients and the immigrants' young age show the importance of E. canadensis as a cause of human Cystic Echinococcosis in Central Europe and accordingly this new species has to be included into future echinococcosis control programs.

The retrospective annual surgical incidence of cystic echinococcosis in Sivas, Turkey.

PubMed

Gonlugur, U; Ozcelik, S; Gonlugur, T E; Arici, S; Celiksoz, A; Elagoz, S; Cevit, R

2009-06-01

Cystic echinococcosis is an important public health problem in our region. The aim of this study was to assess the surgical incidence of cystic echinococcosis in Sivas, Turkey. A retrospective follow-up study was carried out between 1997 and 2004. The mean age of 124 men (39.2%) and 192 women was 36.4 years. The liver was the primary site of cyst development (76.6%), followed by the lungs (19.9%), the spleen (5.4%), the kidneys (3.5%) and the peritoneal cavity (3.2%). Mean annual surgical incidence was 6.4 per 100,000 inhabitants for a 7-year period. The most common site of recurrence was the liver. Hepatic cysts were more frequently infected than pulmonary cysts (P < 0.05). In those patients with an infected cyst, the mean circulating eosinophil level was significantly lower than in those without. Rupture and infection were more common in men (P < 0.01). In conclusion, serious structural, legislative, surveillance, prevention and control measures for echinooccus should be undertaken in our region.

Abattoir based survey of bovine cystic echinococcosis in selected commercial abattoir in Ethiopia

USDA-ARS?s Scientific Manuscript database

Hydatidosis is a widespread parasitic disease posing a significant public health and economic burden in developing countries. Abattoir survey was conducted to determine the prevalence of cystic echinococcosis (CE) in cattle, its organ distribution and to estimate financial loss due to organ condemna...

Echinococcosis of the Liver

DTIC Science & Technology

2007-03-01

and 1b. The lack of internal enhancement confirms the cystic nature of these lesions. Discussion Echinococcosis , also known as Hydatid disease... Echinococcosis of the Liver Radiology Corner Echinococcosis of the Liver Guarantor...abbreviated answer in the March 2007 issue. 1 Echinococcosis is a parasitic infection that poses a potential health threat to military members and

Widespread co-endemicity of human cystic and alveolar echinococcosis on the eastern Tibetan Plateau, northwest Sichuan/southeast Qinghai, China.

PubMed

Li, Tiaoying; Chen, Xingwang; Zhen, Ren; Qiu, Jiamin; Qiu, Dongchuan; Xiao, Ning; Ito, Akira; Wang, Hu; Giraudoux, Patrick; Sako, Yasuhito; Nakao, Minoru; Craig, Philip S

2010-03-01

Cystic echinococcosis (CE) or hydatid disease is known to be cosmopolitan in its global distribution, while alveolar echinococcosis (AE) is a much rarer though more pathogenic hepatic parasitic disease restricted to the northern hemisphere. Both forms of human echinococcosis are known to occur on the Tibetan Plateau, but the epidemiological characteristics remain poorly understood. In our current study, abdominal ultrasound screening programs for echinococcosis were conducted in 31 Tibetan townships in Ganze and Aba Tibetan Autonomous Prefectures of northwest Sichuan Province during 2001-2008. Hospital records (1992-2006) in a major regional treatment centre for echinococcosis in Sichuan Province were also reviewed. Of 10,186 local residents examined by portable ultrasound scan, 645 (6.3%) were diagnosed with echinococcosis: a prevalence of 3.2% for CE, 3.1% for AE and 0.04% for dual infection (both CE and AE). Human cystic and alveolar echinococcosis in pastoral areas was highly co-endemic, in comparison to much lower prevalences in semi-pastoral or farming regions. The high ultrasound prevalence in these co-endemic areas in northwest Sichuan Province was also reflected in the hospital study, and hospital records furthermore indicated another possible highly co-endemic focus in Guoluo Prefecture of Qinghai Province, located at the border of northwest Sichuan. These chronic cestode zoonoses constitute an unparalleled major public health problem for pastoral Tibetan communities, and pose great difficulties for adequate treatment access and effective transmission control in such remote regions. Copyright 2009 Elsevier B.V. All rights reserved.

Genotype identification of human cystic echinococcosis in Isfahan, central Iran.

PubMed

Kia, Eshrat Bigom; Rahimi, Hamidreza; Sharbatkhori, Mitra; Talebi, Ardeshir; Fasihi Harandi, Majid; Mirhendi, Hossein

2010-08-01

Echinococcosis/hydatidosis is one of the most important zoonotic diseases commonly found in different regions of Iran with a major economic and public health importance. In the current study, Echinococcus granulosus isolates were collected from hospitalized patients in Isfahan, central Iran. The genotypes of 30 samples were determined by polymerase chain reaction amplification of internal transcribed spacer-1 region of ribosomal DNA, followed by restriction fragment length polymorphism (RFLP) with two restriction enzymes namely AluI and MspI. As expected, each isolate yielded an approximately 1-kbp DNA fragment on the electrophoresis gel. According to RFLP results for both enzymes, all isolates had an equal pattern indicating the G1 genotype. Our findings confirmed that G1 is the dominant genotype of cystic echinococcosis in human in central Iran, with predilection to different organs including liver, lung, and brain, and warrants the importance of sheep dog cycle in public health.

Global Distribution of Alveolar and Cystic Echinococcosis.

PubMed

Deplazes, P; Rinaldi, L; Alvarez Rojas, C A; Torgerson, P R; Harandi, M F; Romig, T; Antolova, D; Schurer, J M; Lahmar, S; Cringoli, G; Magambo, J; Thompson, R C A; Jenkins, E J

2017-01-01

Alveolar echinococcosis (AE) and cystic echinococcosis (CE) are severe helminthic zoonoses. Echinococcus multilocularis (causative agent of AE) is widely distributed in the northern hemisphere where it is typically maintained in a wild animal cycle including canids as definitive hosts and rodents as intermediate hosts. The species Echinococcus granulosus, Echinococcus ortleppi, Echinococcus canadensis and Echinococcus intermedius are the causative agents of CE with a worldwide distribution and a highly variable human disease burden in the different endemic areas depending upon human behavioural risk factors, the diversity and ecology of animal host assemblages and the genetic diversity within Echinococcus species which differ in their zoonotic potential and pathogenicity. Both AE and CE are regarded as neglected zoonoses, with a higher overall burden of disease for CE due to its global distribution and high regional prevalence, but a higher pathogenicity and case fatality rate for AE, especially in Asia. Over the past two decades, numerous studies have addressed the epidemiology and distribution of these Echinococcus species worldwide, resulting in better-defined boundaries of the endemic areas. This chapter presents the global distribution of Echinococcus species and human AE and CE in maps and summarizes the global data on host assemblages, transmission, prevalence in animal definitive hosts, incidence in people and molecular epidemiology. Copyright © 2017 Elsevier Ltd. All rights reserved.

Field evaluation of an immunochromatographic test for diagnosis of cystic and alveolar echinococcosis.

PubMed

Gao, Chun-Hua; Wang, Jun-Yun; Shi, Feng; Steverding, Dietmar; Wang, Xia; Yang, Yue-Tao; Zhou, Xiao-Nong

2018-05-23

The larval stages of the tapeworms Echinocoocus granulosus and Echinococcus multilocularis are the causative agents of human cystic echinococcosis (CE) and human alveolar echinococcosis (AE), respectively. Both CE and AE are chronic diseases characterised by long asymptomatic periods of many years. However, early diagnosis of the disease is important if treatment and management of echinococcosis patients are to be successful. A previously developed rapid diagnostic test (RDT) for the differential detection of CE and AE was evaluated under field conditions with finger prick blood samples taken from 1502 people living in the Ganzi Tibetan Autonomous Prefecture, China, a region with a high prevalence for both forms of human echinococcosis. The results were compared with simultaneously obtained abdominal ultrasonographic scans of the individuals. Using the ultrasonography as the gold standard, sensitivity and specificity, and the diagnostic accuracy of the RDT were determined to be greater than 94% for both CE and AE. For CE cases, high detection rates (95.6-98.8%) were found with patients having active cysts while lower detection rates (40.0-68.8%) were obtained with patients having transient or inactive cysts. In contrast, detection rates in AE patients were independent of the lesion type. The positive likelihood ratio of the RDT for CE and AE was greater than 20 and thus fairly high, indicating that a patient with a positive test result has a high probability of having echinococcosis. The results suggest that our previously developed RDT is suitable as a screening tool for the early detection of human echinococcosis in endemic areas.

Enhanced chemoprophylactic and clinical efficacy of albendazole formulated as solid dispersions in experimental cystic echinococcosis.

PubMed

Pensel, Patricia E; Castro, Silvina; Allemandi, Daniel; Bruni, Sergio Sánchez; Palma, Santiago D; Elissondo, María Celina

2014-06-16

Cystic echinococcosis is a chronic, complex, and still neglected disease. Although albendazole has demonstrated efficacy, only about one-third of patients experience complete remission or cure and 30-50% of treated patients develop some evidence of a therapeutic response. Different strategies have been developed in order to improve the albendazole water solubility and dissolution rate. The aim of the current work was to investigate the chemoprophylactic and clinical efficacy of an albendazole:poloxamer 188 solid dispersion formulation on mice infected with Echinococcus granulosus metacestodes. Albendazole formulated as solid dispersion had greater chemoprophylactic and clinical efficacy than albendazole alone. The improved in therapeutic efficacy could be a consequence of the increase in the systemic availability of albendazole sulfoxide. The work reported here demonstrates that in vivo treatment with albendazole:poloxamer 188 impairs the development of the hydatid cysts. This new pharmacotechnically based strategy could be a suitable alternative for treating cystic echinococcosis in humans. Copyright © 2014 Elsevier B.V. All rights reserved.

Economic Impact of Cystic Echinococcosis in Peru

PubMed Central

Moro, Pedro L.; Budke, Christine M.; Schantz, Peter M.; Vasquez, Julio; Santivañez, Saul J.; Villavicencio, Jaime

2011-01-01

Background Cystic echinococcosis (CE) constitutes an important public health problem in Peru. However, no studies have attempted to estimate the monetary and non-monetary impact of CE in Peruvian society. Methods We used official and published sources of epidemiological and economic information to estimate direct and indirect costs associated with livestock production losses and human disease in addition to surgical CE-associated disability adjusted life years (DALYs) lost. Findings The total estimated cost of human CE in Peru was U.S.$2,420,348 (95% CI:1,118,384–4,812,722) per year. Total estimated livestock-associated costs due to CE ranged from U.S.$196,681 (95% CI:141,641–251,629) if only direct losses (i.e., cattle and sheep liver destruction) were taken into consideration to U.S.$3,846,754 (95% CI:2,676,181–4,911,383) if additional production losses (liver condemnation, decreased carcass weight, wool losses, decreased milk production) were accounted for. An estimated 1,139 (95% CI: 861–1,489) DALYs were also lost due to surgical cases of CE. Conclusions This preliminary and conservative assessment of the socio-economic impact of CE on Peru, which is based largely on official sources of information, very likely underestimates the true extent of the problem. Nevertheless, these estimates illustrate the negative economic impact of CE in Peru. PMID:21629731

Pulmonary complications of cystic echinococcosis in children in Uruguay.

PubMed

Sakamoto, Tsukasa; Gutierrez, Carmen

2005-08-01

Cystic echinococcal lesions surgically removed from 99 patients aged 1-15 years in Uruguay were histopathologically studied. The lesions were collected from the lungs, liver, peritoneum, omentum, pleura, ovary, appendix and perinephrium. All intact cysts were well-developed and unilocular. Pulmonary and hepatic fertile cysts in which protoscoleces were produced were in 75% and 47%, respectively, of all patients. Fertile cysts having many protoscoleces were in the lungs of a 1-year-old infant. Sixteen ruptured pulmonary cysts were found in the lungs of 13 patients and four ruptured hepatic cysts were found in the liver of three patients. Pulmonary complications were seen in patients having pulmonary echinococcal cysts. In lung tissue surrounding echinococcal cysts, pulmonary complications included chronic congestion, hemorrhage, bronchopneumonia, as well as interstitial pneumonia that included histological changes caused by bronchiolitis, organizing pneumonia and obstructive pneumonia. Liver tissue adjacent to echinococcal cysts showed atrophy of hepatic cells, proliferation of interstices, small focal hemorrhages, and infiltration of mononuclear cells. The present study shows that a close relationship exists between pulmonary echinococcosis and pneumonia. The term 'echinococcal pneumonia' is proposed for pneumonia accompanying pulmonary echinococcosis.

Cystic echinococcosis in cattle slaughtered at Gondar Elfora export Abattoir, northwest Ethiopia.

PubMed

Abebe, Abebaw; Beyene, Desta; Kumsa, Bersissa

2014-12-01

A cross sectional and retrospective studies were conducted from November 2010 to April 2011 to determine the prevalence and characteristics of hydatid cysts in cattle slaughtered at Gondar Elfora Abattoir in northern Ethiopia. Out of the 308 cattle examined for the presence of hydatid cysts, 63 (20.5 %) of them were found harboring hydatid cysts in one or more of their internal organs. Results of the study showed higher (P < 0.05) prevalence of cystic echinococcosis in adult (22.4 %; 49/219) than in young (15.7 %; 14/89) cattle. Lowest prevalence of hydatid cysts was observed in cattle from Debark (7.7 %; 2/26) whereas highest prevalence of cystic echinococcosis was recorded in cattle from Fogera (28.2 %; 11/39). The lungs (69.3 %; 61/88) were the most affected organs followed by the livers (28.4 %; 25/88). A total of 230 hydatid cysts from different internal organs of 63 infected cattle were differentiated into 60.4 % calcified, 30 % fertile and 9.6 % sterile cysts. Likewise, a total of 13 % (30/230) small, 15.6 % (36/230) medium, and 10.9 % (25/230) large hydatid cysts were recorded. Lung harbored the highest overall count (76.9 %; 177/230) and greatest proportion of large size cysts (13.5 %; 103/177) than all the other organs. Liver harbored the highest proportion of calcified cysts (68 %; 34/50). An overall proportion of 30 % (69/230) fertile cysts were recorded. The greatest proportion of fertile cysts (33.3 %; 59/177) was recorded in lungs followed by the livers (20 %; 10/50). A retrospective data of 5 years (2004/5-2009/10) revealed an overall prevalence of 30.4 % (9,106/29,951) and highest overall prevalence of 65.5 % hydatid cysts in the lungs followed by livers (33.5 %) and least in spleen (0.04 %). In conclusion the findings reported herein show that cystic echinococcosis is widespread in cattle slaughtered in Gondar export Abattoir and suggests that the lung is the most important source of hydatid cysts for definitive hosts in

[Echinococcosis hyper-endemic and cystic fertility in pigs of the inter-andean valley of Huancarama, Peru].

PubMed

Sierra-Ramos, Rony; Valderrama-Pomé, Aldo

2017-01-01

The objective of the research was to determine the percentage of echinococcosis infection in swine, cystic fertility and associated factors. Cross-sectional study. The population was formed by 579 pigs slaughtered in the slaughterhouse of Huancarama. Hydatid cysts were detected by postmortem evaluation. Fertility was determined by measuring the diameter of the cysts and microscopically observing the germinative membrane in search of protoscoleces. The percentage of cystic echinococcosis infection was 76.7% (CI 95%: 73.2-80.3). Pigs older than two years were more likely to have cysts (RP 1.03; CI 95%: 1.0-1.1; (P < 0.01), as well as those coming from the Andahuaylas district (RP 1.2; CI 95%: 1.1-1.4; P < 0.01). Lung cysts showed a lower percentage of infection than hepatic ones. We examined 346 hydatid cysts of 334 pigs, a total fertility of 42.8% was found (CI 95%: 37.4-48.1).

Spinal Cystic Echinococcosis – A Systematic Analysis and Review of the Literature: Part 2. Treatment, Follow-up and Outcome

PubMed Central

Neumayr, Andreas; Tamarozzi, Francesca; Goblirsch, Sam; Blum, Johannes; Brunetti, Enrico

2013-01-01

Bone involvement in human cystic echinococcosis (CE) is rare, but affects the spine in approximately 50% of cases. Despite significant advances in diagnostic imaging techniques, surgical treatment and introduction of pharmacological therapy, spinal echinococcosis remains associated with a high degree of morbidity, disability and mortality. We systematically reviewed the published literature of the last five decades to update and summarize the currently existing data on treatment, follow-up and outcome of spinal CE. PMID:24069501

Comparative evaluation of western blotting in hepatic and pulmonary cystic echinococcosis.

PubMed

Akisu, C; Delibas, S B; Bicmen, C; Ozkoc, S; Aksoy, U; Turgay, N

2006-12-01

Many serological tests are widely used in the diagnosis of cystic echinococcosis (CE), caused by the larval stages of Echinococcus granulosus. The present study was carried for differentiation between hepatic and pulmonary cystic echinococcosis by Western Blotting (WB). A total of 121 sera from patients with hepatic CE (37), pulmonary CE (31) and controls (53; consisting of six healthy, seven Hymenolepis nana infection, 20 hepatic and 20 pulmonary diseases other than CE) were examined. In all of the CE patients, E. gronulosus infection was confirmed by surgical intervention. Sera were previously tested using IHA and ELISA to detect the E. gronulosus specific antibodies. Sera from hepatic cases of CE reacted with 16 polypeptides of 6-116 kDa and sera from pulmonary cases of CE reacted with 14 polypeptides of 4-130 kDa by Western Blotting. The WB test enabled the detection of antibodies in the hepatic CE samples for proteins of 24, 32 34, 44-46 and 52-54 kDa in molecular weight in 78.4%, 75.7%, 78.4% and 89.2% of the patients, respectively. In the pulmonary CE samples sera WB test enabled the detection of antibodies 24, 44-46, 100, 110, 116 and 120 124 kDa in molecular weight in 81.3%, 75.0%, 87.5%, 71.9%, 84.4% and 65.6% of the patients, respectively. We indicated that the antigenic components of high molecular weight can be good candidates for differentiation of hepatic CE from pulmonary CE.

[Targeting abattoirs to control cystic echinococcosis in Algeria].

PubMed

Benchikh ElFegoun, M C; Kohil, K; L'Ollivier, C; Lleu, M; Babelhadj, B; Piarroux, M; Gharbi, M; Piarroux, R

2016-08-01

Cystic echinococcosis (CE) is an important anthropozoonotic parasitic common in Algeria. The predominant life cycle of E. granulosus is a synanthropic cycle with domestic dogs as definitive hosts and livestock animals as intermediate hosts. Slaughter activity represents a potential source for dogs to access infected offal. The aim of the present study was to determine if the contact between dogs and potentially infected offal was possible in licensed abattoirs. Eighty-one private and public abattoirs located in eastern Algeria were assessed with respect to their level of protection against the intrusion of dogs.We have demonstrated that in 42 % of these abattoirs, dogs could easily come in contact with potentially parasitized offal. The most common incorrect practices were the dumping of offal freely into the environment, the feeding of dogs with offal, and the leaving of unattended offal in an unsealed chamber. Overall, some hazardous practices remained common customs of workers, and enough abattoirs remain non-compliant that the cattledog domestic cycle of CE is unlikely to be broken. Hence, some recommended measures to interrupt parasite transmission include the following: recognition of the importance of abattoirs in the maintenance of canine echinococcosis, the controlled and proper disposal of offal, the abolishment of the custom of feeding dogs with infected offal and improvements in the level of health education of abattoir staff.

Ultrasonographic features of the liver with cystic echinococcosis in sheep

PubMed Central

Hussein, Hussein Awad; Elrashidy, Mohammed

2014-01-01

Objectives The present study was designed to gain information about the ultrasonographic features of livers with cystic echinococcosis, as well as to evaluate the use of ultrasonography for diagnosis of such disease in sheep. Design This was a retrospective study during the period April 2011 to March 2013. Participants A total of 22 Baladi sheep (aged three to six years) were included in this study. Based on clear hepatic ultrasonographic findings, all animals were classified into two groups: those with hepatic cysts (n=9) and without liver cysts (healthy liver, n=13). Results Biochemically, serum concentrations of γ-glutamyl transferase, aspartate aminotransferase, total bilirubin and globulins were significantly increased (P<0.01), while albumin was lowered (P<0.01) in sheep with cystic livers. Ultrasonographic findings of diseased sheep livers revealed the presence of rounded, anechoic and unilocular hydatid cysts with ellipse circumference ranged from 6–10 cm. The borders of cysts were mostly well defined. The interior of cysts contained echogenic particulate materials, septations, or fine echoes. At the 10th intercostal space, the ventral margin, size, thickness and angle of livers were higher (P<0.01), while the diameter of portal vein was lower (P<0.01) in sheep with liver cysts than control ones. Furthermore, at the 9th intercostal space, the circumference of the gall bladder was decreased in sheep with hepatic cysts (P<0.01). The sensitivity, specificity, and positive and negative predictive values of ultrasonography for diagnosis of hepatic hydatid cysts were 80 per cent and 100 per cent, and 100 per cent and 83 per cent, respectively. Conclusions Cystic echinococcosis is associated with a number of anatomical alterations in the liver tissues that can be easily recognised by ultrasound. Furthermore, ultrasonography alone or in combination with analysis of biochemical parameters reflecting liver function could be helpful for diagnosis of hepatic

Cystic echinococcosis of the pelvic bone with recurrences: a case report.

PubMed

Jain, Sunila; Chopra, Prem

2011-09-01

Hydatid cysts commonly affect the liver and the lung. However, they rarely involve bones with vertebral column. We hereby report a case of a female patient with cystic echinococcosis of the hip bone and ilium. She presented with a long history of frequent recurrences highlighting the dismal prognosis at this rare site. Resection of the hydatid cyst from the sacroiliac region was done with allograft and autograft (rib graft) with lumbosacroiliac fixation. Follow-up of the patient at 6 months showed no detectable abnormality on radiology and the patient was doing well.

Modelling the transmission dynamics of cystic echinococcosis in donkeys of different ages from Tunisia.

PubMed

Lahmar, Samia; Boufana, Belgees; Jebabli, Leila; Craig, Phillip S; Ayari, Hayet; Basti, Talha; Dhibi, Mokhtar; Torgerson, Paul R

2014-09-15

During the period from March 2006 to July 2009 a total of 2040 slaughtered donkeys were examined for cystic echinococcosis (CE). CE prevalence in donkeys was 8.48% and the infection pressure (0.0088 infections per year) and infection rate (0.0448 cysts per year) appeared to be lower than those previously reported for cattle, sheep, dromedaries and goats in Tunisia. However, the number of cysts per infection was relatively high (5.07 cysts per infection). Among the 901 collected hydatid cysts the majority were located in the liver (89.9%), 10.09% in the lungs and 4.77% were fertile (43/901). The amplification of a fragment within the mitochondrial gene coding for cytochrome c oxidase subunit 1 (cox1) revealed that donkeys were infected with both Echinococcus equinus (horse strain, G4 genotype) and Echinococcus granulosus (sheep strain, G1 genotype). E. granulosus G1 developed into fertile cysts (15,112 protoscoleces/ml) with a protoscoleces viability of 65.78%. This investigation is the first detailed epidemiological report on cystic echinococcosis infection in donkeys for any endemic region. Copyright © 2014 Elsevier B.V. All rights reserved.

Cystic echinococcosis in Central Saudi Arabia: a 5-year experience.

PubMed

Fahim, Fraz; Al Salamah, Saleh Mohammad

2007-03-01

In this series of patients we aim to describe aspects of presentation, diagnosis and management of patients suffering from Echinococcosis in an endemic region. This is an observational study done at Riyadh Medical Complex, Saudi Arabia from 1999 to 2004. All adult patients admitted with the primary or incidental diagnosis of echinococcosis were included. These patients were followed up for 6 months. Data relating to patients' demographic characteristics, mode and duration of presentation, investigations, complications and treatment offered was collected. This data was then analyzed using SPSS 11.0. 117 patients with a mean age of 40.9+/-20.7 years were admitted, male to female ratio being 1.7:1. 114 (97.4%) originated from Middle East which is an endemic area. Pain right upper quadrant (RUQ), followed by cough were the commonest symptoms. Nine patients presented with jaundice +/- choleangitis: 3 patients had intrabiliary rupture of the hydatid cyst, while 6 had extrinsic compression. Six patients had infected cyst. Fourteen patients had intrabronchial rupture diagnosed on bronchoscopy. Twenty five (21.4%) patients presented with recurrent disease. The Haemagglutination Inhibition test gave a sensitivity of 78.6. All cysts were visualized using USG, CXR and CT scan. Endocystectomy was the most frequent procedure. Post op 8 patients had biliary leakage and 3 had bronchopleural fistula a majority of whom settled conservatively while two required ERCP and one patient with bronchopleural fistula required surgery. Fifteen patients had infection related complications. During our follow up period no recurrences were recorded. All patients undergoing surgery also received medical treatment. Twenty five patients (21.4%) were unable to undergo operative treatment due to multiple reasons. They were medically treated. Cystic Echinococcosis is a disease of the middle aged. Ultrasonogram combined with a serological assay is the best diagnostic tool available for abdominal

Control programme for cystic echinococcosis in Uruguay.

PubMed

Irabedra, Pilar; Ferreira, Ciro; Sayes, Julio; Elola, Susana; Rodríguez, Miriam; Morel, Noelia; Segura, Sebastian; Santos, Estela Dos; Guisantes, Jorge A

2016-05-24

Cystic echinococcosis is a highly endemic parasitic zoonosis that is present in the Southern Cone countries of America. For several decades, various prevention and control programmes have been implemented in different countries and regions, with varying results. In Uruguay, a new control programme was implemented in 2006 that employed new strategies for canine diagnosis and treatment, dog population control, diagnosis in humans, epidemiological surveillance, and health education, including community participation. The control programme in Uruguay addresses the control and surveillance of the disease from a holistic perspective based on Primary Health Care, which has strengthened the community's participation in developing and coordinating activities in an interdisciplinary manner. Similarly, the control programme that is currently implemented is based on a risk-focused approach. The surveillance and control measures were focused on small villages and extremely poor urban areas. In this study, the strategies used and the results obtained from 2008-2013 are analysed and discussed.

Recombinant antigens for immunodiagnosis of cystic echinococcosis

PubMed Central

Li, Jun; Zhang, Wen-Bao

2004-01-01

Three cDNAs, termed EpC1, TPxEg and EgG5, were isolated by immunoscreening from an Echinococcus granulosus cDNA library. The recombinant phages exhibited strong reactivity with sera from humans with confirmed cystic echinococcosis (CE) and with sera from mice infected with E. granulosus oncospheres. The cDNAs were subcloned into a pET vector, expressed as fusion proteins tagged with GST and affinity purified against the GST tag. Of the three recombinant proteins, EpC1 achieved the highest performance for serodiagnosis of CE in Western blot analysis using a panel of clinically defined human sera to initially address the sensitivity and specificity of the molecules. The protein yielded an overall sensitivity of 92.2% and specificity of 95.6%, levels unprecedented taking into account the large panel of 896 human sera that were tested. The strategy used may also prove suitable for improved immunodiagnosis of other parasitic infections. PMID:15188015

Cystic echinococcosis in the sheep: causes of its persistence in Sardinia.

PubMed

Scala, A; Mazzette, R

2009-09-01

The authors analyse the current epidemiological situation of sheep Cystic Echinococcosis (CE) in Sardinia, where the prevalence of ovine CE is still very high (70%) and this parasitosis is also frequently found in the human population. For this reason Sardinia represents a peculiar model for CE study, because there are particular conditions that allow Echinococcus granulosus to complete its life cycle. Some social and economical factors support the persistence of sheep CE: over 3 million Sarda breed heads are extensively bred; the presence, at farm level, of a strong relationship between dog, sheep and human; the low commercial value of meat coming from adult sheep, which results in a large number of sheep being slaughtered at home or on the farm rather than in authorized slaughterhouses. Moreover Sardinia is the only Italian region where partially successful echinococcosis control plans have been applied. The authors suggest some indirect and direct initiatives, including the use of a new vaccine against the development of this metacestode in sheep, to reduce the prevalence of CE.

Primary cystic echinococcosis in the subcutaneous gluteal region - a case report.

PubMed

Steurer, Stefan; Auer, Herbert

2008-01-01

Cystic echinococcosis (hydatidosis, hydatid disease) belongs to the group of the most severe parasitic diseases of man and is caused by metacestodes (larvae) of Echinococcus granulosus. Dogs and other canid mammals are the definitive hosts harbouring the adult stages of the tape worm in their intestines. Proglottids containing eggs are passed in the faeces of the definitive host. The eggs are ingested by intermediate hosts (i.e. sheep, cattle, swine etc.), in which the metacestode stage develops. Humans are accidental hosts and are infected by ingestion of eggs upon contact with contaminated water, vegetables, fur (of the dogs) or contaminated hands, the subsequently developing metacestodes are usually localized in the liver, the lungs and many other organs. We report on a case of a 57 year old female patient of Turkish origin suffering from a multicystic gluteal lesion diagnosed by CT- and MRT scan. Surgical intervention revealed a subcutaneous hydatid cyst of 15 cm in diameter. Parasitological-serological examinations revealed a highly positive specific antibody level against E. granulosus arc 5 antigen. The surgically resected cysts could be identified as E. granulosus sheep strain (genotype G1). Additionally to the surgical treatment the patient received albendazole postoperatively. Primary subcutaneous cystic echinococcosis without involving thoracic or abdominal organs is extremely rare and it has not been reported so far. Since soft tissue tumors may resemble hydatid cysts, careful preoperative evaluation is critical for proper handling during surgery to avoid possible anaphylactic reactions or spillage of protoscoleces, particularly when patients are deriving from endemic areas.

Cystic echinococcosis due to Echinococcus equinus in a horse from southern Germany.

PubMed

Blutke, Andreas; Hamel, Dietmar; Hüttner, Marion; Gehlen, Heidrun; Romig, Thomas; Pfister, Kurt; Hermanns, Walter

2010-05-01

In Europe, cystic echinococcosis is rare in horses and is mostly diagnosed at slaughter or postmortem examination. Equine cystic echinococcosis can be caused by various Echinococcus taxa, but only Echinococcus equinus (the "horse strain") is known to produce fertile cysts. In Europe, E. equinus appears to be endemic in Great Britain, Ireland, Spain, and Italy and has sporadically been reported in Belgium and Switzerland. The present report describes the first case of a molecularly confirmed E. equinus infection in a horse foaled and raised in Germany. The 19-year-old mare was presented for examination of inappetence, emaciation, and respiratory symptoms. X-ray radiographs of the thorax showed 2 well-circumscribed tumor-like masses, each approximately 10 cm in diameter in the caudal lung field. The horse was euthanized as its condition rapidly deteriorated. Necropsy revealed 2 thick-walled hydatid cysts, each 7-8 cm in diameter in the lung. The tri-layered cyst walls consisted of an outer adventitial layer, a laminated acellular intermediate layer, and an inner germinal membrane. Grossly, the cysts contained a clear, amber liquid with hydatid sand. Light microscopy of the hydatid sand revealed free protoscoleces, intact and ruptured brood capsules, calcareous corpuscles, and debris. Samples of protoscoleces underwent molecular characterization, and the diagnosis of E. equinus was confirmed by restriction fragment length polymorphism-polymerase chain reaction and sequence analysis of the complete mitochondrial nicotinamide adenine dinucleotide dehydrogenase subunit 1 gene.

CT Scanning in Identification of Sheep Cystic Echinococcosis.

PubMed

Mao, Rui; Qi, Hongzhi; Pei, Lei; Hao, Jie; Dong, Jian; Jiang, Tao; Ainiwaer, Abudula; Shang, Ge; Xu, Lin; Shou, Xi; Zhang, Songan; Wu, Ge; Lu, Pengfei; Bao, Yongxing; Li, Haitao

2017-01-01

We aim to determine the efficiency of CT in identification of cystic echinococcosis in sheep. Fifty-three sheep with liver cysts confirmed by ultrasonography were subject to CT scan to evaluate the number, size, and type of the cysts in liver and lung, confirmed using necropsy. The correlation of numbers between liver cysts and lung cysts was calculated using Pearson analysis. Necropsy indicated a 98% consensus on size, location, number, and activity compared with CT scan. The viable cysts were 53.1% and 50.6% in the liver and lung, respectively. Among the cysts in liver, 35.5%, 9.5%, 5.7%, 10.2%, and 39.1% were Types CE1, CE2, CE3, CE4, and CE5, respectively. The cysts in the lungs, 17.4%, 26.9%, 12.1%, 11.6%, and 32.1%, were Types CE1, CE2, CE3, CE4, and CE5, respectively. A significant correlation was noticed between the number of cysts in liver and those in lung ( R = 0.770, P < 0.001). CT scan is a suitable tool in determining the size and type of cystic hydatid cysts in both liver and lung of sheep. A significant correlation was noticed between the numbers in liver and lung, indicating that lung infection was likely due to the expansion of liver cyst burden pressure.

CT Scanning in Identification of Sheep Cystic Echinococcosis

PubMed Central

Mao, Rui; Qi, Hongzhi; Pei, Lei; Hao, Jie; Dong, Jian; Jiang, Tao; Ainiwaer, Abudula; Shang, Ge; Xu, Lin; Shou, Xi; Zhang, Songan; Wu, Ge; Lu, Pengfei

2017-01-01

Objective We aim to determine the efficiency of CT in identification of cystic echinococcosis in sheep. Methods Fifty-three sheep with liver cysts confirmed by ultrasonography were subject to CT scan to evaluate the number, size, and type of the cysts in liver and lung, confirmed using necropsy. The correlation of numbers between liver cysts and lung cysts was calculated using Pearson analysis. Results Necropsy indicated a 98% consensus on size, location, number, and activity compared with CT scan. The viable cysts were 53.1% and 50.6% in the liver and lung, respectively. Among the cysts in liver, 35.5%, 9.5%, 5.7%, 10.2%, and 39.1% were Types CE1, CE2, CE3, CE4, and CE5, respectively. The cysts in the lungs, 17.4%, 26.9%, 12.1%, 11.6%, and 32.1%, were Types CE1, CE2, CE3, CE4, and CE5, respectively. A significant correlation was noticed between the number of cysts in liver and those in lung (R = 0.770, P < 0.001). Conclusions CT scan is a suitable tool in determining the size and type of cystic hydatid cysts in both liver and lung of sheep. A significant correlation was noticed between the numbers in liver and lung, indicating that lung infection was likely due to the expansion of liver cyst burden pressure. PMID:29082246

Experimental cystic echinococcosis therapy: In vitro and in vivo combined 5-fluorouracil/albendazole treatment.

PubMed

Pensel, Patricia E; Elissondo, Natalia; Gambino, Guillermo; Gamboa, Gabriela Ullio; Benoit, J P; Elissondo, María C

2017-10-15

Human cystic echinococcosis is a zoonosis caused by the larval stage of the tapeworm Echinococcus granulosus sensu lato (s. l.). Although benzimidazole compounds such as albendazole (ABZ) and mebendazole have been the cornerstone of chemotherapy for the disease, there is often no complete recovery after treatment. Hence, new strategies are required to improve treatment of human cystic echinococcosis. The goals of the current study were as follows: (i) to evaluate the in vitro efficacy of the 5-fluorouracil (5-FU) and ABZ combination against E. granulosus s. l. protoscoleces and cysts, (ii) to compare the clinical efficacy of 5-FU alone or in combination with ABZ in infected mice. The combination of 5-FU+ABZ had a stronger in vitro effect against larval stage than that did both drugs alone. Even at the lowest concentration of 5-FU+ABZ combination (1μg/ml), the reduction of the viability of protoscoleces and cysts was greater than that observed with drugs alone at 10μg/ml. The results were confirmed at the ultrastructural level by scanning electron microscopy. These data helped to justify the in vivo investigations assessing the therapeutic potential of the combination of 5-FU and ABZ suspension in CF-1 mice infected with E. granulosus sensu stricto (s. s.) metacestodes. Treatment with 5-FU (10mg/kg) or 5-FU (10mg/kg) + ABZ suspension (5mg/kg) reduced the weight of cysts recovered from mice compared with control groups. Interestingly, the effect of 5-FU given weekly for 5 consecutive weeks was comparable to that observed with ABZ suspension under a daily schedule during 30days. Co-administration of 5-FU with ABZ did not enhance the in vivo efficacy of drugs alone calculated in relation to cysts weights. However, the combination provoked greater ultrastructural alterations compared to the monotherapy. In conclusion, we demonstrated the efficacy of 5-FU either alone or co-administrated with ABZ against murine experimental cystic echinococcosis. Since 5-FU treatments

Diagnosis of cystic echinococcosis, central Peruvian Highlands.

PubMed

Gavidia, Cesar M; Gonzalez, Armando E; Zhang, Wenbao; McManus, Donald P; Lopera, Luis; Ninaquispe, Berenice; Garcia, Hector H; Rodríguez, Silvia; Verastegui, Manuela; Calderon, Carmen; Pan, William K Y; Gilman, Robert H

2008-02-01

We evaluated prevalence of cystic echinococcosis (CE) in a central Peruvian Highland district by using 4 diagnostic methods: ultrasonography for 949 persons, radiography for 829, and 2 serologic tests for 929 (2 immunoblot formats using bovine hydatid cyst fluid [IBCF] and recombinant EpC1 glutathione S-transferase [rEpC1-GST] antigens). For the IBCF and rEpC1-GST testing, prevalence of liver and pulmonary CE was 4.7% and 1.1% and seropositivity was 8.9% and 19.7%, respectively. Frequency of seropositive results for IBCF and rEpC1-GST testing was 35.7% and 16.7% (all hepatic cysts), 47.1% and 29.4% (hepatic calcifications excluded), and 22.2% and 33.3% (lung cysts), respectively. Weak immune response against lung cysts, calcified cysts, small cysts, and cysts in sites other than lung and liver might explain the poor performance of the serodiagnostic tests. We confirm that CE is highly endemic to Peru and emphasize the limited performance of available serologic assays in the field.

Cystic Echinococcosis Epidemiology in Spain Based on Hospitalization Records, 1997-2012

PubMed Central

Siles-Lucas, Mar; Aparicio, Pilar; Lopez-Velez, Rogelio; Gherasim, Alin; Garate, Teresa; Benito, Agustín

2016-01-01

Background Cystic echinococcosis (CE) is a parasitic disease caused by the tapeworm Echinococcus granulosus. Although present throughout Europe, deficiencies in the official reporting of CE result in under-reporting and misreporting of this disease, which in turn is reflected in the wrong opinion that CE is not an important health problem. By using an alternative data source, this study aimed at describing the clinical and temporal-spatial characteristics of CE hospitalizations in Spain between 1997 and 2012. Methodology/Principal Findings We performed a retrospective descriptive study using the Hospitalization Minimum Data Set (CMBD in Spanish). All CMBD’s hospital discharges with echinococcosis diagnosis placed in first diagnostic position were reviewed. Hospitalization rates were computed and clinical characteristics were described. Spatial and temporal distribution of hospital discharges was also assessed. Between 1997 and 2012, 14,010 hospitalizations with diagnosis of CE were recorded, 55% were men and 67% were aged over 45 years. Pediatric hospitalizations occurred during the whole study period. The 95.2% were discharged at home, and only 1.7% were exitus. The average cost was 8,439.11 €. The hospitalization rate per 100,000 per year showed a decreasing trend during the study period. All the autonomous communities registered discharges, even those considered as non-endemic. Maximum rates were reached by Extremadura, Castilla-Leon and Aragon. Comparison of the CMBD data and the official Compulsory Notifiable Diseases (CND) reports from 2005 to 2012 showed that official data were lower than registered hospitalization discharges. Conclusions Hospitalizations distribution was uneven by year and autonomous region. Although CE hospitalization rates have decreased considerably due to the success of control programs, it remains a public health problem due to its severity and economic impact. Therefore, it would be desirable to improve its oversight and

Long-term albendazole effectiveness for hepatic cystic echinococcosis.

PubMed

Salinas, Jorge Luis; Vildozola Gonzales, Herman; Astuvilca, Juan; Arce-Villavicencio, Yanet; Carbajal-Gonzalez, Danny; Talledo, Lety; Willig, James H

2011-12-01

Little is known about the long-term effectiveness of albendazole in the medical therapy of non-complicated hepatic cystic echinococcosis (HCE) in resource-constrained settings. We performed a retrospective review of patients starting albendazole for HCE in Lima, Peru from January 1997 to December 2007. Patients successfully recontacted underwent chart abstraction and clinical and ultrasonographic reevaluation. Descriptive statistics were used to delineate patient characteristics and treatment effectiveness at the conclusion of albendazole and after reevaluation. Patients (N = 27) were primarily female, mean age was 51. Initial treatment success at albendazole conclusion was 26% (N = 7) per patient and 37.5% (N = 24) per cyst. After 3.8 ± 2.5 years, albendazole success was 34% (N = 9) per patient and 40% (N = 24) per cyst. We found a gap in the effectiveness of albendazole HCE therapy compared with the efficacy reported in clinical trials. This underscores the need for further investigation into alternate therapeutic strategies for this neglected disease.

Long-Term Albendazole Effectiveness for Hepatic Cystic Echinococcosis

PubMed Central

Salinas, Jorge Luis; Gonzales, Herman Vildozola; Astuvilca, Juan; Arce-Villavicencio, Yanet; Carbajal-Gonzalez, Danny; Talledo, Lety; Willig, James H.

2011-01-01

Little is known about the long-term effectiveness of albendazole in the medical therapy of non-complicated hepatic cystic echinococcosis (HCE) in resource-constrained settings. We performed a retrospective review of patients starting albendazole for HCE in Lima, Peru from January 1997 to December 2007. Patients successfully recontacted underwent chart abstraction and clinical and ultrasonographic reevaluation. Descriptive statistics were used to delineate patient characteristics and treatment effectiveness at the conclusion of albendazole and after reevaluation. Patients (N = 27) were primarily female, mean age was 51. Initial treatment success at albendazole conclusion was 26% (N = 7) per patient and 37.5% (N = 24) per cyst. After 3.8 ± 2.5 years, albendazole success was 34% (N = 9) per patient and 40% (N = 24) per cyst. We found a gap in the effectiveness of albendazole HCE therapy compared with the efficacy reported in clinical trials. This underscores the need for further investigation into alternate therapeutic strategies for this neglected disease. PMID:22144447

Intra-cystic concentrations of albendazole-sulphoxide in human cystic echinococcosis: a systematic review and analysis of individual patient data.

PubMed

Lötsch, Felix; Naderer, Judith; Skuhala, Tomislava; Groger, Mirjam; Auer, Herbert; Kaczirek, Klaus; Waneck, Fredrik; Ramharter, Michael

2016-08-01

Cystic echinococcosis (CE) is a widespread zoonosis caused by the species complex Echinococcus granulosus. Albendazole (ABZ)-the first-line anthelminthic drug for medical treatment of CE-is metabolized in vivo to the active derivative ABZ-sulphoxide (ABZ-SO). Target-site ABZ-SO concentrations in the hydatid cyst mediate the anthelminthic effect in CE. Primary outcome of this systematic review of individual patient data was the intra-cystic ABZ-SO concentration stratified by cyst size, location, calcification status and use of praziquantel. Studies reporting intra-cystic ABZ-SO concentrations in humans were identified by a systematic search. A pooled analysis of individual patient data was performed to assess intra-cystic concentrations. Pharmacokinetic data of 121 individual cysts were analysed. There was no correlation between plasma and intra-cystic ABZ-SO concentrations (rho = -0.03, p = 0.76). Intra-cystic drug concentrations were also not associated with sex and treatment duration. Use of praziquantel in combination with ABZ was associated with higher plasma (median 540 vs. 240 μg/L; p = 0.04) but not intra-cystic ABZ-SO concentrations (median 220 vs. 199 μg/L; p = 0.36). Relative drug concentrations in hepatic cysts were higher than in other cysts (0.8 vs. 0.4; p = 0.05). Intra-cystic concentrations were higher in calcified than non-calcified cysts (median 897 vs. 245 μg/L; p = 0.03). There was a trend towards higher intra-cystic concentrations in smaller sized cysts (β = -17.2 μg/L/cm; 95th CI, -35.9 to 1.6; p = 0.07). This study demonstrates that mean intra-cystic drug concentrations are similar to plasma concentrations on a population level. However, in individual patients plasma concentrations are not directly predictive for intra-cystic concentrations. The use of booster drugs was not associated with higher intra-cystic ABZ-SO concentrations in this analysis.

Hepatic echinococcosis: Clinical and therapeutic aspects

PubMed Central

Nunnari, Giuseppe; Pinzone, Marilia R; Gruttadauria, Salvatore; Celesia, Benedetto M; Madeddu, Giordano; Malaguarnera, Giulia; Pavone, Piero; Cappellani, Alessandro; Cacopardo, Bruno

2012-01-01

Echinococcosis or hydatid disease (HD) is a zoonosis caused by the larval stages of taeniid cestodes belonging to the genus Echinococcus. Hepatic echinococcosis is a life-threatening disease, mainly differentiated into alveolar and cystic forms, associated with Echinoccus multilocularis (E. multilocularis) and Echinococcus granulosus (E. granulosus) infection, respectively. Cystic echinococcosis (CE) has a worldwide distribution, while hepatic alveolar echinococcosis (AE) is endemic in the Northern hemisphere, including North America and several Asian and European countries, like France, Germany and Austria. E. granulosus young cysts are spherical, unilocular vesicles, consisting of an internal germinal layer and an outer acellular layer. Cyst expansion is associated with a host immune reaction and the subsequent development of a fibrous layer, called the pericyst; old cysts typically present internal septations and daughter cysts. E. multilocularis has a tumor-like, infiltrative behavior, which is responsible for tissue destruction and finally for liver failure. The liver is the main site of HD involvement, for both alveolar and cystic hydatidosis. HD is usually asymptomatic for a long period of time, because cyst growth is commonly slow; the most frequent symptoms are fatigue and abdominal pain. Patients may also present jaundice, hepatomegaly or anaphylaxis, due to cyst leakage or rupture. HD diagnosis is usually accomplished with the combined use of ultrasonography and immunodiagnosis; furthermore, the improvement of surgical techniques, the introduction of minimally invasive treatments [such as puncture, aspiration, injection, re-aspiration (PAIR)] and more effective drugs (such as benzoimidazoles) have deeply changed life expectancy and quality of life of patients with HD. The aim of this article is to provide an up-to-date review of biological, diagnostic, clinical and therapeutic aspects of hepatic echinococcosis. PMID:22509076

Serological evidence for human cystic echinococcosis in Slovenia.

PubMed

Logar, Jernej; Soba, Barbara; Kotar, Tadeja

2008-05-09

Cystic echinococcosis (CE) is caused by the larva of tapeworm Echinococcus granulosus. Dogs and other canids are the primary definitive hosts for this parasite. CE may develop after accidental ingestion of tapeworm eggs, excreted with the feces of these animals. In the intestine, the larvae released from the eggs are nested in the liver, lungs or other organs of livestock as intermediate hosts and humans as aberrant hosts. The aim of this study was to examine serologically whether some of the patients in Slovenia, suspected of CE by imaging findings in the liver or lungs had been infected with the larva of Echinococcus granulosus. Between January 1, 2002 and the end of December 2006, 1323 patients suspected of having echinococcosis were screened serologically by indirect haemagglutination assay (IHA). For confirmation and differentiation of Echinococcus spp. infection, the sera of IHA-positive patients were then retested by western blot (WB). Out of 127 IHA-positive sera, 34 sera were confirmed by WB and considered specific for CE. Of 34 sera of CE-positive patients sera, 32 corresponded to the characteristic imaging findings of a liver cysts and 2 to those of lung cysts. The mean age of CE-positive patients was 58.3 years. No significant differences were found between the CE-positive patients in regard to their sex. In the study, it was found out that CE was mostly spread in the same area of Slovenia as in the past, but its prevalence decreased from 4.8 per 105 inhabitants in the period 1956-1968 to 1.7 per 105 inhabitants in the period 2002-2006. In spite of the decreased prevalence of CE in the last years, it is suggested that clinicians and public health authorities, especially in the eastern parts of Slovenia where the most CE patients come from, should pay greater attention to this disease in the future.

Ultrasound wall-sign in pulmonary echinococcosis (new application).

PubMed

El Fortia, M; El Gatit, A; Bendaoud, M

2006-12-01

We report our experience in diagnosing pulmonary cystic echinococcosis using an ultrasound sign related to the cystic wall. 40 patients with 46 cysts, suspected of pulmonary echinococcosis, based on plain chest radiographs and clinical findings, were examined by ultrasound over a 9-year period (1996 - 2004), and followed up until discharge. We applied our long experience with echinococcal cysts utilising the wall sign (WS) to diagnose pulmonary hydatid disease. All cysts were subject to surgical removal, and postoperative histopathology was the gold standard. There were 34 (74 %) unilocular and 12 (26 %) multivesicular echinococcal cysts. In the univesicular cysts, the WS was found in 20 cases (66.7 %) while it was present in all multivesicular cysts (100 %). Following surgical removal, echinococcosis was confirmed by histopathology in all cases. We conclude that a double layered border in univesicular and double layered internal septum in multivesicular pulmonary echinococcal cysts is a reliable indicator of pulmonary echinococcosis, with a specificity of 66 % and 100 %, respectively.

Epidemiological evaluations of the efficacy of slow-released praziquantel-medicated bars for dogs in the prevention and control of cystic echinococcosis in man and animals.

PubMed

Wei, Jiao; Cheng, Fu; Qun, Qu; Nurbek; Xu, Shi-Dong; Sun, Li-Feng; Han, Xin-Kui; Muhan; Han, Ling-Ling; Irixiati; Jie, Peng; Zhang, Ke-Jiu; Islayin; Chai, Jun-jie

2005-12-01

To assess the epidemiological efficacy of type SRP III slow-released praziquantel-medicated bars for dogs in the prevention and control of cystic echinococcosis in man and livestock, praziquantel-medicated bars were implanted subcutaneously in over 90% of dogs in villages in north Xinjiang, China, where cystic echinococcosis is highly endemic. After implantation, infection rate of Echinococcus granulosus in dogs, specific antibodies in children and prevalence of echinococcosis in one-year-old lambs were observed for 3 years. Coproantigen of E. granulosus was positive in 41.2% of the dogs at the start of experiment. In the second and third year after medicated-bar implantation, coproantigen was undetectable in any dogs examined, while 3.0% of dogs were positive at the end of the fourth year. The antibody positive rate in 7-year-old pupils, that was 41.2% before the experiment, declined gradually and it was 5.4% in the fourth year, while children in the non-intervention control area showed 30.6% seropositivity. The prevalence of hydatid disease in children aged 7-16 years also declined significantly. The prevalence of hydatidosis in lambs one year of age was 44.8% in the first year, dropping to 10.7% in the fourth year, while in the non-intervention control area the level of infection was 46.4%. These results demonstrated not only that the slow released praziquantel-medicated bars efficiently blocked reinfection in dogs at least for 2 years, but also the measure was effective in preventing transmission of cystic echinococcosis to both man and livestock.

Cystic Echinococcosis in a Single Tertiary Care Center in Rome, Italy

PubMed Central

Cuzzi, Gilda; Ettorre, Giuseppe Maria; Busi-Rizzi, Elisa; Schininà, Vincenzo; Pucillo, Leopoldo; Pane, Stefania; Bordi, Eugenio; Pozio, Edoardo; Corpolongo, Angela; Teggi, Antonella; Brunetti, Enrico

2013-01-01

Background. Cystic echinococcosis (CE) is a chronic, clinically complex, and neglected disease. Its prevalence in Italy, a country of medium to high endemicity, remains poorly defined, as notification has long ceased to be mandatory. Methods. We set up a retrospective cohort study involving all CE patients followed at our institute between January 2005 and December 2012. Demographical and clinical features were recorded and analyzed. Results. CE was found in 28 patients (64.3%), mostly Italians from the central regions (50%), followed by subjects from the islands (33.3%) and Southern Italy (16.7%). Their median age was 45 years (IQR: 38.5–66.5), with Eastern Europeans being significantly younger (28 years, IQR: 19–39) than other patients (P ≤ 0.0001). A total of 149 cysts, mostly with hepatic localization (96%), were described. Based on the WHO classification, the cysts were mainly small (80.5%) and active (CE1 (73.8%); CE2 (7.4%)). Active cysts were more common in Eastern Europeans (85.7%) than Italians (66.7%). Conclusion. Our data confirm CE occurrence in Italy. We emphasize the importance to have a national CE registry, opportunely recently introduced. This is essential to assess CE prevalence in this country, implement appropriate control measures, and improve patient management. PMID:24151631

Evaluation of Patients with Cystic Echinococcosis.

PubMed

Türkoğlu, Emine; Demirtürk, Neşe; Tünay, Havva; Akıcı, Murat; Öz, Gürhan; Baskin Embleton, Didem

2017-03-01

Cystic echinococcosis (CE) is a globally prevalent zoonotic disease. Demographic, clinical, laboratory, and follow-up data of patients between May 2009 and 2015 were retrospectively analyzed by screening data from a hospital automation system. A total of 238 (females, n=139 and males, n=99) patients with a mean age of 40.6±20.58 years were included. Less than half (40.8%) of the patients were living in the countryside. Hepatic involvement of CE was most frequently (72.2%) seen. A majority (75.6%) of the patients were symptomatic, but abdominal pain was the most frequently seen symptom. For diagnosis, in all patients, imaging modalities were used, while in 66% of the patients, serological methods were also employed. The patients received both medical and surgical treatments (78.5%, n=187), only surgical treatment (10.5%, n=25), or only medical treatment (8.8%, n=21). Surgical treatment was performed for patients with hepatic (n=139/176, 80.6%), pulmonary (n=78/94, 82.9%), splenic (n=7/9; 77.7%), and mesenteric (n=6/7, 85.1%) cysts, and patients cases with brain, bone, muscle, omentum, bladder, and adrenal cysts had undergone surgical intervention. Publication of regional data is important in terms of epidemiological considerations and may aid in the formulation of standard treatment approaches.

Prevalence of cystic echinococcosis in Iran: a systematic review and meta-analysis.

PubMed

Khalkhali, H R; Foroutan, M; Khademvatan, S; Majidiani, H; Aryamand, S; Khezri, P; Aminpour, A

2018-05-01

As a significant zoonosis, cystic echinococcosis (CE) is endemic in some parts of the world, such as the Middle East. There are studies on the prevalence of this infection in animal and human reservoirs in Iran; hence, we conducted this meta-analysis to elucidate the prevalence of CE in Iran. English (PubMed, Scopus, Web of Science, Science Direct and Google Scholar) and Persian (Magiran, Iran Medex, Iran Doc and SID) databases were explored. In the case of definitive, animal and human intermediate hosts, 37, 90 and 33 studies, respectively, have been included in the current review from January 1990 to December 2015. According to outcomes of the heterogeneity test, either Der Simonian and Laird's random-effects method or Mantel-Haenszel's fixed-effects method were employed to pool the estimations. The pooled prevalence of Echinococcus granulosus infection in definitive hosts was calculated as 23.6% (95% confidence interval (CI) = 17.6-30.1%). The weighted prevalence of animal and human hydatidosis was calculated as 15.6% (95% CI = 14.2-17.1%) and 4.2% (95% CI = 3.0-5.5%), respectively. Meanwhile, most cases of human hydatidosis were in southern Iran, with a prevalence of 5.8% (3.2-9.2%). In terms of human hydatidosis, more infections were found in rural regions, and mostly in female individuals. Egger's regression test revealed publication bias, with a remarkable impact on total prevalence of the infection in animal intermediate hosts (P < 0.001), while it was not significant in human hosts (P = 0.4) and definitive hosts (P = 0.3). According to the weighted estimated prevalence of cystic echinococcosis and its financial burden, implementing appropriate control programmes should be compulsory to decrease the burden of the disease in Iran.

Preliminary assessment of the diagnostic performances of a new rapid diagnostic test for the serodiagnosis of human cystic echinococcosis.

PubMed

Vola, Ambra; Tamarozzi, Francesca; Noordin, Rahmah; Yunus, Muhammad Hafiznur; Khanbabaie, Sam; De Silvestri, Annalisa; Brunetti, Enrico; Mariconti, Mara

2018-04-14

Rapid diagnostic tests for cystic echinococcosis (CE) are convenient to support ultrasound diagnosis in uncertain cases, especially in resource-limited settings. We found comparable diagnostic performances of the experimental Hyd Rapid Test and the commercial VIRapid HYDATIDOSIS Test, used in our diagnostic laboratory, using samples from well-characterized hepatic CE cases. Copyright © 2018 Elsevier Inc. All rights reserved.

Demographics of patients with surgical and nonsurgical cystic echinococcosis in East Azerbaijan from 2001 to 2012.

PubMed

Vahedi, Md Amir; Vahedi, Md Leila

2012-02-15

Echinococcosis is very frequent helminthic infection in human being. The disease is endemic in Iran and particularly in its northwestern region, East Azerbaijan. The objective of this study was to study demographics of patients with cystic echinococcosis in East Azerbaijan; as well as to repot the sites of involvements. In this cross-sectional study, 318 patients with surgical and nonsurgical cystic echinococcosis were recruited from three referral centers in Tabriz within a 10-year period of time. The patients' demographics (age and gender) and the site of infection were investigated. Among the patients, females were predominant (57.5 vs. 42.5%). The mean age of patients was 32.59 +/- 18.47 (range: < 1-87) years and the most frequently age group was 20-30 years (23.6%), followed by the age groups of 30-40 years (21.7%), 10-20 years (14.2%), 40-50 years (11.9%), 50-60 years (11.3%), < 1 year (6%), 60-70 years (4.7%), 1-10 years (4.1%) and finally, > 70 years (2.5%). The lung was the most frequently infected organ (48.1%), followed by the liver (28.9%), gallbladder (3.5%), spleen and kidney (each one in 2.2%), abdomen, intestine, pleura, bronchus and mediastinum (each one in 1.6%), chest wall and heart (each one in 1.3%), brain, pericardium, facial sinus, pancreas and esophagus (each one in 0.6%) and the ovary, uterus, axillary region, pelvis and femur (each one in 0.3%). In conclusion, this study showed that the patients with hydatid cysts in East Azerbaijan Province are mainly females in the age group of 20-40 years. The lung and liver are the major sites of infection in these patients.

Cystic echinococcosis in South America: systematic review of species and genotypes of Echinococcus granulosus sensu lato in humans and natural domestic hosts.

PubMed

Cucher, Marcela Alejandra; Macchiaroli, Natalia; Baldi, Germán; Camicia, Federico; Prada, Laura; Maldonado, Lucas; Avila, Héctor Gabriel; Fox, Adolfo; Gutiérrez, Ariana; Negro, Perla; López, Raúl; Jensen, Oscar; Rosenzvit, Mara; Kamenetzky, Laura

2016-02-01

To systematically review publications on Echinococcus granulosus sensu lato species/genotypes reported in domestic intermediate and definitive hosts in South America and in human cases worldwide, taking into account those articles where DNA sequencing was performed; and to analyse the density of each type of livestock that can act as intermediate host, and features of medical importance such as cyst organ location. Literature search in numerous databases. We included only articles where samples were genotyped by sequencing since to date it is the most accurate method to unambiguously identify all E. granulosus s. l. genotypes. Also, we report new E. granulosus s. l. samples from Argentina and Uruguay analysed by sequencing of cox1 gene. In South America, five countries have cystic echinococcosis cases for which sequencing data are available: Argentina, Brazil, Chile, Peru and Uruguay, adding up 1534 cases. E. granulosus s. s. (G1) accounts for most of the global burden of human and livestock cases. Also, E. canadensis (G6) plays a significant role in human cystic echinococcosis. Likewise, worldwide analysis of human cases showed that 72.9% are caused by E. granulosus s. s. (G1) and 12.2% and 9.6% by E. canadensis G6 and G7, respectively. E. granulosus s. s. (G1) accounts for most of the global burden followed by E. canadensis (G6 and G7) in South America and worldwide. This information should be taken into account to suit local cystic echinococcosis control and prevention programmes according to each molecular epidemiological situation. © 2015 John Wiley & Sons Ltd.

Cardiac and Pulmonary Cystic Echinococcosis with Massive Obstruction of the Pulmonary Vessel System in a 16 Year Old Girl.

PubMed

Kohlmaier, Benno; Trobisch, Andreas; Pfurtscheller, Klaus; Knez, Igor; Klepetko, Walter; Pilhatsch, Alexander; Schweintzger, Sabrina; Zenz, Werner

2018-04-09

We describe herein the management of a 16-year-old girl with cystic echinococcosis (CE) of the right ventricle and massive obstruction of the pulmonary vessel system by parasitic metastatic dissemination. After resection of the cardiac cyst, pulmonary thromboendarterectomy was performed to remove parts of the obstructive parasitic material. The treatment reduced the elevated pulmonary arterial pressure, improving the patient's overall condition.

Management and Outcome of Cardiac and Endovascular Cystic Echinococcosis

PubMed Central

Díaz-Menéndez, Marta; Pérez-Molina, José Antonio; Norman, Francesca Florence; Pérez-Ayala, Ana; Monge-Maillo, Begoña; Fuertes, Pilar Zamarrón; López-Vélez, Rogelio

2012-01-01

Background Cystic echinococcosis (CE) can affect the heart and the vena cava but few cases are reported. Methods A retrospective case series of 11 patients with cardiac and/or endovascular CE, followed-up over a period of 15 years (1995–2009) is reported. Results Main clinical manifestations included thoracic pain or dyspnea, although 2 patients were asymptomatic. Cysts were located mostly in the right atrium and inferior vena cava. Nine patients were previously diagnosed with disseminated CE. Echocardiography was the diagnostic method of choice, although serology, electrocardiogram, chest X-ray, computed tomography/magnetic resonance imaging and histology aided with diagnosis and follow-up. Nine patients underwent cardiac surgery and nine received long-term antiparasitic treatment for a median duration of 25 months (range 4–93 months). One patient died intra-operatively due to cyst rupture and endovascular dissemination. Two patients died 10 and 14 years after diagnosis, due to pulmonary embolism (PE) and cardiac failure, respectively. One patient was lost to follow-up. Patients who had cardiac involvement exclusively did not have complications after surgery and were considered cured. There was only one recurrence requiring a second operation. Patients with vena cava involvement developed PEs and presented multiple complications. Conclusions Cardiovascular CE is associated with a high risk of potentially lethal complications. Clinical manifestations and complications vary according to cyst location. Isolated cardiac CE may be cured after surgery, while endovascular extracardiac involvement is associated with severe chronic complications. CE should be included in the differential diagnosis of cardiovascular disease in patients from endemic areas. PMID:22235354

Knowledge, attitudes and practices with regard to the presence, transmission, impact, and control of cystic echinococcosis in Sidi Kacem Province, Morocco.

PubMed

El Berbri, Ikhlass; Ducrotoy, Marie J; Petavy, Anne-Françoise; Fassifihri, Ouaffa; Shaw, Alexandra P; Bouslikhane, Mohammed; Boue, Franck; Welburn, Susan C; Dakkak, Allal

2015-11-09

This study is a component of a large research project on five major neglected zoonotic diseases (NZDs) including cystic echinococcosis and was undertaken in the Province of Sidi Kacem over a period of four years (April 2009-March 2013). Questionnaires were administered at community level in a total of 27 communes and visits were made to all of the 10 abattoirs situated in the Province, to collect qualitative data on determinants of transmission for disease in humans and animals. More specifically, community knowledge, attitudes and practices related to cystic echinococcosis were assessed, as well as the extent to which local customs and behaviours may promote transmission. Abattoir infrastructure and practices, and their role in perpetuating disease transmission were also critically evaluated. The results show that only 50 % of people have heard of the disease, and of those, only 21 % are aware of the dog's role in disease transmission. Sixty-seven per cent of respondents stated that dogs are fed ruminant organs deemed unfit for human consumption. Owned dogs have access to the family home, including the kitchen, in 39 % of households. The extent of this close proximity between humans and animals is even more pertinent when one considers that dogs are omnipresent in the community, with an average of 1.8 dogs owned per household. The unrestricted access of dogs to abattoirs is a huge issue, which further promotes disease transmission. This study would suggest that the high prevalence of cystic echinococcosis in humans and animals in Morocco is largely due to three factors: 1) abundance of dogs 2) engagement in risky behaviour of the local population and 3) poor abattoir infrastructure and practices. This has serious implications in terms of the socio-economic impact of the disease, especially for rural poor communities.

[Study of the pathogenesis of paecilomycosis-complicated echinococcosis].

PubMed

Abdiev, F T; Mamysheva, N O; Streliaeva, A V; Gasparian, E R; Sagieva, A T; Sadykov, R V; Lazareva, N B; Kurilov, D V; Akhmedov, Iu M; Sadykov, V M; Shcheglova, T A; Zuev, S S; Mukhitdinov, Sh M; Vakhidova, A V

2012-01-01

Experiments were carried out on 35 lambs infected with echinococcus eggs taken from canine donors, 5 lambs with echinococcosis, 5 lambs with echinococcosis + paecilomycosis, 5 lambs with coenurosis, 5 with cysticercosis ovis, 5 with alveococcosis, 5 with paecilomycosis, inoculated by the material taken from patients; and 5 healthy lambs formed a control group. Cystic hydatidosis was found to induce substantial cardiovascular changes.

Economic impact of cystic echinococcosis in peru.

PubMed

Moro, Pedro L; Budke, Christine M; Schantz, Peter M; Vasquez, Julio; Santivañez, Saul J; Villavicencio, Jaime

2011-05-01

Cystic echinococcosis (CE) constitutes an important public health problem in Peru. However, no studies have attempted to estimate the monetary and non-monetary impact of CE in Peruvian society. We used official and published sources of epidemiological and economic information to estimate direct and indirect costs associated with livestock production losses and human disease in addition to surgical CE-associated disability adjusted life years (DALYs) lost. The total estimated cost of human CE in Peru was U.S.$2,420,348 (95% CI:1,118,384-4,812,722) per year. Total estimated livestock-associated costs due to CE ranged from U.S.$196,681 (95% CI:141,641-251,629) if only direct losses (i.e., cattle and sheep liver destruction) were taken into consideration to U.S.$3,846,754 (95% CI:2,676,181-4,911,383) if additional production losses (liver condemnation, decreased carcass weight, wool losses, decreased milk production) were accounted for. An estimated 1,139 (95% CI: 861-1,489) DALYs were also lost due to surgical cases of CE. This preliminary and conservative assessment of the socio-economic impact of CE on Peru, which is based largely on official sources of information, very likely underestimates the true extent of the problem. Nevertheless, these estimates illustrate the negative economic impact of CE in Peru.

Spinal Cystic Echinococcosis – A Systematic Analysis and Review of the Literature: Part 1. Epidemiology and Anatomy

PubMed Central

Neumayr, Andreas; Tamarozzi, Francesca; Goblirsch, Sam; Blum, Johannes; Brunetti, Enrico

2013-01-01

Bone involvement in human cystic echinococcosis (CE) is rare, but affects the spine in approximately 50% of cases. Despite significant advances in diagnostic imaging techniques as well as surgical and medical treatment of spinal CE, our basic understanding of the parasite's predilection for the spine remains incomplete. To fill this gap, we systematically reviewed the published literature of the last five decades to summarize and analyze the currently existing data on epidemiological and anatomical aspects of spinal CE. PMID:24086783

Cystic echinococcosis in Southern Israel.

PubMed

Ben-Shimol, Shalom; Sagi, Orli; Houri, Ohad; Bazarsky, Elina; Berkowitz, Anat; Bulkowstein, Shlomi; Barrett, Chiya; Greenberg, David

2016-01-01

The aim of this retrospective, population-based study was to characterize demographically and clinically cystic-echinococcosis (CE) in southern Israel, between 2005 and 2012. Newly-diagnosed (nd-CE) and past-diagnosed (pd-CE, diagnosed before the study) cases were defined. Two populations live in southern-Israel, receiving medical treatment at a single hospital: the Jewish and the Bedouin populations (resembling resource-rich and resource-poor populations, respectively). 126 CE cases were identified; 55 nd-CE and 71 pd-CE. Mean annual nd-CE incidence per 100,000 in the Bedouin and Jewish populations were 2.7 ± 1.2 and 0.4 ± 0.3, respectively (P<0.001). None of the Bedouin and 86.5% of the Jewish patients were born outside Israel. Liver and lung involvement were recorded in 85.7% and 15.1% of overall-CE, respectively. Abdominal pain, cough, fever, eosinophilia and asymptomatic disease were documented in 63.6%, 32.7%, 27.3%, 41.5% and 12.7% of nd-CE, respectively. Serology sensitivity for first test and any positive test were 67.3% and 83.3%, respectively. Computed tomography, ultrasonography and X-ray diagnosis were documented in 79.2%, 58.4% and 17.0% of overall-CE, respectively, with ultrasonography mainly used in liver-CE and X-ray in lung-CE. Treatment included surgery and albendazole in 50.0% and 55.3% of CE, respectively. We conclude that CE is endemic in southern-Israel among the Bedouin population, while disease is probably mainly imported in the Jewish population. Liver involvement and eosinophilia rates were high compared with those of other endemic regions, possibly due to differences in the timing of diagnosis. These findings may help developing treatment and prevention strategies.

Ruptured Pulmonary Cystic Echinococcosis Mimicking Tuberculosis in Childhood: A Case Report.

PubMed

Ünver Korğalı, Elif; Kaymak Cihan, Meriç; Ceylan, Özgür; Kaptanoğlu, Melih

2017-06-01

Cystic echinococcosis (CE) is a zoonotic disease; in places such as Turkey where livestock is common, it is an endemic health concern. The most commonly involved organ is the lungs in children. Pulmonary cysts can be asymptomatic; in some cases, they may rupture and become symptomatic. Ruptured lung hydatid cysts may often be confused with tuberculosis (Tbc) radiologically and clinically. . In this report, we present an 8-year-old female patient admitted with cough, fever, and sputum persisting since 2 weeks; her chest radiography and computed tomography (CT) findings initially indicated Tbc, but the follow-up surgery led to a diagnosis of ruptured lung CE. We want to emphasize that in children belonging to places where livestock is common, if respiratory symptoms are observed, CE and tuberculosis must be considered in the differential diagnosis, and the final diagnosis should be supported by other microbiological-serological tests.

A correlative study of ultrasound with serology in an area in China co-endemic for human alveolar and cystic echinococcosis.

PubMed

Yang, Y R; Craig, P S; Ito, A; Vuitton, D A; Giraudoux, P; Sun, T; Williams, G M; Huang, Z; Li, Z; Wang, Y; Teng, J; Li, Y; Huang, L; Wen, H; Jones, M K; McManus, D P

2007-05-01

We correlated ultrasound (US) imaging classifications for human alveolar echinococcosis (AE) and cystic echinococcosis (CE) with serology (ELISA and immunoblotting (IB) incorporating native and recombinant/purified echinococcal antigens) in community surveys (2001-2003) and follow-up (2002 and 2003) of US-confirmed cases in Ningxia, China. One hundred and seventy-one cases (96 with AE, 75 with CE) were identified; of these, US classification and serological data were obtained for 142 and 112 cases, respectively. Seropositive-rates increased in CE patients with highly viable unilocular cyst lesions (Types CL, CE 1 or CE 2) to degenerating primary lesions (CE 3), but then decreased in subjects with inactive (CE 4) or dead (CE 5) cysts. In contrast, there was a constant increase in seropositivity from the early (P1, P2) to the advanced stages (P3, P4) with AE cases. For US-confirmed cases, follow-up by US combined with serology is invaluable for studying the clinical progression of echinococcosis and for detecting recurrent cysts or reinfection post-treatment.

[Cystic echinococcosis in children in Tunisia: fertility and case distribution of hydatid cysts].

PubMed

Oudni-M'Rad, M; M'Rad, S; Gorcii, M; Mekki, M; Belguith, M; Harrabi, I; Nouri, A; Azaiez, R; Mezhoud, H; Babba, H

2007-02-01

Cystic echinococcosis, which commonly starts during childhood or adolescence, is a serious problem of public health in Tunisia. For 121 children (161 cysts), the localization and fertility of cysts as well as viability of their protoscoleces were determined. Results indicated that the lung was the primary localization of cyst (59%) followed by the liver (35%). Children's infection is more frequent in male than in female (sex ratio 1.96) and the greatest number of cases is observed in the 4-9 year age groups (94 cases). The fertility of the cyst was independent of its site or its size and no incidence of age of children was detected. Nevertheless, the fertility rate is higher in females than in males for the liver localization.

Knowledge, Awareness and Practices Regarding Cystic Echinococcosis among Livestock Farmers in Basrah Province, Iraq

PubMed Central

Abdulhameed, Mohanad F.; Al-Azizz, Suzan A.

2018-01-01

Cystic echinococcosis (CE) is an endemic neglected parasitic zoonosis in many of the countries of the Middle East. The disease poses a remarkable economic burden for both animals and humans. In this study, we conducted a questionnaire survey among livestock farmers in Basrah province, southern Iraq, in order to evaluate their knowledge and awareness about CE, and to understand some of the risky practices that could contribute to spread and persistence of such disease. Of the interviewed participants (N = 314), 27.4% owned dogs on their farms. Among farmers owning dogs, 76.7% (66/86) never tied up their dogs, and 43% (37/86) indicated feeding uncooked animal viscera to their dogs. The majority (96.5%) of the farmers indicated that they did not de-worm their dogs at all. Only 9.8% (31/314) of the respondents indicated eating raw leafy vegetables without washing. Added to that, 32% of the interviewees indicated that they source water for domestic use from a river; meanwhile 94.3% (296/314) of them do not boil water before using it for domestic purposes. Half of the interviewed livestock farmers in Basrah were not aware about how humans get infected with CE disease, and 41.4% (130/314) did not even realize that CE is a dangerous disease to human health. Almost one in three of the respondents who owned dogs on their farms viewed de-worming of their dogs as a low priority practice. This study highlights the gap in knowledge and awareness about CE among the study population. Risky practices associated with dog keeping management and food and water handling practices were identified. The insight from this research could be used to improve the delivery of a health education message relevant to cystic echinococcosis control at the human-animal interface in Iraq. PMID:29415425

Performance of polymerase chain reaction for the diagnosis of cystic echinococcosis using serum, urine, and cyst fluid samples

PubMed Central

Chaya, DR; Parija, Subhash Chandra

2014-01-01

Introduction: Cystic echinococcosis (CE) is a chronic zoonosis which presents with variable clinical manifestations. Currently the diagnosis of this disease is based on radiological findings and serological tests which lack specificity. Although antigen detection from the cyst fluid is the most specific, it is seldom done due to the complications involved. Detecting the presence of Echinococcus granulosus specific deoxyribonucleic acid (DNA) by the polymerase chain reaction (PCR) could provide a definitive diagnosis of CE. Materials and Methods: An in-house PCR assay was devised to detect E. granulosus specific DNA in serum, urine and hydatid cyst fluid. The ability of the PCR to detect E. granulosus in the above mentioned samples were observed in comparison with other antigen and antibody detection tests. Results: Serum samples from surgically confirmed patients of CE with ruptured cysts contained the corresponding DNA while the in the majority of cases who had an intact cyst had no DNA of E. granulosus in their serum. DNA of E. granulosus was not found to be excreted in urine. PCR performed equal to antigen detection ELISA while testing hydatid cyst fluid samples. Conclusions: Serum and urine might not serve as useful samples for the molecular diagnosis of cystic echinococcosis. However, PCR can be useful on serum samples to detect ruptured hydatid cysts and on hydatid cyst fluid to confirm the parasitic diagnosis. PMID:24754027

Performance of polymerase chain reaction for the diagnosis of cystic echinococcosis using serum, urine, and cyst fluid samples.

PubMed

Chaya, Dr; Parija, Subhash Chandra

2014-01-01

Cystic echinococcosis (CE) is a chronic zoonosis which presents with variable clinical manifestations. Currently the diagnosis of this disease is based on radiological findings and serological tests which lack specificity. Although antigen detection from the cyst fluid is the most specific, it is seldom done due to the complications involved. Detecting the presence of Echinococcus granulosus specific deoxyribonucleic acid (DNA) by the polymerase chain reaction (PCR) could provide a definitive diagnosis of CE. An in-house PCR assay was devised to detect E. granulosus specific DNA in serum, urine and hydatid cyst fluid. The ability of the PCR to detect E. granulosus in the above mentioned samples were observed in comparison with other antigen and antibody detection tests. Serum samples from surgically confirmed patients of CE with ruptured cysts contained the corresponding DNA while the in the majority of cases who had an intact cyst had no DNA of E. granulosus in their serum. DNA of E. granulosus was not found to be excreted in urine. PCR performed equal to antigen detection ELISA while testing hydatid cyst fluid samples. Serum and urine might not serve as useful samples for the molecular diagnosis of cystic echinococcosis. However, PCR can be useful on serum samples to detect ruptured hydatid cysts and on hydatid cyst fluid to confirm the parasitic diagnosis.

Human cystic echinococcosis in Turkey: a preliminary study on DNA polymorphisms of hydatid cysts removed from confirmed patients.

PubMed

Orsten, Serra; Boufana, Belgees; Ciftci, Turkmen; Akinci, Devrim; Karaagaoglu, Ergun; Ozkuyumcu, Cumhur; Casulli, Adriano; Akhan, Okan

2018-04-01

Cystic echinococcosis caused by the larval stages of Echinococcus granulosus sensu lato s.l is endemic in Turkey with a high public health impact particularly in rural areas. The aim of this study was to investigate the genetic variation and population structure of E. granulosus s.s using metacestode isolates removed from surgically confirmed patients originating from several regions in Turkey and to investigate the occurrence of autochthonous transmission. Using DNA extracted from a total of 46 human-derived CE isolates, we successfully analysed an 827-bp fragment within the cox1 mitochondrial gene and confirmed the causative agent of human cystic echinococcosis in patients included in this study to be Echinococcus granulosus s.s (G1 and G3 genotypes). The haplotype parsimony network consisted of 28 haplotypes arranged within three main clusters and the neutrality indices were both negative and significant indicating negative selection or population expansion. The assessment carried out in this study using GenBank nucleotide sequence data from Turkey for sheep and cattle hosts demonstrated the importance of autochthonous transmission with sheep, cattle and humans harbouring the same haplotypes. Further studies are required to investigate the biological significance, if any, of E. granulosus s.s haplotypes and the genetic variability of CE from human patients using longer nucleotide sequences and a larger sample set.

Clinical characterization of unusual cystic echinococcosis in southern part of Turkey.

PubMed

Akcam, Atlgan Tolga; Ulku, Abdullah; Koltas, Ismail Soner; Izol, Volkan; Bicer, Omer Sunkar; Kilicbagir, Emine; Sakman, Gurhan; Poyrazoglu, Hakan; Erman, Tahsin; Aridogan, Ibrahim Atilla; Parsak, Cem Kaan; Inal, Mehmet; Iskit, Serdar

2014-01-01

The incidence of primary extrahepatic cystic echinococcosis (CE) is rare. Generally, radiological and serological findings can help establish the diagnosis of hepatic and pulmonary CE, but a CE in an unusual location with atypical radiological findings may complicate the differential diagnosis. The objective of this study is to present the characteristics of cases with extrahepatic CE in respect of sites of involvement, clinical presentations, radiological findings, serological diagnostic evaluations, and outcomes of infected patients. A retrospective analysis of surgically treated CE was conducted between January 1993 and January 2014 in the General Surgery, Pediatric Surgery, Urology, Cardiovascular Surgery, Neurosurgery, and Orthopedics departments of University of Cukurova, Faculty of Medicine, Balcal Hospital. Among the 661 patients managed for CE, 134 had unusual sites of involvement. Radiological and serological examinations were used to differentiate CE from alveolar echinococcosis. Of 134 cases with unusual sites of involvement, 32 cases had liver CE (23.9%), 7 cases had lung CE (5.2%), and 2 cases had concomitant liver and lung CE (1.5%). In 93 (69.4%) cases, unusual organ involvement was isolated without any liver or lung involvement. The mean age was 45 years. Abdominal pain was the main symptom and was found in 104 patients. Thirty-one (23.1%) of 134 extrahepatic CE cases were evaluated as negative with indirect hemagglutination (IHA). However, positive results were obtained in 54 cases evaluated with Echinococcus granulosus IgG Western blot (WB), including 10 IHA-negative cases. CE with unusual localizations may cause serious problems of diagnostic confusion. The combination of clinical history, radiological findings, and serological test results (especially the WB) are valuable in diagnosing extrahepatic CE.

Laparoscopic approach for total cystectomy in treating hepatic cystic echinococcosis

PubMed Central

Li, Haitao; Shao, Yingmei; Aji, Tuerganaili; Zhang, Jinhui; Kashif, Kafayat; Ma, Qinglong; Ran, Bo; Wen, Hao

2014-01-01

Background: The laparoscopic approach has been proposed for treating hepatic cystic echinococcosis (HCE) and has already been used in clinical practice, mostly for non-radical operations. In this study, we aimed to evaluate the feasibility of total cystectomy of HCE under laparoscopy (LS). Results: A retrospective review of the medical records obtained from 22 patients diagnosed with HCE between June 2009 and June 2013 and treated with an LS approach was conducted in the First Affiliated Hospital of Xinjiang Medical University. A total of 15 patients underwent total cystectomy of HCE using LS. The average time of surgery was 174 min (160–210 min). Intraoperative bleeding was 103 mL (80–200 mL). The mean duration of hospitalization was 7 days (6–15 days). Seven patients were transferred to open surgery (OS). For these patients, the average duration of surgery was 177 min (150–230 min). Intraoperative bleeding was 237 mL (160–350 mL), and the mean duration of hospitalization was 10 days (8–15 days). The most frequent postoperative complications were hydrops in the surgical area (two cases in LS and three cases in OS), and temporary bile leakage (one patient in the LS group). Recurrence was not seen in any cases in either group with a follow-up of 6–12 months. Conclusions: Total cystectomy of HCE appears to be safe and effective in selected patients with unique, small-sized, superficially located cysts. To establish precise recommendations about the technique and its indications, prospective studies are necessary. PMID:25489977

Importance of cystic echinococcosis in slaughtered herbivores from Iran.

PubMed

Ezatpour, Behrouz; Farhadi, Seid Jafar; Azami, Mehdi; Alirezaei, Masoud; Ebrahimzadeh, Farzad

2015-06-01

Cystic echinococcosis (CE) or hydatidosis is well-known as one of the zoonotic diseases in world-wide including Iran. Hydatidosis was considered as a disease that causes severe reduction in meat wool and milk in livestock animals which all of them indicating its importance. Thus, present study was designed to evaluated prevalence of CE in slaughtered animals from Delfan region in Lorestan province of Iran. The samples 6,885 animals were considered based on type of species in a slaughter from Lorestan province. The study performed from 3 April 2009 to 3 April 2012 and inspection carried out from 4,101 cattle, 2,150 sheep and 634 goat. The liver and lungs examined based on CE and showed the highest prevalence in cattle (25.7 %) and the lowest 3.8 % in goat, likewise, CE was more in the lung than to liver. There was significant difference between species of animals and infected organ (P < 0.001). The highest prevalence was seen in winter (32.8 % for cattle, 8.1 % for goat) while, it was 15.7 % for sheep in summer (P = 0.04). Overall these data indicate the necessity of disease control strategy for reduction of CE.

Echinococcosis: Control and Prevention.

PubMed

Craig, P S; Hegglin, D; Lightowlers, M W; Torgerson, P R; Wang, Q

2017-01-01

Human cystic echinococcosis (CE) has been eliminated or significantly reduced as a public health problem in several previously highly endemic regions. This has been achieved by the long-term application of prevention and control measures primarily targeted to deworming dogs, health education, meat inspection, and effective surveillance in livestock and human populations. Human CE, however, remains a serious neglected zoonotic disease in many resource-poor pastoral regions. The incidence of human alveolar echinococcosis (AE) has increased in continental Europe and is a major public health problem in parts of Eurasia. Better understanding of wildlife ecology for fox and small mammal hosts has enabled targeted anthelmintic baiting of fox populations and development of spatially explicit models to predict population dynamics for key intermediate host species and human AE risk in endemic landscapes. Challenges that remain for echinococcosis control include effective intervention in resource-poor communities, better availability of surveillance tools, optimal application of livestock vaccination, and management and ecology of dog and wildlife host populations. Copyright © 2017 Elsevier Ltd. All rights reserved.

Cystic echinococcosis in cattle slaughtered at Shashemanne Municipal Abattoir, south central Oromia, Ethiopia: prevalence, cyst distribution and fertility.

PubMed

Negash, Kedir; Beyene, Desta; Kumsa, Bersissa

2013-04-01

Cystic echinococcosis is one of the most widespread zoonoses, causing morbidity and mortality in humans and huge economic losses in livestock. It is caused by metacestodes of the tapeworm Echinococcus granulosus. The metacestodes cause hydatid cysts in the lungs, liver and other organs of intermediate hosts. A study was made from December 2010 through March 2011 to determine the prevalence, organ distribution and characteristics of hydatid cysts in cattle slaughtered at Shashemanne Municipal Abattoir in Oromia, Ethiopia. Antemortem examination of 384 cattle was followed by standard postmortem inspection of their internal organs including lungs, liver, kidneys, spleen and heart for the presence of hydatid cysts. The overall prevalence of hydatid cysts recorded in cattle slaughtered at Shashemanne Municipal Abattoir was 49.5% (190/384). Hydatid cyst prevalence was significantly higher in cattle more than 7 years old compared with those aged 7 years or less, in male cattle compared with female cattle (51.9% vs 31.9%), and in cattle with a body condition score of lean or medium rather than fat (54.05% and 83.2% vs 22.9%). The greatest proportions of cysts were recorded in the lungs (71.6%) and liver (24.1%). Lungs and liver were more commonly infected (95.5%) than other organs. Of the cysts recorded, 15.9% were fertile, 71.7% sterile and 12.2% calcified. The percentage of fertile cysts in the lungs was higher than that in any other organ. Our study showed widespread occurrence of cystic echinococcosis in cattle, which may have a role in the lifecycle of this serious zoonosis.

Cystic echinococcosis in Romania: the pediatric approach.

PubMed

Neghina, Raul; Neghina, Adriana M; Marincu, Iosif; Iacobiciu, Ioan

2011-08-01

Cystic echinococcosis (CE) has represented a leading parasitic disease in Romania since 1862 when the first case was detected. Following the extremely high number of human and animal cases registered in 1995, Romania was framed in the forefront of Europe and among the first countries worldwide. This review aims to overview and analyze important data concerning CE in Romania from a pediatric point of view. To fulfill these goals, we focused on information provided by major epidemiological studies and rare/unusual case presentations. CE represents a serious concern for both public health services and the society, mainly because of prolonged hospitalization and convalescence periods. Moreover, the situation is much more problematic in children because physical, psychical, or intellectual impairments may impact negatively on their developmental and educational progress. The management of the disease consumes considerable healthcare resources estimated at about 1500-2000 United States Dollar (USD) for each uncomplicated case and approximately double for the complicated ones. A systematic analysis of data framed important epidemiological characteristics that may be useful when assessing individuals at risk: children (0-19 years old) represented 22.1% of the total number of cases, patients aged 13 years were most frequently affected (p < 0.0001), male patients and rural inhabitants predominated (p = 0.01 and p < 0.0001, respectively), and pulmonary involvement was detected most frequently (p < 0.0001). Considering the immense burden of this disease in Romania, emphasis should be given to the wide-scale implementation of effective prophylactic measures toward its eradication. Among them, education of the population plays an important role and should begin in early childhood.

[Knowledge and practices regarding cystic echinococcosis and trichinellosis in users and companions of users of the Hospital Comunitario de Salud Familiar El Carmen, Biobio Region, Chile].

PubMed

Lisboa-Navarro, Raúl; González, Jorge; Junod, Tania; Melín-Coloma, Millaray; Landaeta-Aqueveque, Carlos

2016-08-01

We surveyed users of the Hospital Comunitario de Salud Familiar El Carmen and their companions to analyze knowledge and practices regarding cystic echinococcosis and trichinellosis. Most people recognized risky practices. Previous attendance at talks and working in agricultural-livestock enhanced the knowledge. Age, sex and formal education were not associated with the knowledge. Knowledge was not associated with practices.

[Rapid diagnostic tests for the serodiagnosis of human cystic echinococcosis].

PubMed

Tamarozzi, F; Mariconti, M; Covini, I; Brunetti, E

2017-02-01

Cystic echinococcosis (CE) is a parasitic zoonosis especially affecting resource-poor populations in livestock raising areas. Imaging, in particular ultrasound (US), is crucial for the diagnosis, staging, and clinical management of abdominal CE in humans. Serology is a valuable complement to imaging, especially when ultrasound features of CE are absent or unclear. In rural endemic areas, where expertise in US is scant, and conventional serology techniques are unavailable due to lack of laboratory equipment, rapid diagnostic tests (RDTs) may be very useful. Several reports have described the performance of commercial and experimental RDTs in the diagnosis of CE, including a recent study by our group that compared the diagnostic performances of three commercial RDTs for the diagnosis of hepatic CE. To put RDTs for CE in context, we reviewed the available literature in English on this topic. Overall, RDTs appear to be useful in resourcepoor settings where they may replace conventional serodiagnostic tests. However, like other serodiagnostic tests, RDTs lack standardization and show unsatisfactory sensitivity and specificity. An important issue that needs to be addressed is that studies on the diagnostic performance of RDTs fail to take into account the variables known to influence results such as anatomical location and cyst stage.

Evaluation of human cystic echinococcosis before and after surgery and chemotherapy by demonstration of antibodies in serum.

PubMed

Tenguria, Rajesh Kumar; Naik, Mohd Irfan

2014-01-01

Human cystic echinococcosis (CE), caused by Echinococcus granulosus, is one of the most important and widespread parasitic zoonoses. As one of the problems that can be encountered after treating CE patients is the risk of postsurgical relapses or treatment failure, a long-term clinical and serological follow-up is required to evaluate the success and failure of therapy. Therefore, the aim of the present study was to identify the best diagnostic and prognostic ELISA markers in patients with CE. The cohort comprised 50 patients with symptomatic CE treated with antihelminthic drugs and surgery, who were followed up clinically and radiologically for a mean of 6 years (range 4-8 years). The results clearly indicate that the hydatid specific antibodies of IgE, IgG1 and IgG4 are the most important antibodies for the serological diagnosis of cystic echinococcosis during the active stage of the disease. None of the serum samples from healthy controls gave a non-specific reaction with IgE, IgG1 or IgG4, and a considerably reduced cross-reaction was observed with these antibodies. During post-operative follow-up, the IgM, IgE, IgG1, IgG2 and IgG4 antibody response provided the best correlate of disease activity. The detection of total IgG and IgG3 subclass antibody response for the assessment of post-treatment disease activity among CE patients was insensitive. All patients responded to treatment except 2 women (32 and 36 years old), in whom multiple cysts (12 and 7 cysts) were detected in the liver and lung two years after the first operation. Hence, it can be concluded that the CE-specific antibodies of IgE, IgG1 and IgG4 are the best immunological markers for diagnosis and prognosis of CE patients.

Cystic echinococcosis: late rupture and complication of a stable pulmonary cyst.

PubMed

Fisher, J; Shargall, Y; Krajden, S; Moid, F; Hoffstein, V

2011-01-01

Cystic echinococcosis is observed worldwide. Traditional management includes an invasive surgical approach with adjunctive chemotherapy. It has been suggested that observation alone may be appropriate in asymptomatic individuals with stable cysts. A case involving a 38-year-old Peruvian man with an asymptomatic bronchogenic cyst (suspected to be due to echinococcus, but never definitely diagnosed) is presented. The cyst was first noted in 1998, and was followed for 10 years during which time he remained asymptomatic with minimal radiographic change. One year later, in 2009, he presented with acute rupture of the cyst causing empyema. The patient required thoracotomy, decortication and resection of the ruptured cyst. Final pathology showed Echinococcus organisms. The patient responded well to treatment with albendazole and praziquantel, and became completely asymptomatic within six months. The present case demonstrates that echinococcal cysts may be at risk of spontaneous rupture, even after many years of clinical stability, thus supporting the case for resection of asymptomatic cysts suspected of being echinococcal at the time of diagnosis. In addition, the case illustrates that medical therapy with albendazole and praziquantel, in conjunction with surgical drainage, can be successful in the treatment of echinococcal empyema.

Performance of the commercially available SERION ELISA classic Echinococcus IgG test for the detection of cystic echinococcosis in clinical practice.

PubMed

Sarink, M J; Koelewijn, R; Slingerland, B C G C; Tielens, A G M; van Genderen, P J J; van Hellemond, J J

2018-06-28

Diagnosis of cystic echinococcosis (CE) is at present mainly based on imaging techniques. Serology has a complementary role, partly due to the small number of standardized and commercially available assays. Therefore we examined the clinical performance of the SERION ELISA classic Echinococcus IgG test. Using 10 U/ml as a cut-off point, and serum samples from 50 CE patients and 105 healthy controls, the sensitivity and specificity were 98.0% and 96.2%, respectively. If patients with other infectious diseases were used as negative controls, the specificity decreased to 76.9%, which causes poor positive predictive values. However, if results between 10 and 15 U/ml are classified as indecisive, the specificity of positive results (≥15 U/ml) increased to 92.5% without greatly affecting the sensitivity (92.0%). Using this approach in combination with imaging studies, the SERION ELISA classic Echinococcosis IgG test can be a useful aid in the diagnosis of CE.

Demographic and clinical characteristics of patients with anaphylactic shock after surgery for cystic echinococcosis.

PubMed

Li, Yimei; Zheng, Hong; Cao, Xinghua; Liu, Zaoling; Chen, Lili

2011-09-01

We reviewed the records of 446 patients who were treated surgically for cystic echinococcosis (CE) to identify risk factors for anaphylactic shock. Of 446 patients, 10 had final diagnoses of anaphylactic shock induced by CE; none died. The incidence of anaphylactic shock was significantly higher in younger age groups (P < 0.001) and in patients with pulmonary cysts. Anaphylactic shock induced by CE appears to differ from type I immediate hypersensitivity shock, which suggests that in CE, shock may be caused by a combination of immediate hypersensitivity and endotoxic shock. This possibility suggests that additional precautions should be taken during surgery. These precautions include reducing intracystic pressure, which would prevent possible leaked liquid from reaching other organs by surrounding the cyst with sterile gauze and decrease the chance of spreading the echinococcus; preventing antigen from contacting other tissues where it might trigger anaphylaxis; and resecting the cyst completely when feasible.

Echinococcosis: An Economic Evaluation of a Veterinary Public Health Intervention in Rural Canada

PubMed Central

Farag, Marwa; Zeng, Wu; Jenkins, Emily J.

2015-01-01

Echinococcosis is a rare but endemic condition in people in Canada, caused by a zoonotic cestode for which the source of human infection is ingestion of parasite eggs shed by canids. The objectives of this study were to identify risk factors associated with infection and to measure the cost-utility of introducing an echinococcosis prevention program in a rural area. We analyzed human case reports submitted to the Canadian Institutes for Health Information between 2002 and 2011. Over this 10 year period, there were 48 cases associated with E. granulosus/E. canadensis, 16 with E. multilocularis, and 251 cases of echinococcosis for which species was not identified (total 315 cases). Nationally, annual incidence of echinococcosis was 0.14 cases per 100 000 people, which is likely an underestimate due to under-diagnosis and under-reporting. Risk factors for echinococcosis included female gender, age (>65 years), and residing in one of the northern territories (Nunavut, Yukon, or Northwest Territories). The average cost of treating a case of cystic echinococcosis in Canada was $8,842 CAD. Cost-utility analysis revealed that dosing dogs with praziquantel (a cestocide) at six week intervals to control cystic echinococcosis is not currently cost-effective at a threshold of $20,000-100,000 per Quality Adjusted Life Year (QALY) gained, even in a health region with the highest incidence rate in Canada ($666,978 -755,051 per QALY gained). However, threshold analysis demonstrated that the program may become cost-saving at an echinococcosis incidence of 13-85 cases per 100,000 people and therefore, even one additional CE case in a community of 9000 people could result in the monetary benefits of the program outweighing costs. PMID:26135476

Diagnostic Accuracy of Antigen 5-Based ELISAs for Human Cystic Echinococcosis

PubMed Central

Pagnozzi, Daniela; Addis, Maria Filippa; Biosa, Grazia; Roggio, Anna Maria; Tedde, Vittorio; Mariconti, Mara; Tamarozzi, Francesca; Meroni, Valeria; Masu, Gabriella; Masala, Giovanna; Brunetti, Enrico; Uzzau, Sergio

2016-01-01

Background Clinical diagnosis and follow up of cystic echinococcosis (CE) are based on imaging complemented by serology. Several immunodiagnostic tests are commercially available, but the development of new tools is still needed to overcome the lack of standardization of the target antigen, generally consisting of a crude extract of Echinococcus granulosus hydatid cyst fluid. In a previous work, we described a chromatographic method for the preparation of a highly enriched Antigen 5 fraction from hydatid cyst fluid. The high reactivity of patient sera against this preparation prompted us to evaluate further this antigen for the serodiagnosis of CE on a larger cohort of samples. Methodology/Principal Findings A total of 327 sera from CE patients with heterogeneous conditions for cyst stage, cyst number, organ localization, drug therapy, and surgical intervention, together with 253 sera from healthy controls, were first analyzed by an ELISA based on the Ag5 preparation in two different experimental setups and, in parallel, by a commercial ELISA routinely used in clinical laboratories for CE serodiagnosis. The Ag5 ELISAs revealed different sensitivity (88.3% vs 95.3%) without significant differences in specificity (94.1% vs 92.5%), for the two setups, respectively. Moreover, possible relationships between the Ag5 ELISA absorbance results and clinical variables were investigated. Chi squared test, bivariate logistic regression and multiple regression analyses highlighted differences in the serology reactivity according to pharmacological treatment, cyst activity, and cyst number. Conclusions/Significance The two Ag5 ELISAs revealed different performances depending on the setup. The good diagnostic sensitivity and the high reliability of the Ag5 preparation method make this antigen a promising candidate for the serodiagnosis of CE. Further studies will be needed to evaluate the ability of our test to provide useful information on specific CE clinical traits. PMID

Cystic Echinococcosis in Spain: Current Situation and Relevance for Other Endemic Areas in Europe

PubMed Central

Rojo-Vazquez, Francisco A.; Pardo-Lledias, Javier; Francos-Von Hunefeld, Marcelo; Cordero-Sanchez, Miguel; Alamo-Sanz, Rufino; Hernandez-Gonzalez, Ana; Brunetti, Enrico; Siles-Lucas, Mar

2011-01-01

Cystic echinococcosis (CE) remains an important health problem in many regions of the world, both where no control measures have been implemented, and where control programs have been incompletely successful with ensuing re-emergence of the disease. In Spain, official data on CE show an increase in the proportion of intermediate hosts with CE during the last few years, and autochthonous pediatric patients have been reported, a sign of active local transmission of disease. A similar picture emerges from data reported to the European Food Safety Authority by other European countries. Nevertheless, several crucial aspects related to CE that would help better understand and control the disease have not been tackled appropriately, in particular the emergence of infection in specific geographical areas. In this respect, while some data are missing, other data are conflicting because they come from different databases. We review the current situation of CE in Spain compared with areas in which similar problems in the CE field exist, and offer recommendations on how to overcome those limitations. Specifically, we believe that the introduction of national registries for CE with online data entry, following the example set by the European Registry for Alveolar Echinococcosis, would help streamline data collection on CE by eliminating the need for evaluating and integrating data from multiple regions, by avoiding duplication of data from patients who access several different health facilities over time, and by providing much needed clinical and epidemiological data that are currently accessible only to clinicians. PMID:21283615

Prevalence of cystic echinococcosis in slaughtered cattle in Afyonkarahisar.

PubMed

Köse, Mustafa; Sevimli, Feride Kircali

2008-01-01

This study was carried out in 1001 cattle that were slaughtered in 3 abattoirs in the Afyonkarahisar district of western Turkey. Abattoirs were visited twice a week and internal organs of the cattle were examined for cystic echinococcosis (CE). The number of organs discarded because of CE was recorded. CE was found in 295 (29.47%) out of 1001 cattle studied. CE was found in 113 (44.31%) out of 255 cows and 182 (24.39%) out of 746 bulls. This difference was statistically significant (p < 0.001). CE was observed only in liver of 130 (44.06%) cattle, only in lungs of 91 (30.85%); only in hearts of 2 (0.68%) and only in the kidney of 1 (0.34%); in both livers and lungs of 70 (23.73%), and in both hearts and lungs of 1 (0.34%). Also, the rate of CE was significantly higher in cows than bulls in terms of organ predilection (p < 0.001). Forty three livers were completely discarded along with half of 18 livers, a quarter of 37 livers and the remaining livers in various degrees. Forty four lungs were completely destroyed along with half of 13 and a quarter of 21 whereas 3 hearts and one kidney were completely destroyed. The fertility rate of cysts was found to be 5.42%. In this study the prevalence of CE in cattle in Afyonkarahisar district has been documented and possible economic losses due to CE were emphasized.

Cystic echinococcosis in slaughtered domestic ruminants from Tunisia.

PubMed

Lahmar, S; Trifi, M; Ben Naceur, S; Bouchhima, T; Lahouar, N; Lamouchi, I; Maâmouri, N; Selmi, R; Dhibi, M; Torgerson, P R

2013-09-01

A total of 10,818 domestic ruminants (3913 cattle, 2722 sheep, 3779 goats, 404 dromedaries) slaughtered in various abattoirs in Tunisia between 2003 and 2010 were examined for the presence of Echinococcus granulosus hydatid cysts. The prevalence of cystic echinococcosis (CE) was 16.42% in sheep, 8.56% in cattle, 5.94% in dromedaries and 2.88% in goats. CE prevalence increased with age according to an asymptotic model and there was evidence of variation in infection pressure depending on the region of Tunisia where the animals were slaughtered. Cattle appeared to have the highest infection pressure of the species examined. The mean intensity of hepatic cysts was higher than that of pulmonary cysts in all species. The highest mean intensity of infection with E. granulosus larvae was observed in cattle (18.14) followed by sheep (9.58), goats (2.31) and dromedaries (2.12). The abundance of infection increased in a linear fashion with age in all animal species. Cyst abundance varied with species of animal and district of Tunisia. Cysts from dromedaries were more fertile (44.44%) than those from sheep (30.25%), goats (30.32%) and cattle (0.95%). The viability of the protoscoleces from fertile cysts from cattle (78.45%) was higher than those from sheep (70.71%) and camels (69.57%). The lowest protoscolex viability was recorded for hydatid cysts from goats (20.21%). This epidemiological study confirms the importance of CE in all domestic ruminant species, particularly in sheep, throughout Tunisia and emphasizes the need to interrupt parasite transmission by preventive integrated approaches in a CE control programme.

Alveolar echinococcosis in Europe and Poland. Threats to humans

PubMed

Gawor, Jakub

The increasing number of detected cases of alveolar echinococcosis (AE) in recent years in Central-Eastern Europe strongly support the need of accurate diagnosis of the disease in patients and its differentiation from cystic echinococcosis. Accordingly to the EU notification requirements both parasitoses are reported jointly as echinococcosis, which makes it impossible to assess epidemiological situation of each disease. AE threat to humans is due to the environmental contamination with eggs of the tapeworm Echinococcus multilocularis excreted by infected foxes. Also contribution of dogs to the parasite transmission should be taken into account in endemic areas. The article presents current data on human cases of AE registered in humans in Europe and Poland, threats of this parasitosis to humans and results of studies on E. multilocularis infection in animals, the definitive hosts.

Canine echinococcosis: global epidemiology and genotypic diversity.

PubMed

Carmena, David; Cardona, Guillermo A

2013-12-01

Canine echinococcosis is a potential zoonotic infection caused by the adult form of several cestode species belonging to the genus Echinococcus, of which E. granulosus sensu lato and E. multilocularis are the most epidemiologically relevant. Dogs infected with E. granulosus and E. multilocularis are widely regarded as the main source of infection for human cystic and alveolar echinococcosis, diseases that cause substantial morbidity and socio-economic burden in several regions of the world. Following our previous review on the global situation of cystic echinococcosis in livestock species (Cardona and Carmena. Vet. Parasitol. 2013;192:10-32), we summarize here current knowledge on the global epidemiology, geographical distribution and molecular diversity of Echinococcus spp. infection in dogs. We address relevant topics including the implications of the increasing urbanization of wildlife species such as foxes, coyotes, and dingoes in the establishment of urban cycles of Echinococcus spp., or the rising concerns regarding the role of unsupervised translocation of infected dogs in spreading the infection to Echinococcus-free areas. The involvement of wildlife species as natural reservoirs of disease to domestic animals and humans and the epidemiological significance of the sympatric occurrence of different Echinococcus species in the same geographical region are also debated. Data presented are expected to be useful for policy makers, educational and health authorities responsible for designing and implementing effective measures for disease control and prevention. Copyright © 2013 Elsevier B.V. All rights reserved.

Ultrasound as a monitoring tool for cystic echinococcosis in sheep.

PubMed

Dore, F; Varcasia, A; Pipia, A P; Sanna, G; Pinna Parpaglia, M L; Corda, A; Romig, T; Scala, A

2014-06-16

An ultrasound-based survey for cystic echinococcosis (CE) in sheep was carried out in Sardinia in 2012. The study was done on three farms (A, B, C) which had been pre-selected for different CE prevalence levels (A: >80%, B: 50-80%, C: <50%). In total, 129 sheep were examined on the farms using portable ultrasound equipment (A: n=51, B: n=30, C: n=48). Within a period of 20 days after ultrasound examination, all sheep were slaughtered and underwent a parasitological post-mortem examination for cysts in the liver and lungs. With post-mortem as gold standard, ultrasonography gave a test sensitivity of 88.7% and a specificity of 75.9%, while the positive and negative predictive values were 81.8% and 84.6%, respectively. When only sheep with fertile cysts were considered, the sensitivity of the test increased to 100%. We conclude that the ultrasound examination of the liver in sheep - using state-of-the-art technology - is a sensitive and specific diagnostic tool, which is cost-effective, highly appropriate for field use and requires only moderate time (no shaving required). The method can also be applied to other livestock species and will be useful tool in epidemiological studies, monitoring schemes and vaccination/control trials. Copyright © 2014 Elsevier B.V. All rights reserved.

Cystic echinococcosis: A neglected disease at usual and unusual locations.

PubMed

Sarkar, Soma; Roy, Himansu; Saha, Puranjay; Sengupta, Mallika; Sarder, Krisnendu; Sengupta, Manideepa

2017-01-01

Echinococcus granulosus causes a zoonotic infection called cystic echinococcosis (CE) or more commonly known as hydatid disease. Although the two most common locations of hydatid cyst are liver and lung, it may also appear in other parts of the body. Clinical presentation of the hydatid disease depends on the site and size of the lesion. A retrospective study was done in Medical College and Hospital, Kolkata, from January 2012 to June 2014, to find the site of involvement, distribution, clinical features, history of contact, mode of presentation, laboratory diagnosis, and treatment modalities of the cases of hydatid cyst. The cases were identified by radiological and laboratory methods, the data were entered in Excel spreadsheet, and analysis was done. Among the 21 cases of hydatid cyst included in the study, solitary hepatic involvement was seen in 11 (52.38%), pulmonary involvement in 4 (19%), and 6 (28.71%) were in unusual locations such as liver cyst extending as retroperitoneal, omental cyst, choledochal cyst, splenic cyst, and in hepatorenal pouch. History of contact with dog was seen in 15 (71.43%). All the patients were treated with surgery and albendazole and were discharged in healthy condition. CE may be present in usual and unusual locations with a lot of variations in the clinical features. Hence, proper radiological and laboratory diagnosis is required for accurate diagnosis and appropriate management of these cases.

Quantitative assessment of serum-specific IgE in the diagnosis of human cystic echinococcosis.

PubMed

Marinova, I; Nikolov, G; Michova, A; Kurdova, R; Petrunov, B

2011-07-01

Anti-Echinococcus serum immunoglobulin (Ig)E was assessed by the ImmunoCAP system and compared with anti-Echinococcus serum IgG assessed by enzyme-linked immunosorbent assay (ELISA) and Western blot. The ImmunoCAP system revealed very high specificity (one false positive of 110 healthy individuals), low cross-reactivity (one false positive of 58 patients with other diseases) and decreased sensitivity (73.55%). Receiver operating characteristic analysis displayed a beneficial diagnostic value with high accuracy. Comparison of the ImmunoCAP system with ELISA and Western blot showed significantly higher specificity and significantly lower cross-reactivity compared with the ELISA. Examination of sera from 155 patients with cystic echinococcosis (CE) showed varying levels of anti-Echinococcus IgE (range, 0.01-118.33 kUA/L). However, most samples had moderately elevated IgE levels. Analysis of serum-specific IgE revealed significantly higher sensitivity of the ImmunoCAP system and significantly higher antibody levels in hepatic CE compared with pulmonary CE. © 2011 Blackwell Publishing Ltd.

[Determination of the incidence of toxoplasmosis and cystic echinococcosis in Kocaeli].

PubMed

Sönmez Tamer, Gülden

2009-01-01

We determined the incidence of toxoplasmosis and cystic echinococcosis (CE) in 388 healthy high school students who reside in Kocaeli region and were not previously diagnosed as toxoplasmosis and CE positive. The collected serum samples were tested with ELISA for the presence of toxoplasmosis and CE. Toxoplasmosis serology was positive at 61 people (18%). Among them, 90% had the raw meat consumption habit and 81.7% had the history of close contact with cats. There was a statistically positive correlation between the raw-meat-consuming females and toxoplasmosis seropositivity (p < 0.05). The serology of CE was positive in 30 (8.9%) samples. Eighty percent of these, eozinophili was detected and a positive correlation was observed with CE (p < 0.05). Also, a positive relationship was detected between CE and males who had close contact with dogs (p < 0.05). Two students who were serologically positive with high titrations and one of them had the ultrasound test. A cyst with a diameter of 4-cm was observed in his liver. The results of this study led us conclude that the real incidence of the parasitic diseases and the endemic sites should be determined with seroepidemiological studies and the citizens, especially students should be informed about the contagion ways and precautions.

Assessment of the effect of Allium sativum on serum nitric oxide level and hepatic histopathology in experimental cystic echinococcosis in mice.

PubMed

Ali, Nehad Mahmoud; Ibrahim, Ayman Nabil; Ahmed, Naglaa Samier

2016-09-01

The current study was carried out to evaluate the prophylactic and therapeutic effects of Allium sativum on experimental cystic echinococcosis by measuring the serum nitric oxide level and studying hepatic histopathological changes. The experimental animals were divided into five groups, ten mice in each, group (I): prophylactic; group (II): therapeutic; group (III): prophylactic and therapeutic; group (IV): infected nontreated; group (V): non infected non treated. The results showed that serum nitric oxide was significantly increased as a result of infection in all infected groups compared to group V. Statistical significant difference was noted in serum nitrate level in group I at 1st and 8th week post infection compared to the same time interval in group IV. In group II, statistical significance was noticed only at the 1st week post infection. Statistical significant difference was noted in serum nitrate level in group III at 1st, 4th, 6th and 8th week post infection compared to same time interval in group IV. Hydatid cysts developed in livers of mice of group IV as early as 4 weeks of infection while no cysts were found in groups I,II and III. Histopathologically there were moderate pathological changes in group I and group II as hepatocytes showed moderate steatosis, moderate venous congestion and inflammatory cellular infiltrate with foci of degeneration and necrosis. While livers of mice of group III showed mild steatosis, mild venous congestion, mild inflammatory cellular infiltrate, no necrosis and no biliary hyperplasia. Accordingly, that garlic (Allium sativum) may be a promising phototherapeutic agent for cystic echinococcosis.

Management and outcome of cardiac and endovascular cystic echinococcosis.

PubMed

Díaz-Menéndez, Marta; Pérez-Molina, José Antonio; Norman, Francesca Florence; Pérez-Ayala, Ana; Monge-Maillo, Begoña; Fuertes, Pilar Zamarrón; López-Vélez, Rogelio

2012-01-01

Cystic echinococcosis (CE) can affect the heart and the vena cava but few cases are reported. A retrospective case series of 11 patients with cardiac and/or endovascular CE, followed-up over a period of 15 years (1995-2009) is reported. Main clinical manifestations included thoracic pain or dyspnea, although 2 patients were asymptomatic. Cysts were located mostly in the right atrium and inferior vena cava. Nine patients were previously diagnosed with disseminated CE. Echocardiography was the diagnostic method of choice, although serology, electrocardiogram, chest X-ray, computed tomography/magnetic resonance imaging and histology aided with diagnosis and follow-up. Nine patients underwent cardiac surgery and nine received long-term antiparasitic treatment for a median duration of 25 months (range 4-93 months). One patient died intra-operatively due to cyst rupture and endovascular dissemination. Two patients died 10 and 14 years after diagnosis, due to pulmonary embolism (PE) and cardiac failure, respectively. One patient was lost to follow-up. Patients who had cardiac involvement exclusively did not have complications after surgery and were considered cured. There was only one recurrence requiring a second operation. Patients with vena cava involvement developed PEs and presented multiple complications. Cardiovascular CE is associated with a high risk of potentially lethal complications. Clinical manifestations and complications vary according to cyst location. Isolated cardiac CE may be cured after surgery, while endovascular extracardiac involvement is associated with severe chronic complications. CE should be included in the differential diagnosis of cardiovascular disease in patients from endemic areas. © 2012 Díaz-Menéndez et al.

Cystic echinococcosis in immigrant from Peru: first case treated with percutaneous treatment in Japan.

PubMed

Sakaguchi, Hiroshi; Tanaka, Toshihiro; Marugami, Nagaaki; Kichikawa, Kimihiko; Horiuchi, Hazuki; Morioka, Chie; Toyohara, Masahisa; Moriya, Kei; Nishiofuku, Mariko; Mitoro, Akira; Fukui, Hiroshi; Hirai, Toshiko; Yamashita, Namiko; Ouji, Yukiteru; Ishizaka, Shigeaki; Yoshikawa, Masahide

2007-09-01

We report a case of cystic echinococcosis (CE) caused by Echinococcusgranulosus, for which a modified percutaneous evacuation (PEVAC) treatment was applied. The patient had immigrated from Peru to Japan and had 2 hydatid cystic masses, 1 located in segment (S)5 of the liver and the other in S3 (5.3 and 3.5 cm in diameter, respectively), both of which were visualized as pseudotumors by ultrasound (US) examinations. Albendazole treatment showed no effects and surgical treatment was refused. After punctuation of the S5 cyst under US guidance and S3 with CT guidance, 10- and 12-French gauge catheters, respectively, with multiple side holes were inserted. About 60 ml of the cyst contents was drawn out from the S5 lesion and 2 ml from the S3 lesion. Using repetitive manual injections and aspiration of small amounts of hypertonic saline, the remaining cyst content was removed as much as possible, after which 20 and 10 ml of 98% ethanol was injected into the S5 and S3 lesions, respectively. A short-term evaluation during the 4 month-period following the procedure using US revealed nearly complete evacuation of the S5 lesion, whereas that at S3 remained as a pseudo-solid mass. We consider that percutaneous treatment is a safe therapeutic modality for hydatid cysts. This is the first case report of CE treated percutaneously in Japan.

Combating echinococcosis in China: strengthening the research and development.

PubMed

Qian, Men-Bao; Abela-Ridder, Bernadette; Wu, Wei-Ping; Zhou, Xiao-Nong

2017-11-21

Echinococcosis is a neglected zoonotic disease, causing great morbidity and mortality due to the wide distribution of its endemic areas. China holds a high percentage in the global burden of both cystic and alveolar echinococcosis. A national survey conducted between 2012 and 2016 showed that an estimated 50 million people are at risk of contracting the disease in western China, of whom about 0.17 million are cases with echinococcosis.Despite this, research and development on echinococcosis in China is greatly inadequate compared to that in other countries. In this paper, we argue that there is a need for more research and work to be conducted in China on echinococcosis, including researching techniques in regards to diagnosis, treatment, and vaccination, and developing products through technical transformation and piloting strategies to control and even elimination.However, great opportunities exist for China to strengthen the research and development on this disease through initiatives such as Health China 2030, the Belt and Road Initiative, the China-Africa cooperation, as well as through further cooperation between China and the World Health Organization. All of these can bring us closer to controlling echinococcosis in China as well as in other countries. One element of crucial importance will be the training and development of professionals, which can be strengthened through international cooperation.

Echinococcosis: disease, detection and transmission.

PubMed

Craig, P S; Rogan, M T; Campos-Ponce, M

2003-01-01

Echinococcosis is one of the world's most geographically widespread parasitic zoonoses, with transmission occurring in tropical, temperate and arctic biomes. Most human infections are due to Echinococcus granulosus transmitted between domestic dogs and livestock, but this cosmopolitan species also cycles between wild carnivores (principally canids) and wild ungulates. The other species with significant zoonotic potential is E. multilocularis that occurs naturally in fox definitive hosts and small mammal intermediate hosts. These two species cause human cystic or alveolar echinococcosis respectively, which may be considered serious public health problems in several regions including developed countries. This review provides an introductory overview to the Supplement and summarises the biology and epidemiology of these two related cestodes with an emphasis on applied aspects relating to detection, diagnosis and surveillance in animal and human populations, and includes aspects of transmission ecology, and also considers aspects of community epidemiology and potential for control.

Percutaneous Treatment of Splenic Cystic Echinococcosis: Results of 12 Cases

DOE Office of Scientific and Technical Information (OSTI.GOV)

Akhan, Okan, E-mail: akhano@tr.net; Akkaya, Selçuk, E-mail: selcuk.akkaya85@gmail.com; Dağoğlu, Merve Gülbiz, E-mail: drmgkartal@gmail.com

PurposeCystic echinococcosis (CE) in the spleen is a rare disease even in endemic regions. The aim of this study was to examine the efficacy of percutaneous treatment for splenic CE.Materials and MethodsTwelve patients (four men, eight women) with splenic CE were included in this study. For percutaneous treatment, CE1 and CE3A splenic hydatid cysts were treated with either the PAIR (puncture, aspiration, injection, respiration) technique or the catheterization technique.ResultsEight of the hydatid cysts were treated with the PAIR technique and four were treated with catheterization. The volume of all cysts decreased significantly during the follow-up period. No complication occurred inmore » seven of 12 patients. Abscess developed in four patients. Two patients underwent splenectomy due to cavity infection developed after percutaneous treatment, while the spleen was preserved in 10 of 12 patients. Total hospital stay was between 1 and 18 days. Hospital stay was longer and the rate of infection was higher in the catheterization group. Follow-up period was 5–117 months (mean, 44.8 months), with no recurrence observed.ConclusionThe advantages of the percutaneous treatment are its minimal invasive nature, short hospitalization duration, and its ability to preserve splenic tissue and function. As the catheterization technique is associated with higher abscess risk, we suggest that the PAIR procedure should be the first percutaneous treatment option for splenic CE.« less

Humoural immune response and pathological analysis in patients with false immune diagnosis of cystic echinococcosis

PubMed Central

Chen, X; Zhang, J; Feng, X; Chen, X; Yin, S; Wen, H; Zheng, S

2014-01-01

The patients with false immune diagnosis of hydatid disease were investigated for the humoural immune response to analyse the possible reasons and mechanism leading to false immune diagnosis. Two hundred and thirty-nine patients with nature-unknown cysts and 30 healthy controls were detected by immunological assays (four hydatid antigen-based immunogold filtration assay and enzyme-linked immune absorbent assay) and ultrasound. Sensitivity of and specificity of immunological assay and ultrasound were calculated, respectively. The serological diagnosis was compared with surgical pathology to screen the patients with false immune diagnosis for the immunoglobulin measurement and pathological analysis. The history and cyst characteristics were also reviewed. The results indicate the immunoglobulin has little influence on false immunodiagnosis. The false-negative immunodiagnosis was caused by the cysts' inactive status while the false positive caused by previous rupture, antigen cross-reaction. The clinical diagnosis of cystic echinococcosis requires a combination of immunodiagnosis and ultrasonography, which is the necessary complementary confirmation. PMID:24372157

Cystic echinococcosis: a disease mimicking cancer in a non-endemic country report of two cases.

PubMed

Comrnejo-Juárez, P; Espinosa-Altamirano, A; Ibarra-del-Río, M; Pacheco-Bravo, I; Volkow-Fernández, P

2013-03-01

Echinococcosis is a parasitic disease that involves dogs as definitive host and sheep as intermediate host. Humans become infected incidentally through fecal-oral contact, particularly in the course of playful and close contact with an infected dog. Mexico is considered a region that is virtually free of cystic echinoccocosis. This manuscript describes two cases that were referred to a tertiary-care oncology hospital with a diagnosis of cancer. In one case, the presumptive diagnosis was liver cancer because abdominal ultrasonography revealed a low-density mass in the right hepatic lobe. Drainage was performed and cytologic examination of the fluid showed multiple Echinococcus cyst as well as prostoscolex. The case was resolved with percutaneous drainage and administration of albendazole for two months. In the second case, the patient was referred with a diagnosis of disseminated cervical cancer A cyst was identified in the upper right lung lobe; a diagnostic puncture was performed showing an Echinococcus cyst. This resolved solely with two months of albendazole administration.

Brain and lung metastasis of alveolar echinococcosis in a refugee from a hyperendemic area.

PubMed

Tappe, Dennis; Weise, David; Ziegler, Uwe; Müller, Andreas; Müllges, Wolfgang; Stich, August

2008-11-01

Alveolar echinococcosis (AE) of the liver with cerebral and pulmonary metastasis was diagnosed in a Tibetan monk who initially presented with severe headache to an emergency department in Germany. Multiple lesions with perifocal oedema and severe compression of the third ventricle were seen with computed tomography (CT) of the brain. Glioma or cerebral metastasis of a hitherto undiagnosed abdominal or pulmonary malignancy was suspected. CT scans of the lung and liver demonstrated further tumorous masses. Magnetic resonance imaging of the brain revealed the cystic nature of the cerebral lesions and the patient had a highly positive serology for AE. The echinococcal aetiology of the brain lesions was confirmed by PCR for this refugee from an area where two disease entities, AE and cystic echinococcosis, are hyperendemic.

Expression of Toll-Like Receptors 2 and 4 and Related Cytokines in Patients with Hepatic Cystic and Alveolar Echinococcosis

PubMed Central

Tuxun, Tuerhongjiang; Ma, Hai-Zhang; Apaer, Shadike; Zhang, Heng; Aierken, Amina; Li, Yu-Peng; Lin, Ren-Yong; Zhao, Jin-Ming; Zhang, Jin-Hui; Wen, Hao

2015-01-01

Several studies have demonstrated the important role of Toll-like receptors in various parasitic infections. This study aims to explore expression of Toll-like receptors (TLRs) and related cytokines in patients with human cystic echinococcosis (CE) and alveolar echinococcosis (AE). 78 subjects including AE group (N = 28), CE group (N = 22), and healthy controls (HC, N = 28) were enrolled in this study. The mRNA expression levels of TLR2 and TLR4 in blood and hepatic tissue and plasma levels related cytokines were detected by using ELISA. Median levels of TLR2 mRNA in AE and CE groups were significantly elevated as compared with that in healthy control group. Median levels of TLR4 expression were increased in AE and CE. Plasma concentration levels of IL-5, IL-6, and IL-10 were slightly increased in AE and CE groups compared with those in HC group with no statistical differences (p > 0.05). The IL-23 concentration levels were significantly higher in AE and CE groups than that in HC subjects with statistical significance. The increased expression of TLR2 and IL-23 might play a potential role in modulating tissue infiltrative growth of the parasite and its persistence in the human host. PMID:26635448

The clinical burden of human cystic echinococcosis in Palestine, 2010-2015

PubMed Central

Al-Jawabreh, Amer; Ereqat, Suheir; Dumaidi, Kamal; Nasereddin, Abdelmajeed; Al-Jawabreh, Hanan; Azmi, Kifaya; Al-Laham, Nahed; Nairat, Moath; Casulli, Adriano; Maqboul, Husni; Abdeen, Ziad

2017-01-01

Background Cystic echinococcosis (CE) is classified by the WHO as a neglected disease inflicting economic losses on the health systems of many countries worldwide. The aim of this case-series study was to investigate the burden of human CE in Palestine during the period between 2010 and 2015. Methodology/Principal findings Records of surgically confirmed CE patients from 13 public and private hospitals in the West Bank and Gaza Strip were reviewed. Patients’ cysts were collected from surgical wards and formalin-fixed paraffin-embedded (FFPE) blocks were collected from histopathology departments. Molecular identification of CE species /genotypes was conducted by targeting a repeat DNA sequence (EgG1 Hae III) within Echinococcus nuclear genome and a fragment within the mitochondrial cytochrome c oxidase subunit 1, (CO1). Confirmation of CE species/genotypes was carried out using sequencing followed by BLAST analysis and the construction of maximum likelihood consensus dendrogram. CE cases were map-spotted and statistically significant foci identified by spatial analysis. A total of 353 CE patients were identified in 108 localities from the West Bank and Gaza Strip. The average surgical incidence in the West Bank was 2.1 per 100,000. Spot-mapping and purely spatial analysis showed 13 out of 16 Palestinian districts had cases of CE, of which 9 were in the West Bank and 4 in Gaza Strip. Al-Khalil and Bethlehem were statistically significant foci of CE in Palestine with a six-year average incidence of 4.2 and 3.7 per 100,000, respectively. Conclusions/Significance To the best of our knowledge, this is the first confirmation of human CE causative agent in Palestine. This study revealed that E. granulosus sensu stricto (s.s.) was the predominating species responsible for CE in humans with 11 samples identified as G1 genotype and 2 as G3 genotype. This study emphasizes the need for a stringent surveillance system and risk assessment studies in the rural areas of high

Epidemiology of Echinococcosis Among Schoolchildren in Golog Tibetan Autonomous Prefecture, Qinghai, China.

PubMed

Cai, Huixia; Guan, Yayi; Ma, Xiao; Wang, Liying; Wang, Hu; Su, Guoming; Zhang, Xuefei; Han, Xiumin; Ma, Junying; Liu, Yu Fang; Li, Jun; Zhang, Jingxiao; Wang, Yongshun; Wang, Wei; Du, Rui; Lei, Wen; Wu, Weiping

2017-03-01

Echinococcosis is a serious zoonotic parasitic disease that is highly endemic in Qinghai Province. The present study aimed to investigate the prevalence of echinococcosis among schoolchildren in Golog Tibetan Autonomous Prefecture to improve early diagnosis and treatment of patients and to provide information for echinococcosis prevention and control. A total of 11,260 schoolchildren from five counties (Maqin, Gander, Dari, Jiuzhi, and Banma) in Golog Tibetan Autonomous Prefecture, Qinghai Province, were screened for echinococcosis. Screening involved ultrasound imaging combined with serologic examination as an auxiliary diagnostic test. The prevalence of echinococcosis in the schoolchildren was 2.1% (235/11,260), with a rate of 0.8% for cystic echinococcosis (CE; 89/11,260) and 1.3% for alveolar echinococcosis (AE; 146/11,260). Additionally, one child had a mixed infection. The prevalence ranged between 1.1% and 4.1% among the five investigated counties, and was highest in Dari County (4.1%). The prevalence of echinococcosis was higher in girls than in boys and gradually increased with age. In addition, children with CE mainly had type 1 (CE1) and type 3 (CE3) lesions, and children with AE mainly had small-diameter calcified lesions, suggesting that they were in the early asymptomatic stage of echinococcosis. In conclusion, children of Golog Tibetan Autonomous Prefecture appear to exhibit the highest recorded prevalence of CE and AE globally. Ultrasound is useful for screening populations in regions where both CE and AE are endemic.

Epidemiology of Echinococcosis among Schoolchildren in Golog Tibetan Autonomous Prefecture, Qinghai, China

PubMed Central

Cai, Huixia; Guan, Yayi; Ma, Xiao; Wang, Liying; Wang, Hu; Su, Guoming; Zhang, Xuefei; Han, Xiumin; Ma, Junying; Liu, Yu Fang; Li, Jun; Zhang, Jingxiao; Wang, Yongshun; Wang, Wei; Du, Rui; Lei, Wen; Wu, Weiping

2017-01-01

Echinococcosis is a serious zoonotic parasitic disease that is highly endemic in Qinghai Province. The present study aimed to investigate the prevalence of echinococcosis among schoolchildren in Golog Tibetan Autonomous Prefecture to improve early diagnosis and treatment of patients and to provide information for echinococcosis prevention and control. A total of 11,260 schoolchildren from five counties (Maqin, Gander, Dari, Jiuzhi, and Banma) in Golog Tibetan Autonomous Prefecture, Qinghai Province, were screened for echinococcosis. Screening involved ultrasound imaging combined with serologic examination as an auxiliary diagnostic test. The prevalence of echinococcosis in the schoolchildren was 2.1% (235/11,260), with a rate of 0.8% for cystic echinococcosis (CE; 89/11,260) and 1.3% for alveolar echinococcosis (AE; 146/11,260). Additionally, one child had a mixed infection. The prevalence ranged between 1.1% and 4.1% among the five investigated counties, and was highest in Dari County (4.1%). The prevalence of echinococcosis was higher in girls than in boys and gradually increased with age. In addition, children with CE mainly had type 1 (CE1) and type 3 (CE3) lesions, and children with AE mainly had small-diameter calcified lesions, suggesting that they were in the early asymptomatic stage of echinococcosis. In conclusion, children of Golog Tibetan Autonomous Prefecture appear to exhibit the highest recorded prevalence of CE and AE globally. Ultrasound is useful for screening populations in regions where both CE and AE are endemic. PMID:28070013

T-lymphocyte cytokine mRNA expression in cystic echinococcosis.

PubMed

Fauser, S; Kern, P

1997-04-01

In the present study we investigated cytokine mRNA expression by peripheral blood mononuclear cells (PBMC) from patients with cystic echinococcosis (CE) after stimulation with different antigens. By using reverse transcriptase polymerase chain reaction (RT-PCR) we could demonstrate that restimulation with crude Echinococcus granulosus antigen (Eg-Ag) induced or enhanced Th2 cytokine mRNA expression, especially IL-5 (by using antigen from sheep cyst fluid) in 23 out of 26 investigated CE patients and IL-10 (by using antigen from camel cyst fluid) in 10 out of 10 investigated CE patients. In contrast, IL-5 mRNA expression was absent in PBMC of healthy controls after Eg-Ag stimulation. To determine the specificity of this reaction we stimulated PBMC from 11 CE patients with crude Echinococcus multilocularis antigen (Em-Ag) and PBMC from 8 CE patients with Toxocara canis antigen (Tc-Ag). We found that the PBMC of patients showed a similar mRNA cytokine pattern on stimulation with Em-Ag when compared with Eg-Ag stimulation. The cytokine mRNA pattern on stimulation with Tc-Ag, however, resembled the cytokine mRNA pattern of unstimulated PBMC. Furthermore, the stimulation of PBMC with crude Mycobacterium tuberculosis antigen (H37Ra) and purified protein derivative (PPD) of M. tuberculosis revealed distinct IL-5 mRNA expression in all investigated CE patients, whereas in healthy controls IL-5 mRNA expression was very weak or totally absent. Thus, our results indicate an induction of Th2 cytokine mRNA expression in CE patients, which is frequently observed in parasite infections. Interestingly, this response persists after stimulation with tuberculosis antigens, which normally induce Th1 response.

Immunoblotting with human native antigen shows stage-related sensitivity in the serodiagnosis of hepatic cystic echinococcosis.

PubMed

Mariconti, Mara; Bazzocchi, Chiara; Tamarozzi, Francesca; Meroni, Valeria; Genco, Francesca; Maserati, Roberta; Brunetti, Enrico

2014-01-01

The diagnosis of hepatic cystic echinococcosis is based on ultrasonography and confirmed by serology. However, no biological marker of cyst viability is currently available implying years-long patient follow-up, which is not always feasible in endemic areas. We characterized the performance of an immunoblotting test based on human hydatid cyst fluid with particular regard to its ability to distinguish between cyst stages. Sera from patients with cysts in different stages showed distinctive band pattern recognition. Most importantly, the test discriminated in 80% of cases CE3a from CE3b transitional cysts, known to have different viability profiles. Interestingly, we observed a rapid change in band pattern recognition of sera from one patient at time points when his cyst passed from active to transitional to inactive stages. Further identification of different antigens expressed by different cyst stages will support the development of diagnostic tools that could early define cyst viability, to guide clinical decision making, and shorten patient follow-up.

A review of the global prevalence, molecular epidemiology and economics of cystic echinococcosis in production animals.

PubMed

Cardona, Guillermo A; Carmena, David

2013-02-18

Cystic echinococcosis (CE) is an important and widespread zoonotic infection caused by the larval stages of taeniid cestodes of the genus Echinococcus. The disease represents a serious animal health concern in many rural areas of the world, causing important economic losses derived from decreased productivity and viscera condemnation in livestock species. In this review we aim to provide a comprehensive overview on recent research progress in the epidemiology of CE in production animals from a global perspective. Particular attention has been paid to the discussion of the extent and significance of recent molecular epidemiologic data. The financial burden associated to CE on the livestock industry has also been addressed. Data presented are expected to improve our current understanding of the parasite's geographical distribution, transmission, host range, immunogenicity, pathogenesis, and genotype frequencies. This information should be also valuable for the design and implementation of more efficient control strategies against CE. Copyright © 2012 Elsevier B.V. All rights reserved.

The monetary burden of cystic echinococcosis in Iran.

PubMed

Fasihi Harandi, Majid; Budke, Christine M; Rostami, Sima

2012-01-01

Cystic echinococcosis (CE) is a globally distributed parasitic infection of humans and livestock. The disease is of significant medical and economic importance in many developing countries, including Iran. However, the socioeconomic impact of the disease, in most endemic countries, is not fully understood. The purpose of the present study was to determine the monetary burden of CE in Iran. Epidemiological data, including prevalence and incidence of CE in humans and animals, were obtained from regional hospitals, the scientific literature, and official government reports. Economic data relating to human and animal disease, including cost of treatment, productivity losses, and livestock production losses were obtained from official national and international datasets. Monte Carlo simulation methods were used to represent uncertainty in input parameters. Mean number of surgical CE cases per year for 2000-2009 was estimated at 1,295. The number of asymptomatic individuals living in the country was estimated at 635,232 (95% Credible Interval, CI 149,466-1,120,998). The overall annual cost of CE in Iran was estimated at US$232.3 million (95% CI US$103.1-397.8 million), including both direct and indirect costs. The cost associated with human CE was estimated at US$93.39 million (95% CI US$6.1-222.7 million) and the annual cost associated with CE in livestock was estimated at US$132 million (95% CI US$61.8-246.5 million). The cost per surgical human case was estimated at US$1,539. CE has a considerable economic impact on Iran, with the cost of the disease approximated at 0.03% of the country's gross domestic product. Establishment of a CE surveillance system and implementation of a control program are necessary to reduce the economic burden of CE on the country. Cost-benefit analysis of different control programs is recommended, incorporating present knowledge of the economic losses due to CE in Iran.

The Monetary Burden of Cystic Echinococcosis in Iran

PubMed Central

Fasihi Harandi, Majid; Budke, Christine M.; Rostami, Sima

2012-01-01

Cystic echinococcosis (CE) is a globally distributed parasitic infection of humans and livestock. The disease is of significant medical and economic importance in many developing countries, including Iran. However, the socioeconomic impact of the disease, in most endemic countries, is not fully understood. The purpose of the present study was to determine the monetary burden of CE in Iran. Epidemiological data, including prevalence and incidence of CE in humans and animals, were obtained from regional hospitals, the scientific literature, and official government reports. Economic data relating to human and animal disease, including cost of treatment, productivity losses, and livestock production losses were obtained from official national and international datasets. Monte Carlo simulation methods were used to represent uncertainty in input parameters. Mean number of surgical CE cases per year for 2000–2009 was estimated at 1,295. The number of asymptomatic individuals living in the country was estimated at 635,232 (95% Credible Interval, CI 149,466–1,120,998). The overall annual cost of CE in Iran was estimated at US$232.3 million (95% CI US$103.1–397.8 million), including both direct and indirect costs. The cost associated with human CE was estimated at US$93.39 million (95% CI US$6.1–222.7 million) and the annual cost associated with CE in livestock was estimated at US$132 million (95% CI US$61.8–246.5 million). The cost per surgical human case was estimated at US$1,539. CE has a considerable economic impact on Iran, with the cost of the disease approximated at 0.03% of the country's gross domestic product. Establishment of a CE surveillance system and implementation of a control program are necessary to reduce the economic burden of CE on the country. Cost-benefit analysis of different control programs is recommended, incorporating present knowledge of the economic losses due to CE in Iran. PMID:23209857

Detection of G1 genotype of human cystic echinococcosis in Egypt.

PubMed

Abd El Baki, Mohammad H; El Missiry, Adel M G; Abd El Aaty, Heba E M; Mohamad, Anhar A; Aminou, Heba A R

2009-12-01

The first trial to detect G1 genotype in Egyptian human isolates of hydatid cysts (HC) and serum samples to approach diagnosis of cystic echinococcosis (CE) using human sera by PCR. Using strain specific primers, 27/36 confirmed CE patients (75%) showed G1 specific band in their sera at 254 bp. Specificity was 100% without detecting bands for either other parasitosis, or mass occupying lesions. Using PCR, G1 genotype was detected in 83.3% of HC samples, without significant difference between types of human isolates (pulmonary, hepatic, or multi-organ). G1 genotype detection in human sera was in 75% of CE patients compared to 83.3% in HC samples of the same group of patients proved satisfactory, simple and safer than HCF sampling. IHAT gave sensitivity of 58.3% compared to histopathological examination of surgically removed cysts or examination of hydatid cyst fluid (HCF) for protoscolices (gold standards). The specificity was 70% with false positive reactions with other parasitic infections and mass occupying lesions. PCR detection of G1 genotype in Egyptian animal hydatid cysts showed 90% in camel isolates and 80% in sheep isolates, but pig isolates were negative. The presence of this genotype in a high percentage in camel isolates incriminated sheep strain as the source of CE camel infection. The results may give an explanation to the contradicting results of other studies that did not relay upon molecular aspects.

A Systematic Review of the Epidemiology of Echinococcosis in Domestic and Wild Animals

PubMed Central

Otero-Abad, Belen; Torgerson, Paul R.

2013-01-01

Background Human echinococcosis is a neglected zoonosis caused by parasites of the genus Echinococcus. The most frequent clinical forms of echinococcosis, cystic echinococcosis (CE) and alveolar echinococcosis (AE), are responsible for a substantial health and economic burden, particularly to low-income societies. Quantitative epidemiology can provide important information to improve the understanding of parasite transmission and hence is an important part of efforts to control this disease. The purpose of this review is to give an insight on factors associated with echinococcosis in animal hosts by summarising significant results reported from epidemiological studies identified through a systematic search. Methodology and Principal Findings The systematic search was conducted mainly in electronic databases but a few additional records were obtained from other sources. Retrieved entries were examined in order to identify available peer-reviewed epidemiological studies that found significant risk factors for infection using associative statistical methods. One hundred studies met the eligibility criteria and were suitable for data extraction. Epidemiological factors associated with increased risk of E. granulosus infection in dogs included feeding with raw viscera, possibility of scavenging dead animals, lack of anthelmintic treatment and owners' poor health education and indicators of poverty. Key factors associated with E. granulosus infection in intermediate hosts were related to the hosts' age and the intensity of environmental contamination with parasite eggs. E. multilocularis transmission dynamics in animal hosts depended on the interaction of several ecological factors, such as hosts' population densities, host-prey interactions, landscape characteristics, climate conditions and human-related activities. Conclusions/Significance Results derived from epidemiological studies provide a better understanding of the behavioural, biological and ecological factors

Evaluation of nitazoxanide for the treatment of disseminated cystic echinococcosis: report of five cases and literature review.

PubMed

Pérez-Molina, José A; Díaz-Menéndez, Marta; Gallego, José I; Norman, Francesca; Monge-Maillo, Begoña; Ayala, Ana Pérez; López-Vélez, Rogelio

2011-02-01

We aimed to evaluate the effectiveness of nitazoxanide in disseminated cystic echinococcosis (DCE) that failed to respond to surgical and antiparasitic therapy. We report on seven patients (five of them with bony involvement): two cases from the literature and five patients who were included in a compassionate trial of nitazoxanide therapy in our hospital. Median follow-up time until nitazoxanide therapy was 12 years and all patients had received prior medical treatment and extensive surgery. Nitazoxanide (500 mg/12 h) in combination with albendazole, with/without praziquantel, was administered for 3-24 months. Three patients improved: one with muscle involvement (clinico-radiological response), one with lung involvement (radiological response), and another with soft tissue and bony involvement (clinico-radiological response of soft tissue cysts). There was one discontinuation after 15 days of starting therapy. Nitazoxanide combination therapy could have a role in the treatment of DCE when there is no bony involvement. Long-term safety profile seems to be favorable.

Immunological Characteristics of Recurrent Echinococcosis-Induced Anaphylactic Shock

PubMed Central

Ye, Jianrong; Zhang, Qin; Ma, Long; Zheng, Hong

2016-01-01

Anaphylactic shock represents a serious complication of echinococcosis as up to 4.6% of patients die as a result of its severity and improper handling. Once a definite diagnosis is made, effective treatments need to be immediately initiated. Here, we report the immunological characteristics and management of two patients with recurrent anaphylactic shock concurrent with the surgical removal of hydatid cysts. Both patients had systemic echinococcosis classified as cystic echinococcosis type 2 (CE2) with multiple, immature cysts (absence of calcification and necrosis). In addition, both patients had increased eosinophils and basophils before surgery, as well as elevated crude hydatid cyst fluid antigen (anti-EgCF) and hydatid cyst fluid native antigen B (anti-EgB) antibodies and high IgG levels. Although we cannot definitively predict which patients are at risk for cyst fluid leakage or anaphylactic shock at present, clinicians may consider taking precautions before surgery on encountering patients with a similar profile to prevent the occurrence of anaphylactic shock and the likelihood of a second incident. However, these observations need to be confirmed in further studies with a larger number of patients. PMID:26711523

Immunological Characteristics of Recurrent Echinococcosis-Induced Anaphylactic Shock.

PubMed

Ye, Jianrong; Zhang, Qin; Ma, Long; Zheng, Hong

2016-02-01

Anaphylactic shock represents a serious complication of echinococcosis as up to 4.6% of patients die as a result of its severity and improper handling. Once a definite diagnosis is made, effective treatments need to be immediately initiated. Here, we report the immunological characteristics and management of two patients with recurrent anaphylactic shock concurrent with the surgical removal of hydatid cysts. Both patients had systemic echinococcosis classified as cystic echinococcosis type 2 (CE2) with multiple, immature cysts (absence of calcification and necrosis). In addition, both patients had increased eosinophils and basophils before surgery, as well as elevated crude hydatid cyst fluid antigen (anti-EgCF) and hydatid cyst fluid native antigen B (anti-EgB) antibodies and high IgG levels. Although we cannot definitively predict which patients are at risk for cyst fluid leakage or anaphylactic shock at present, clinicians may consider taking precautions before surgery on encountering patients with a similar profile to prevent the occurrence of anaphylactic shock and the likelihood of a second incident. However, these observations need to be confirmed in further studies with a larger number of patients. © The American Society of Tropical Medicine and Hygiene.

Endosonography of a Pulmonary Artery Obstruction in Echinococcosis.

PubMed

Schuuring, Mark J; Bonta, Peter I; van Vugt, Michele; Smithuis, Frank; van Delden, Otto M; Annema, Jouke T; Stijnis, Kees

2016-01-01

A 44-year-old woman with a history of pulmonary embolism and abdominal echinococcosis complained of sudden thoracic pain and shortness of breath. A D-dimer of 77.5 mg/l (reference ≤0.5 mg/l) was found. Chest CT scan revealed obstruction of the right lower and middle lobe pulmonary artery (PA). Anticoagulation therapy was initiated for the presumed diagnosis of recurrent pulmonary embolism. However, due to persistent symptoms of dyspnea, follow-up CT angiography of the chest was performed 3 months later. A persistent PA obstruction was found and the presumed diagnosis of embolism was questioned. Subsequently, endobronchial ultrasound (EBUS) imaging was performed to support an alternative diagnosis. EBUS imaging showed an inhomogeneous, sharply demarcated, intravascular lesion with round hypoechoic areas compatible with cysts. The diagnosis of embolism was rejected and treatment with albendazole was initiated for pulmonary echinococcosis. Echinococcosis is a parasitic disease and cystic spread in the PA is exceptional. The patient has remained stable for more than 4 years. In case of disease progression, including progressive PA obstruction or life-threatening hemoptysis, surgical resection will be considered. © 2016 S. Karger AG, Basel.

The natural history of cystic echinococcosis in untreated and albendazole-treated patients.

PubMed

Solomon, N; Kachani, M; Zeyhle, E; Macpherson, C N L

2017-07-01

The World Health Organization (WHO) treatment protocols for cystic echinococcosis (CE) are based on the standardized ultrasound (US) classification. This study examined whether the classification reflected the natural history of CE in untreated and albendazole-treated patients. Data were collected during mass US screenings in CE endemic regions among transhumant populations, the Turkana and Berber peoples of Kenya and Morocco. Cysts were classified using the WHO classification. Patient records occurring prior to treatment, and after albendazole administration, were selected. 852 paired before/after observations of 360 cysts from 257 patients were analyzed. A McNemar-Bowker χ 2 test for symmetry was significant (p<0.0001). 744 observations (87.3%) maintained the same class, and 101 (11.9%) progressed, consistent with the classification. Regression to CE3B occurred in seven of 116 CE4 cyst observations (6.0%). A McNemar-Bowker χ 2 test of 1414 paired before/after observations of 288 cysts from 157 albendazole-treated patients was significant (p<0.0001). 1236 observations (87.4%) maintained the same class, and 149 (10.5%) progressed, consistent with the classification. Regression to CE3B occurred in 29 of 206 CE4 observations (14.1%). Significant asymmetry confirms the WHO classification's applicability to the natural history of CE and albendazole-induced changes. Regressions may reflect the stability of CE3B cysts. Copyright © 2017. Published by Elsevier B.V.

Disseminated alveolar echinococcosis resembling metastatic malignancy: a case report.

PubMed

Caire Nail, Laura; Rodríguez Reimundes, Ezequiel; Weibel Galluzzo, Christelle; Lebowitz, Dan; Ibrahim, Yasmine Lucile; Lobrinus, Johannes Alexander; Chappuis, François

2017-04-18

Alveolar echinococcosis is a potentially lethal zoonosis caused by larval forms of the tapeworm Echinococcus multilocularis. Humans are aberrant intermediate hosts who become infected by ingestion of egg-contaminated food or water or via physical contact with domestic or wild animals that carry the parasite in their small intestine. In humans, the disease usually affects the liver and can spread to other organs causing metastatic infiltration. In this report, we describe an advanced presentation of human alveolar echinococcosis mimicking metastatic malignancy. A 62-year-old white woman was evaluated for fever, jaundice, and abdominal pain, associated with significant weight loss. She lived in a rural area in Switzerland and used to eat wild forest fruits and mushrooms. She owned cats that used to hunt rodents. On physical examination, she appeared severely ill with cachexia, altered mental status, jaundice, and massive hepatomegaly. Laboratory tests showed cholestasis with preserved liver function. An abdominal computed tomography scan showed an enlarged liver with a huge cystic mass in the right lobe extending into the left lobe, infiltrating her hepatic hilum, causing intrahepatic bile duct dilation and occlusion of her right portal vein. A chest computed tomography scan showed multiple calcified bilateral pulmonary nodules. Her clinical and radiological presentation resembled an advanced neoplastic disease. Serologic tests for Echinococcus multilocularis were positive. The diagnosis of alveolar echinococcosis was established on her past history of exposure, imaging, and serology results. Clinical presentation and radiologic imaging findings of disseminated alveolar echinococcosis can mimic metastatic malignancy, and diagnosis can be challenging in atypically advanced cases. As the incidence of human alveolar echinococcosis appears to be increasing in Europe and Switzerland, physicians should be aware of alveolar echinococcosis, its epidemiology, and its clinical

Human cystic echinococcosis in Heilongjiang Province, China: a retrospective study.

PubMed

Zhang, Tiemin; Zhao, Wei; Yang, Dong; Piao, Daxun; Huang, Shibo; Mi, Yuanyuan; Zhao, Xianqi; Cao, Jianping; Shen, Yujuan; Zhang, Weizhe; Liu, Aiqin

2015-03-10

Cystic echinococcosis (CE) is one of emerging zoonotic parasitic diseases throughout the world, having significant medical and economic importance in developing countries. The western and northwestern China is considered as CE endemic areas. In northeastern China's Heilongjiang Province, the increasing number of sporadic human CE cases has attracted more and more attention. The aims of the present study were to understand the clinical characteristics of human CE in the investigated area and to compare the coincidence rates of CT, ultrasound and serological test against the histopathology results among CE patients. Hospital data of 183 human CE cases in the period from January 2004 to July 2013 were collected from the two largest hospitals in Heilongjiang Province. Clinical data were analyzed, including age, gender, occupation and living residence of CE patients and localization, size and number of CE cysts as well as the diagnosis methods of CE before operation. The results revealed that the incidence of CE reached a peak in the age group of 41-50 years. Among the 183 CE patients, the females were observed to have a higher percentage of CE patients (60.66%, 111/183) than males (39.34%, 72/183). The majority of CE patients were farmers, followed by workers, employees, public servants, students and so on. CE cysts were most commonly found in the livers, with a 30 cm cyst in diameter being detected. CT showed the highest coincidence rate (96.64%) for hepatic CE among the three common diagnosis methods (CT, ultrasound imagine and serological test) compared against the histopathology results. This is the first retrospective analysis of human CE cases in Heilongjiang Province in recent ten years. Clinical characteristics of human CE were described here. CT appeared to be the most effective diagnosis method for hepatic CE.

Serological Diagnosis and Follow-Up of Human Cystic Echinococcosis: A New Hope for the Future?

PubMed Central

Manzano-Román, Raúl; Sánchez-Ovejero, Carlos; Hernández-González, Ana; Siles-Lucas, Mar

2015-01-01

Cystic echinococcosis (CE) is an important helminthic zoonotic disease caused by the Echinococcus granulosus complex. In humans, CE is a chronic disease driven by the growth of echinococcal cysts in different organs. Prognosis of this disease depends on multiple factors, including location, number, size, and stage of the cysts, making CE a disease of complex management. CE is usually asymptomatic for years and attracts limited attention from funding organizations and health authorities. For this reason, only experts' recommendations are available but no evidence-based conclusions have been drawn for CE clinical management. One of those pitfalls refers to the lack of evidence to support the use of serological tools for the diagnosis and follow-up of CE patients. In this respect, crude antigens are used to detect specific antibodies in patients, giving rise to false positive results. The advent of molecular techniques allowing the production of recombinant proteins has provided a number of candidate antigens that could overcome the problems associated with the use of crude parasite extracts in the serological assays. In this review, we present the last advances in this field, proposing the use of serology to support cyst stage-specific diagnosis and follow-up. PMID:26504805

[Pulmonary Echinococcosis: Surgical Aspects].

PubMed

Eichhorn, M E; Hoffmann, H; Dienemann, H

2015-10-01

Pulmonary cystic echinococcosis is a very rare disease in Germany. It is caused by the larvae of the dog tapeworm (echinococcus granulosus). The liver is the most affected organ, followed by the lungs. Surgery remains the main therapeutic approach for pulmonary CE. Whenever possible, parenchyma-preserving lung surgery should be preferred over anatomic lung resections. To ensure best therapeutic results, surgery needs to be performed under precise consideration of important infectiological aspects and patients should be treated in specialised centres based on interdisciplinary consensus. In addition to surgical aspects, this review summarises special infectiological features of this disease, which are crucial to the surgical approach. Georg Thieme Verlag KG Stuttgart · New York.

Cystic pulmonary hydatidosis

PubMed Central

Sarkar, Malay; Pathania, Rajnish; Jhobta, Anupam; Thakur, Babu Ram; Chopra, Rajesh

2016-01-01

Cystic echinococcosis (CE) is a zoonotic parasitic disease caused by the larval stages of the cestode Echinococcus granulosus. Worldwide, pulmonary hydatid cyst is a significant problem medically, socially, and economically. Surgery is the definitive therapy of pulmonary hydatidosis. Benzimidazoles may be considered in patients with a surgical contraindication. This review will focus on pathogenesis, lifecycle, clinical features, and management of pulmonary hydatid disease. PMID:27051107

Costs Associated with Surgically Treated Cases of Abdominal Cystic Echinococcosis: A Single Center's Experience from 2008 to 2014, Pavia, Italy

PubMed Central

Narra, Roberta; Maestri, Marcello; Budke, Christine M.; Tamarozzi, Francesca; Mariconti, Mara; Nicoletti, Giovanni J.; Rinaldi, Francesca; Brunetti, Enrico

2016-01-01

Cystic echinococcosis (CE) is a globally distributed zoonosis caused by the Echinococcus granulosus sensu lato species complex. Four approaches are available for treatment of abdominal CE: surgery, percutaneous aspiration, chemotherapy with albendazole, and watch-and-wait. Allocation of patients to these different treatment options mainly depends on the stage of the cystic lesion. However, as available guidelines are not widely followed, surgery is often applied even without the correct indication outside referral centers. This is not only a disadvantage for the patient, but also a waste of money. In this study, we evaluated the cost of the surgical approach for abdominal CE by analyzing hospitalization costs for 14 patients admitted to the General Surgery Ward at the “San Matteo” Hospital Foundation in Pavia, Italy, from 2008 through 2014. We found that the total cost of a single hospitalization, including hospital stay, surgical intervention, personnel, drugs, and administrative costs ranged from €5,874 to 23,077 (median €11,033) per patient. Our findings confirm that surgery can be an expensive option. Therefore, surgical intervention should be limited to cyst types that do not benefit from nonsurgical therapies and appropriate case management can best be accomplished by using a cyst stage-specific approach. PMID:27273641

Costs Associated with Surgically Treated Cases of Abdominal Cystic Echinococcosis: A Single Center's Experience from 2008 to 2014, Pavia, Italy.

PubMed

Narra, Roberta; Maestri, Marcello; Budke, Christine M; Tamarozzi, Francesca; Mariconti, Mara; Nicoletti, Giovanni J; Rinaldi, Francesca; Brunetti, Enrico

2016-08-03

Cystic echinococcosis (CE) is a globally distributed zoonosis caused by the Echinococcus granulosus sensu lato species complex. Four approaches are available for treatment of abdominal CE: surgery, percutaneous aspiration, chemotherapy with albendazole, and watch-and-wait. Allocation of patients to these different treatment options mainly depends on the stage of the cystic lesion. However, as available guidelines are not widely followed, surgery is often applied even without the correct indication outside referral centers. This is not only a disadvantage for the patient, but also a waste of money. In this study, we evaluated the cost of the surgical approach for abdominal CE by analyzing hospitalization costs for 14 patients admitted to the General Surgery Ward at the "San Matteo" Hospital Foundation in Pavia, Italy, from 2008 through 2014. We found that the total cost of a single hospitalization, including hospital stay, surgical intervention, personnel, drugs, and administrative costs ranged from €5,874 to 23,077 (median €11,033) per patient. Our findings confirm that surgery can be an expensive option. Therefore, surgical intervention should be limited to cyst types that do not benefit from nonsurgical therapies and appropriate case management can best be accomplished by using a cyst stage-specific approach. © The American Society of Tropical Medicine and Hygiene.

The study of apoptotic bifunctional effects in relationship between host and parasite in cystic echinococcosis: a new approach to suppression and survival of hydatid cyst.

PubMed

Spotin, Adel; Majdi, Monireh Mokhtari Amir; Sankian, Mojtaba; Varasteh, Abdolreza

2012-05-01

Cystic echinococcosis (hydatidosis) is a zoonotic helminthic disease of human and other intermediated hosts wherein infection is caused by the larval stages of tapeworm Echinococcus granulosus. Growth of the larval stage is formed throughout the internal organs, the liver and lung, causing their destruction. Important pathways are unknown about suppression and survival of cysts in human body. In this study, apoptotic bifunctional effects are evaluated in relationship between host and parasite in cystic echinococcosis. Human lymphocytes were treated with hydatid fluid (HF). After 6 h of exposure, caspase-3 activity was measured by fluorometric assay in the HF-treated lymphocytes and control cells. Also, the expression of Bax (as pro-apoptotic protein) and Bcl-2 (an anti-apoptotic protein) mRNA was assessed by semiquantitative reverse transcriptase polymerase chain reaction (RT-PCR) after 12 h of exposure. For surveying of apoptosis-inducing ligands TNF-related apoptosis-inducing ligand and Fas-L, germinal layer and accompaniment peripheral tissues as healthy control were separated by scalpel from each cyst in sterile condition, then were assess by semiquantitative RT-PCR method in mRNA expression. Both the ratio of Bax/Bcl-2 mRNA expression and caspase-3 activity were higher in the fertile fluid-treated lymphocytes relative to infertile fluid-treated lymphocytes and control group versus the expression level of apoptosis-inducing ligands having a relatively high level in germinal layer of infertile cyst in comparison to fertile cyst and healthy tissue. Apoptosis of germinal layer of fertile cysts is possibly one of the suppression mechanisms in hydatidosis patients, in contrast to lymphocytes apoptosis by modulator of hydatid fluid, one of the hydatid cyst survival mechanisms.

Interleukin-17A correlates with interleukin-6 production in human cystic echinococcosis: a possible involvement of IL-17A in immunoprotection against Echinococcus granulosus infection.

PubMed

Mezioug, Dalila; Touil-Boukoffa, Chafia

2012-01-01

Hydatidosis is a parasitic disease caused by the development, in humans and other mammals, of the larval form of Taenia, Echinococcus granulosus. It is one of the world's major zoonotic infections. This study aimed to examine interleukin-6 (IL-6), interferon-γ (IFN-γ) and interleukin-17A (IL-17A) production in patients with cystic echinococcosis (CE), and the role of IL-17A in the modulation of the immune response against the extracellular parasite, E. granulosus. A relationship between IL-6, IL-17A production and C reactive Protein (CRP) levels was also assessed. IL-6, IFN-γ, IL-17A and CRP production were determined in serum from Algerian hydatid patients. Cytokine production was also measured in supernatants from cultures of peripheral blood mononuclear cells (PBMCs) from hydatid patients stimulated by a major parasitic antigen (antigen-5). The increased activity of IL-6, IFN-γ and IL-17A were observed in most serum samples from patients. In contrast, healthy controls showed only minor levels. Similarly, high levels of CRP were detected. Our in vitro results indicate a positive correlation between IL-6, IFN-γ and IL-17A production in PBMC culture supernatants. However, IL-6, IFN-γ and IL-17A activity was low in serum and supernatants of PBMC cultures from relapsing patients, and there was no evidence of an immune response against parasitic antigen. Collectively, our results show that IL-17A was produced during human cystic echinococcosis, and was involved in the host defense mechanisms against the extracellular parasite E. granulosus. Our data suggest that IL-17A plays an immunoprotective role in this parasitic, helminth infection.

Cystic echinococcosis in cattle dairy farms: spatial distribution and epidemiological dynamics.

PubMed

Scala, Antonio; Bosco, Antonio; Pipia, Anna Paola; Tamponi, Claudia; Musella, Vincenzo; Costanzo, Nicola; Testoni, Francesco; Montisci, Antonio; Mocci, Giovanni; Longhi, Alessandro; Tilocca, Laura; Rinaldi, Laura; Cringoli, Giuseppe; Varcasia, Antonio

2017-05-29

A study monitoring cystic echinococcosis (CE) in adult dairy cattle from intensive livestock farms located in the municipality of Arborea (Sardinia, Italy) was carried out between 2012 and 2015. A retrospective study of veterinary reporting forms of post-mortem inspections in 10 different Italian slaughterhouses was also performed. In addition, data on viability and molecular characterisation of hydatid cysts removed from parasitised organs in cattle was carried out. A geographical information system (GIS) with data layers of the study area and the geo-referenced points of 160 cattle farms was constructed. CE was found in 21.9% (35/160) of the surveyed farms. The retrospective study revealed that 0.05% (13/23,656) of adult slaughtered animals (over one year of age) from Arborea had tested positive to CE. The results stratified per year showed the following CE prevalences: 0.09% (5/5673) in 2012; 0.02% (1/5682) in 2013; 0.08% (5/6261) in 2014; and 0.03% (2/6040) in 2015 (χ2 with 3 degrees of freedom=3.81; P=0.282). The E. granulosus sensu stricto (formerly called G1 or sheep strain) was detected in all cysts subjected to molecular analysis. The GIS analysis showed that CE is fairly resilient in the Arborea territory where most of cattle farms are located, while a small cluster of cases was found located in the southeastern part of Arborea, close to districts where sheep farms are situated. The present survey reports the presence of CE in Sardinian dairy cattle intensive farms and suggests that the parasitic pressure of CE in the island continues to be very strong.

PULMONARY CYSTIC ECHINOCOCCOSIS

PubMed Central

Santivanez, Saul; Garcia, Hector H.

2012-01-01

Pulmonary cystic echinococosis, a zoonosis caused by the larvae of the dog tapeworm Echinococcus granulosus, is considered as a major public health problem in those countries where dogs are used to care for large herds because of the incapacitating effects produced in affected population. The ratio lung:liver involvement is higher in children than in adults. A higher proportion of lung cases are discovered incidentally on a routine x-ray evaluation; the majority of infected people remain asymptomatic until the cyst enlarges sufficiently to cause symptoms. The majority of symptoms are caused by mass effect from the cyst volume; the presence of complications caused by cysts broke changes the clinical presentation; the principal complication is cyst rupture, producing cough, chest pain, hemoptysis, or vomica. Diagnosis is obtained by imaging evaluation (Chest X-ray or CT scan), supported by serology in the majority of cases. Surgery is the main therapeutic approach, having as principal objective, the removal of the parasite, preventing intraoperative dissemination; the use of pre surgical chemotherapy reduces the chances of seeding and recurrence; treatment using benzimidazoles is the preferred treatment when surgery is not available, or complete removal is not feasible PMID:20216420

Molecular characterization of Echinococcus granulosus isolates from Bulgarian human cystic echinococcosis patients.

PubMed

Marinova, Irina; Spiliotis, Markus; Wang, Junhua; Muhtarov, Marin; Chaligiannis, Ilias; Sotiraki, Smaro; Rainova, Iskra; Gottstein, Bruno; Boubaker, Ghalia

2017-03-01

Although cystic echinococcosis (CE) is highly endemic in Bulgaria, there is still scarce information about species and/or genotypes of the Echinococcus granulosus complex that infect humans. Our study tackled the genetic diversity of E. granulosus complex in a cohort of 30 Bulgarian CE patients. Ten animal E. granulosus isolates from neighboring Greece were additionally included. Specimens were comparatively analyzed for partial sequences of five mitochondrial (mt) (cox I, nad I, rrnS, rrnL, and atp6) and three nuclear (nc) genes (act II, hbx 2, and ef-1α) using a PCR-sequencing approach. All 30 Bulgarian isolates were identified as E. granulosus sensu stricto (s.s.) and were showing identical sequences for each of the three examined partial nc gene markers. Based upon concatenated sequences from partial mtDNA markers, we detected 10 haplotypes: 6 haplotypes (H1-H6) clustering with E. granulosus s.s. (G1) and 4 haplotypes (H9-H13) grouping with E. granulosus s.s. (G3), with H1 and H10 being the most frequent in Bulgarian patients. The haplotypes H1, H4, and H11 were also present in Greek hydatid cyst samples of animal origin. In conclusion, E. granulosus s.s. (G1 and G3 genotypes) is the only causative agent found so far to cause human CE in Bulgaria. However, further studies including larger sample sizes and other additional geographic regions in Bulgaria will have to be performed to confirm our results.

Cystic echinococcosis in Sardinia: farmers' knowledge and dog infection in sheep farms.

PubMed

Varcasia, A; Tanda, B; Giobbe, M; Solinas, C; Pipia, A P; Malgor, R; Carmona, C; Garippa, G; Scala, A

2011-09-27

Cystic Echinococcosis (CE) is one of the most widespread parasitic diseases in Sardinia, the second largest Mediterranean island where almost 3,558,000 milk sheep were raised extensively. The aim of this survey was to evaluate the level of farmers' knowledge on CE transmission, focusing on the role of human to facilitate the persistence of this zoonosis in Sardinia after 14 years after the last campaign against CE. The other goal of the survey is to update on presence of Echinococcus granulosus in its definitive hosts through three ELISA coproantigen tests. An interview was carried out with 172 farmers. The questionnaire was designed to include possible factors associated with the transmission of Echinococcosis: ownership and number of dogs, the use of anthelmintic drugs against dog cestode, frequency of anthelmintic treatment in dogs, home slaughtering and offal disposal. Individual faecal samples were retrieved from 300 dogs, and after a preliminary macroscopic examination to discover adult worms and/or proglottids, was submitted to copromicroscopic examination. Coproantigens were then extracted according to the protocol described by Allan et al. (1992), and subsequently stored at -20°C until use. Faecal soluble antigens from E. granulosus were detected using three different ELISA coproantigen assays: (a) the commercially produced Chekit Echinotest (Bommeli, Bern, CH) based on polyclonal antibodies against adult excretory/secretory (E/S) antigens; (b) a sandwich ELISA that uses rabbit polyclonal antibodies against adult E/S antigens and biotinylated monoclonal antibody EmA9 produced against adult Echinococcus multilocularis somatic extract (Malgor et al., 1997); and (c) a sandwich assay that uses monoclonal antibody EgC3 produced by immunization with adult E. granulosus E/S products (Casaravilla et al., 2005). Questionnaire results reveal that on all farms home-slaughtering was done, and offal was used as dog meal raw (17%) or after boiling (37%), discarded in

Echinococcosis: Unexpected Occurrence and the Diagnostic Contribution of Routine Histopathology.

PubMed

Taxy, Jerome B; Gibson, William E; Kaufman, Michael W

2017-01-01

Echinococcus is the smallest of the major tapeworms. Humans are incidental hosts who become infected by exposure to soil contaminated with ova from the feces of the canine definitive hosts. The major species are E. granulosus (cystic echinococcosis) and E. multilocularis (alveolar or sylvatic echinococcosis), both common worldwide but unusual and not reportable in the United States. Human disease is characterized by slowly growing visceral cystic masses often containing daughter cysts and hydatid sand. In nonendemic areas, the clinical evaluation includes imaging, serology, and surgery; metastatic tumor is a major consideration. The diagnosis depends on the morphologic recognition of scolices and hooklets, albeit degenerated, within the cyst or attached to the cyst wall. Highly suggestive cyst wall features are an acellular hyaline lining surrounded by focally calcific fibrous tissue and chronic inflammation. We report 7 patients encountered in the Chicago area (5 E. granulosus, 2 E. multilocularis) who presented with cystic masses affecting the liver, lung, soft tissue, and spleen. The initial diagnosis was established or suggested by intraoperative evaluation of cyst fluid or cyst wall histopathology. Organism parts were found in 5 cases; 1 patient had a hyalinized cyst wall with a positive IgG (enzyme-linked immunosorbent assay), and 1 had a positive Western blot and a 30-year history of treatment. Treatment included cyst resection, hepatic lobectomy, intraoperative cyst injection, and benzimidazole therapy. There was 1 death. In a nonendemic area, the awareness of the pathologist to the possibility of this disease will exclude a neoplasm and facilitate prompt antihelminthic treatment.

A study to assess the awareness of risk factors of cystic echinococcosis in Turkey

PubMed Central

Aydın, Mehmet F.; Adıgüzel, Emre; Güzel, Hakan

2018-01-01

Objectives: To determine the knowledge, attitudes, practices, and awareness for risk factors associated with cystic echinococcosis (CE) among animal breeders, nurses, nursing students, dietitians, food professionals and other public groups in Turkey. Methods: Questionnaire forms were prepared in compliance with the individuals’ diversity, and a survey was created about agents, transmission modes, epidemiology, treatment, and risk factors for CE. The descriptive survey was performed between May 2015 and May 2016 by face-to-face communication. Data were collected from a total of 647 individuals, including 95 animal breeders, 92 nurses, 249 nursing students, 49 dietitians, 28 food professionals and 134 people from other public groups from Turkey. Survey data were evaluated in terms of percentages and means and analyzed by the Pearson’s chi-square test and Mann-Whitney U test to determine the relationships between questionnaire answers and socio-demographic attributes, such as age, gender, educational level, and profession. Non-categorical variables were compared via Spearman’s rho correlation analysis with statistical significance set at the 0.05 level. Results: We determined low awareness for vegetable washing, raw vegetables and offal consumption, informative courses, contact with dogs, and administration of antiparasitic drugs to dogs. Conclusion: The participants of this survey were found to be with insufficient knowledge regarding risk factors of CE. We advocate the implementation of training programs to improve public awareness on this important disease. PMID:29543307

A case of pulmonary and hepatic cystic Echinococcosis of CE1 stage in a healthy Japanese female that was suspected to have been acquired during her stay in the United Kingdom.

PubMed

Nakamura, Kiwamu; Ito, Akira; Yara, Satomi; Haranaga, Shusaku; Hibiya, Kenji; Hirayasu, Tsuneo; Sako, Yasuhito; Fujita, Jiro

2011-09-01

We herein report a case of a young Japanese female who was confirmed to have cystic echinococcosis (CE) 1 stage based on the World Health Organization Informal Working Group on Echinococcosis pathological classification of CE, and she was also suspected to be infected with eggs of the G1 Echinococcus granulosus sensu stricto during her stay in the United Kingdom and therefore, suffered from synchronous pulmonary and hepatic CE. Oral albendazole was administered initially, but rupture of a lung hydatid cyst was observed. To avoid additional rupture, we performed two surgeries. CE is very rare in Japan; all CE cases in Japan during the past two decades have been confirmed to be imported, and almost all cases are hepatic CE. This case is the first case report of a Japanese patient who had concomitant giant lung and liver CE with early-stage CE1 and was successfully treated by surgery and pharmacotherapy with a serological follow-up.

Cystic echinococcosis amongst small ruminants and humans in central Ethiopia.

PubMed

Assefa, Habtamu; Mulate, Belay; Nazir, Shahid; Alemayehu, Alula

2015-08-21

This study was conducted to determine the prevalence of cystic echinococcosis (CE) in small ruminants and humans in Addis Ababa, central Ethiopia. A cross-sectional study involving systematic random sampling was conducted to estimate the prevalence of CE in 512 small ruminants (262 sheep and 250 goats) slaughtered at Addis Ababa Abattoir Enterprise between October 2011 and March 2012. Hydatid cysts were identified macroscopically during postmortem examination and their fertility and viability were determined. CE was observed in 21 (8.02%) sheep and 17 (6.80%) goats. In sheep 13 (4.96%) of the lungs, 10 (3.81%) livers and 1 (0.381%) heart were found to be infected with hydatid cysts. Involvement of lung and liver in goats was found to be 10 (4.0%) and 8 (3.2%) respectively, with no cysts recorded in the heart. Of the total of 77 and 47 cysts encountered in sheep and goats, 33 (42.85%) and 15 (31.91%) respectively were fertile. Viability of protoscoleces from fertile cysts in sheep (29 [87.87%]) was higher than in goats (6 [40.0%]). For humans, retrospective analysis covering five years of case reports at two major hospitals in Addis Ababa between January 2008 and December 2012 showed that of the total of 25 840 patients admitted for ultrasound examination, 27 CE cases were registered, a prevalence of 0.1% and mean annual incidence rate of approximately 0.18 cases per 100 000 population. Liver was the major organ affected in humans (81.5% in affected patients) followed by spleen (11.1%) and kidney (7.4%). Logistic regression analysis showed that prevalence of CE varied significantly in relation to host age in the small ruminants (OR = 3.93, P < 0.05) as well as in humans (95% CI, R = 4.8). This epidemiological study confirms the importance of CE in small ruminants and humans in central Ethiopia, emphasising the need for integrated approaches to controlling this neglected preventable disease.

First description of Echinococcus ortleppi and cystic echinococcosis infection status in Chile.

PubMed

Corrêa, Felipe; Stoore, Caroll; Horlacher, Pamina; Jiménez, Mauricio; Hidalgo, Christian; Alvarez Rojas, Cristian A; Figueiredo Barros, Guilherme; Bunselmeyer Ferreira, Henrique; Hernández, Marcela; Cabrera, Gonzalo; Paredes, Rodolfo

2018-01-01

Cystic echinococcosis (CE), a parasitic disease caused by the cestode Echinococcus granulosus sensu lato (s.l.), is a worldwide zoonotic infection. Although endemic in Chile, information on the molecular characteristics of CE in livestock remains scarce. Therefore we aimed to describe the status of infection with E. granulosus s.l. in cattle from central Chile and also to contribute to the study of the molecular epidemiology of this parasite. According to our results, the prevalence of CE is 18.84% in cattle, similar to previous reports from Chile, suggesting that the prevalence in Santiago Metropolitan area has not changed in the last 30 years. Most of the cysts were found only in lungs (51%), followed by concurrent infection in liver and lungs (30%), and only liver (19%). Molecular characterization of the genetic diversity and population structure of E. granulosus s.l. from cattle in central Chile was performed using a section of the cytochrome c oxidase subunit 1 (cox1) mitochondrial gene. E. granulosus sensu stricto (s.s.) (G1-G3 genotypes) was confirmed by RFLP-PCR to be the dominant species affecting cattle (284 samples/290 samples); we also report for the first time in Chile the presence of E. ortleppi (G5 genotype) (2 samples/61 samples). The Chilean E. granulosus s.s. parsimony network displayed 1 main haplotype. Additional studies using isolates from many locations across Chile and different intermediate hosts will provide more data on the molecular structure of E. granulosus s.s. within this region. Likewise, investigations of the importance of E. ortleppi in human infection in Chile deserve future attention.

Post-Treatment Follow-Up Study of Abdominal Cystic Echinococcosis in Tibetan Communities of Northwest Sichuan Province, China

PubMed Central

Li, Tiaoying; Ito, Akira; Pengcuo, Renqing; Sako, Yasuhito; Chen, Xingwang; Qiu, Dongchuan; Xiao, Ning; Craig, Philip S.

2011-01-01

Background Human cystic echinococcosis (CE), caused by the larval stage of Echinococcus granulosus, with the liver as the most frequently affected organ, is known to be highly endemic in Tibetan communities of northwest Sichuan Province. Antiparasitic treatment with albendazole remains the primary choice for the great majority of patients in this resource-poor remote area, though surgery is the most common approach for CE therapy that has the potential to remove cysts and lead to complete cure. The current prospective study aimed to assess the effectiveness of community based use of cyclic albendazole treatment in Tibetan CE cases, and concurrently monitor the changes of serum specific antibody levels during treatment. Methodology/Principal Findings Ultrasonography was applied for diagnosis and follow-up of CE cases after cyclic albendazole treatment in Tibetan communities of Sichuan Province during 2006 to 2008, and serum specific IgG antibody levels against Echinococcus granulosus recombinant antigen B in ELISA was concurrently monitored in these cases. A total of 196 CE cases were identified by ultrasound, of which 37 (18.9%) showed evidence of spontaneous healing/involution of hepatic cyst(s) with CE4 or CE5 presentations. Of 49 enrolled CE cases for treatment follow-up, 32.7% (16) were considered to be cured based on B-ultrasound after 6 months to 30 months regular albendazole treatment, 49.0% (24) were improved, 14.3% (7) remained unchanged, and 4.1% (2) became aggravated. In general, patients with CE2 type cysts (daughter cysts present) needed a longer treatment course for cure (26.4 months), compared to cases with CE1 (univesicular cysts) (20.4 months) or CE3 type (detached cyst membrane or partial degeneration of daughter cysts) (9 months). In addition, the curative duration was longer in patients with large (>10 cm) cysts (22.3 months), compared to cases with medium (5–10 cm) cysts (17.3 months) or patients with small (<5 cm) cysts (6 months). At

Expert Reliability for the World Health Organization Standardized Ultrasound Classification of Cystic Echinococcosis

PubMed Central

Solomon, Nadia; Fields, Paul J.; Tamarozzi, Francesca; Brunetti, Enrico; Macpherson, Calum N. L.

2017-01-01

Cystic echinococcosis (CE), a parasitic zoonosis, results in cyst formation in the viscera. Cyst morphology depends on developmental stage. In 2003, the World Health Organization (WHO) published a standardized ultrasound (US) classification for CE, for use among experts as a standard of comparison. This study examined the reliability of this classification. Eleven international CE and US experts completed an assessment of eight WHO classification images and 88 test images representing cyst stages. Inter- and intraobserver reliability and observer performance were assessed using Fleiss' and Cohen's kappa. Interobserver reliability was moderate for WHO images (κ = 0.600, P < 0.0001) and substantial for test images (κ = 0.644, P < 0.0001), with substantial to almost perfect interobserver reliability for stages with pathognomonic signs (CE1, CE2, and CE3) for WHO (0.618 < κ < 0.904) and test images (0.642 < κ < 0.768). Comparisons of expert performances against the majority classification for each image were significant for WHO (0.413 < κ < 1.000, P < 0.005) and test images (0.718 < κ < 0.905, P < 0.0001); and intraobserver reliability was significant for WHO (0.520 < κ < 1.000, P < 0.005) and test images (0.690 < κ < 0.896, P < 0.0001). Findings demonstrate moderate to substantial interobserver and substantial to almost perfect intraobserver reliability for the WHO classification, with substantial to almost perfect interobserver reliability for pathognomonic stages. This confirms experts' abilities to reliably identify WHO-defined pathognomonic signs of CE, demonstrating that the WHO classification provides a reproducible way of staging CE. PMID:28070008

Slaughterhouse survey of cystic echinococcosis in cattle and sheep from the Republic of Moldova.

PubMed

Chihai, O; Umhang, G; Erhan, D; Boué, F; Tălămbuţă, N; Rusu, Ş; Zamornea, M

2016-05-01

The Echinococcus granulosus tapeworm is responsible for cystic echinococcosis (CE), a zoonotic disease with worldwide distribution. The life cycle of the parasite is mainly domestic and takes place between dogs and livestock species. A slaughterhouse survey was conducted in 2012 in the Republic of Moldova in order to estimate the prevalence of CE. A total of 1525 cattle, 5580 sheep and 12,700 pigs were surveyed. No CE infection was observed in pigs, while prevalence was estimated at 59.3% in cattle and 61.9% in sheep. Infection was significantly higher in animals raised in private households than in those from collective farms. The frequency of infection increased with age in both species. In cattle and in sheep, infection of both the liver and lungs was the most common, while infection in the lungs only was much less frequent. Farm type appears to be an important factor in CE infection in Moldova, due to the extensive farming and the home-slaughtering undertaken in the majority private sector, despite a high prevalence of CE also recorded in the public sector. The low fertility of cysts in cattle (1.1%) compared to sheep (47.6%) confirmed the maintenance of E. granulosus sensu stricto in a dog-sheep life cycle which excludes cattle. Further studies are needed to obtain a complete overview of the parasite's epidemiology in its intermediate and definitive hosts, in order to implement control and preventive measures, with specific attention given to farms in the private sector.

Comparative Evaluation of Liposomal Albendazole and Tablet-Albendazole Against Hepatic Cystic Echinococcosis

PubMed Central

Li, Haitao; Song, Tao; Shao, Yingmei; Aili, Tuergan; Ahan, Ayifuhan; Wen, Hao

2016-01-01

Abstract In this study, we aimed to compare the clinical efficacy of liposomal albendazole (L-ABZ) and tablet-albendazole (T-ABZ) for the treatment of human hepatic cystic echinococcosis (CE). Sixty patients with single cyst (CE1) or daughter cyst (CE2) were included in this study and were nonrandomly divided into the L-ABZ group (n = 30, 10 mg/kg per day, p.o., b.i.d.) and T-ABZ group (n = 30, 12–20 mg/kg per day, p.o., b.i.d.), respectively. The treatment duration lasted for 6 months, during which dynamic follow-up was carried out to evaluate the clinical efficacy through calculating the total effective rates (TERs). Measurement data and numerous data were analyzed by the chi-square test. Two-sided tests were performed for all the statistical tests. In our study, 2 patients were lost in the follow-up in the L-ABZ group. One patient was lost in the follow-up in the T-ABZ group, and 1 patient was withdrawal from the study due to receiving surgery. Significant difference was identified in the 3-month TERs of L-ABZ group and T-ABZ group (33.3% vs 76.7%, P < 0.05). Also, remarkable difference was noted in the 6-month TERs in the L-ABZ group and T-ABZ group (66.7% vs 93.3%, P = 0.01). No statistical difference was noticed in the incidence rate of adverse reactions in both groups (P > 0.05). Based on our study, both T-ABZ and L-ABZ are effective for treating human CE. The TER in the L-ABZ group is superior to that of T-ABZ. PMID:26825878

Morbidity pattern of hydatid disease (cystic echinococcosis) and lack of its knowledge in patients attending Mamata General Hospital, Khammam, Andhra Pradesh.

PubMed

Hemachander, S Suguna; Prasad, C Rajendra; Jessica, M

2008-01-01

There is hearsay that prevalence of hydatid disease in Khammam and Nalgonda districts of Andhra Pradesh is high. We report here a preliminary study conducted to determine the magnitude of the problem of hydatid disease and the morbidity associated with it in patients attending MGH, KMM, A.P. (rural hospital). Eleven cases were identified during the period from November 2005 to May 2006 (seven months). Pain in abdomen, mass per abdomen, loss of appetite, pregnancy complicated by cystic echinococcosis (CE), and jaundice were the main clinical symptoms and signs. Ultrasonography, detection and removal of the cysts on the operation table, microscopic examination of the aspirated hydatid fluid were confirmatory. Ziehl-Neelsen stain of the aspirated fluid revealed acid-fast scolices. Interrogation of the patients and their family members (50) revealed that there was a total lack of knowledge of dog-tapeworm-caused infection in humans. They knew 'rabies' as the only disease man gets from dogs, and tapeworms are from pork and beef.

Multivisceral echinococcosis: concept, diagnosis, management.

PubMed

Grozavu, C; Ilias, M; Pantile, D

2014-01-01

Hydatid disease is in a come-back period. In Romania the incidence is cited at 5-6 cases per 100,000 inhabitants. In this study we define the concept of multivisceral echinococcosis, which is a more serious form of the hydatid disease with implications of diagnosis, treatment,morbidity and mortality. Multivisceral echinococcosis must be differentiated from multiple echinococcosis. The latter is defined as the localization of multiple hydatid cysts in the same organ. In case of multiple echinococcosis, we can describe double echinococcosis (two hydatid cysts located in the same organ), triple, etc. The etiology of multivisceral echinococcosis is similar to mono-visceral echinococcosis.Regarding the pathogenic mechanism, we appreciate that there are two distinct mechanisms: primary infection (most of them) and secondary infection. We propose a classification of multivisceral echinococcosis based on the anatomical compartment involved.The diagnosis of this condition is easy to establish using classicor more recent investigations (CT, MRI). Compared to monovisceral echinococcosis, the symptomatology is louder because of the involvement of several organs and its association with different other conditions. We wish this study to bring more information about hydatid disease, but especially about multivisceral echinococcosis. Celsius.

Recurrence of cystic echinococcosis in an endemic area: a retrospective study.

PubMed

Velasco-Tirado, Virginia; Romero-Alegría, Ángela; Belhassen-García, Moncef; Alonso-Sardón, Montserrat; Esteban-Velasco, Carmen; López-Bernús, Amparo; Carpio-Perez, Adela; Jimenez López, Marcelo Fernando; Muñoz Bellido, Juan Luis; Muro, Antonio; Cordero-Sanchez, Miguel; Pardo-Lledias, Javier; Muñoz-Bellvis, Luis

2017-06-27

Cystic echinococcosis (CE) is a chronic, complex and neglected zoonotic disease. CE occurs worldwide. In humans, it may result in a wide spectrum of clinical manifestations, ranging from asymptomatic infection to fatal disease. Clinical management procedures have evolved over decades without adequate evaluation. Despite advances in surgical techniques and the use of chemotherapy, recurrence remains one of the major problems in the management of hydatid disease. The aim of this study was to determine the frequency of CE recurrence and the risk factors involved in recurrence. A descriptive longitudinal-retrospective study was designed. We reviewed all patients diagnosed with CE according to ICD-9 (code 122-0 to 122-9) criteria admitted at Complejo Asistencial Universitario de Salamanca, Spain, between January 1998 and December 2015. Among the 217 patients studied, 25 (11.5%) had a hydatid recurrence after curative intention treatment. Median duration of recurrence's diagnosis was 12.35 years (SD: ±9.31). The likelihood of recurrence was higher [OR = 2.7; 95% CI, 1.1-7.1; p < 0.05] when the cyst was located in organs other than liver and lung, 22.6% (7/31) vs 14.2% (31/217) in the cohort. We detected a chance of recurrence [OR = 2.3; 95% CI, 1.4-6.5; p > 0.05] that was two times higher in those patients treated with a combination of antihelminthic treatments and surgical intervention (20/141, 14.2%) than in patients treated with surgical intervention alone (5/76, 6.6%). Despite advances in diagnosis and therapeutic techniques in hydatid disease, recurrence remains one of the major problems in the management of hydatid disease. The current management and treatment of recurrences is still largely based on expert opinion and moderate-to-poor quality of evidence. Consequently, large prospective and multicenter studies will be needed to provide definitive recommendations for its clinical management.

Cystic echinococcosis in sheep in Sardinia. Changing pattern and present status.

PubMed

Conchedda, Margherita; Seu, Valter; Capra, Salvatore; Caredda, Alessia; Pani, Sergio Pino; Lochi, Pier Giorgio; Collu, Carlo; Mura, Angelo; Gabriele, Flavio

2012-04-01

The epidemiological status of cystic echinococcosis (CE) in sheep in Sardinia over the 20 years since the last control attempt at the end of the 1980s has been assessed, comparing the results of two surveys carried out in abattoirs in southern Sardinia. In the first, conducted in 1995-1997 (5-7 years after the last control effort), CE prevalence of about 75% was observed in the 1375 sheep sampled, with intensity of 10.0 and mean abundance of 7.5. The most affected organ was the liver, whereas a large percentage of infected animals presented cysts in both the liver and lung. Overall, about 26% of parasitized sheep were found to be heavily infected and 12.15% of infected animals harboured fertile cysts. In the second survey, carried out ten years after the first, during the period 2005-2010 in absence of specific control measures, a total of 1414 sheep were examined. CE prevalence was 65%, 78% in the most rural Oristano province and 58% in the most "urbanised" province of Cagliari. Frequency of sheep infected in both the liver and lung had decreased slightly compared to the first survey, particularly in the Cagliari province, as had intensity and mean abundance, though to a lesser extent in the Oristano province. 14.6% of parasitized sheep were heavily infected, showing a general decline over the 10 years between the two surveys, particularly in the Cagliari area where the figure had more than halved. Conversely, about 14% of infected sheep hosted at least 1 fertile cyst, a slight increase compared to the 1st survey. Finally, the trend of CE transmission in Sardinian sheep according to surveys carried out from 1952 to 2010 has been analysed and the results are discussed in the light of the significant socio-economic and structural transformations that have actually modified the zoonosis scenario on the island. Copyright © 2011 Elsevier B.V. All rights reserved.

The clinical burden of human cystic echinococcosis in Palestine, 2010-2015.

PubMed

Al-Jawabreh, Amer; Ereqat, Suheir; Dumaidi, Kamal; Nasereddin, Abdelmajeed; Al-Jawabreh, Hanan; Azmi, Kifaya; Al-Laham, Nahed; Nairat, Moath; Casulli, Adriano; Maqboul, Husni; Abdeen, Ziad

2017-07-01

Cystic echinococcosis (CE) is classified by the WHO as a neglected disease inflicting economic losses on the health systems of many countries worldwide. The aim of this case-series study was to investigate the burden of human CE in Palestine during the period between 2010 and 2015. Records of surgically confirmed CE patients from 13 public and private hospitals in the West Bank and Gaza Strip were reviewed. Patients' cysts were collected from surgical wards and formalin-fixed paraffin-embedded (FFPE) blocks were collected from histopathology departments. Molecular identification of CE species /genotypes was conducted by targeting a repeat DNA sequence (EgG1 Hae III) within Echinococcus nuclear genome and a fragment within the mitochondrial cytochrome c oxidase subunit 1, (CO1). Confirmation of CE species/genotypes was carried out using sequencing followed by BLAST analysis and the construction of maximum likelihood consensus dendrogram. CE cases were map-spotted and statistically significant foci identified by spatial analysis. A total of 353 CE patients were identified in 108 localities from the West Bank and Gaza Strip. The average surgical incidence in the West Bank was 2.1 per 100,000. Spot-mapping and purely spatial analysis showed 13 out of 16 Palestinian districts had cases of CE, of which 9 were in the West Bank and 4 in Gaza Strip. Al-Khalil and Bethlehem were statistically significant foci of CE in Palestine with a six-year average incidence of 4.2 and 3.7 per 100,000, respectively. To the best of our knowledge, this is the first confirmation of human CE causative agent in Palestine. This study revealed that E. granulosus sensu stricto (s.s.) was the predominating species responsible for CE in humans with 11 samples identified as G1 genotype and 2 as G3 genotype. This study emphasizes the need for a stringent surveillance system and risk assessment studies in the rural areas of high incidence as a prerequisite for control measures.

Human cystic echinococcosis in Morocco: Ultrasound screening in the Mid Atlas through an Italian-Moroccan partnership

PubMed Central

Chebli, Houda; Laamrani El Idrissi, Abderrhamane; Benazzouz, Mustapha; Lmimouni, Badre Eddine; Nhammi, Haddou; Elabandouni, Mourad; Youbi, Mohammed; Afifi, Rajaa; Tahiri, Sara; Essayd El Feydi, Abdellah; Settaf, Adbellatif; Tinelli, Carmine; De Silvestri, Annalisa; Bouhout, Souad; Abela-Ridder, Bernadette; Magnino, Simone; Brunetti, Enrico; Filice, Carlo

2017-01-01

Background Cystic echinococcosis (CE) is a neglected parasitic zoonosis with considerable socioeconomic impact on affected pastoral communities. CE is endemic throughout the Mediterranean, including Morocco, where the Mid Atlas is the most prevalent area for both human and animal infection. The highest hospital annual incidence of human CE is recorded in the provinces of Ifrane and El Hajeb. However, hospital-based statistics likely underestimate the real prevalence of infection, as a proportion of cases never reach medical attention or official records. Methodology/Principal findings In 2012, a project on clinical management of CE in Morocco was launched with the aims of estimating the prevalence of human abdominal CE in selected rural communes of the above mentioned provinces using ultrasound (US) screening and training local physicians to implement US-based focused assessment and rational clinical management of CE according to the WHO-IWGE Expert Consensus. A total of 5367 people received abdominal US during four campaigns in April-May 2014. During the campaigns, 24 local general practitioners received >24 hours of hands-on training and 143 health education sessions were organized for local communities. We found an overall CE prevalence of 1.9%, with significantly higher values in the rural communes of Ifrane than El Hajeb (2.6% vs 1.3%; p<0.001). CE cysts were predominantly in inactive stage, especially in older age groups. However, active cysts were present also in adults, indicating acquisition of infection at all ages. Province of residence was the only risk factor consistently associated with CE infection. Conclusions/Significance Our results show a high prevalence and on-going, likely environmental transmission of CE in the investigated provinces of Morocco, supporting the implementation of control activities in the area by national health authorities and encouraging the acceptance and divulgation of diagnosis and treatment algorithms based on imaging for

The disease burden of human cystic echinococcosis based on HDRs from 2001 to 2014 in Italy

PubMed Central

Brundu, Diego; Stegel, Giovanni; Loi, Federica; Rolesu, Sandro; Masu, Gabriella; Ledda, Salvatore; Masala, Giovanna

2017-01-01

Background Cystic echinococcosis (CE) is an important neglected zoonotic parasitic infection belonging to the subgroup of seven Neglected Zoonotic Disease (NZDs) included in the World Health Organization’s official list of 18 Neglected Tropical Diseases (NTDs). CE causes serious global human health concerns and leads to significant economic losses arising from the costs of medical treatment, morbidity, life impairments and fatality rates in human cases. Moreover, CE is endemic in several Italian Regions. The aim of this study is to perform a detailed analysis of the economic burden of hospitalization and treatment costs and to estimate the Disability Adjusted Life Years (DALYs) of CE in Italy. Methods and findings In the period from 2001 to 2014, the direct costs of 21,050 Hospital Discharge Records (HDRs) belonging to 12,619 patients with at least one CE-related diagnosis codes were analyzed in order to quantify the economic burden of CE. CE cases average per annum are 901 (min—max = 480–1,583). Direct costs include expenses for hospitalizations, medical and surgical treatment incurred by public and private hospitals and were computed on an individual basis according to Italian Health Ministry legislation. Moreover, we estimated the DALYs for each patient. The Italian financial burden of CE is around € 53 million; the national average economic burden per annum is around € 4 million; the DALYs of the population from 2001 to 2014 are 223.35 annually and 5.26 DALYs per 105 inhabitants. Conclusion In Italy, human CE is responsible for significant economic losses in the public health sector. In humans, costs associated with CE have been shown to have a great impact on affected individuals, their families and the community as a whole. This study could be used as a tool to prioritize and make decisions with regard to a surveillance system for this largely preventable yet neglected disease. It demonstrates the need of implementing a CE control program aimed at

Watch and Wait Management of Inactive Cystic Echinococcosis - Does the Path to Inactivity Matter - Analysis of a Prospective Patient Cohort.

PubMed

Stojkovic, Marija; Rosenberger, Kerstin Daniela; Steudle, Franziska; Junghanss, Thomas

2016-12-01

Overdiagnosis and overtreatment are rarely discussed in the context of NTDs despite their relevance for patients under the care of health services with limited resources where the risks of therapy induced complications are often disproportionate to the benefit. The advantages of cyst staging-based management of patients with cystic echinococcosis (CE) are not yet fully explored. Questions are: Do inactive cysts (CE 4 and CE 5) need treatment and is there a difference between cysts which reach CE4 and CE5 naturally or by benzimidazole therapy? Analysis of long-term follow-up data from a prospective CE patient cohort of 223 patients of a national clinical center for echinococcosis. The event of interest "relapse" was defined as the reversal of a cyst from an inactive stage (CE4, CE5) back to an active stage. The watch &wait (ww) group included 30 patients with 46 inactive cysts who never received medical treatment. The benzimidazole-treated (med) group included 15 patients with 17 cysts. There was no relapse in the ww-group whereas 8/17 cysts showed relapse within 18 months after treatment in the med-group. Loss to follow-up was 15.5%. Data from the watch & wait group impressively show how stable naturally inactivated cysts are in contrast to cysts which reach inactivity through treatment with benzimidazoles. A substantial proportion of patients can be spared from treatment through cyst staging. Cysts which inactivated through a natural course do not relapse with very high likelihood. We recommend follow up of 5 years to confirm the stability of the inactive stage. Cysts driven into inactivity through benzimidazole therapy instead need careful monitoring to identify those which reactivate (around 50% within 18 months). 5 years follow-up appears safe to make a final decision on the need for further monitoring.

Economic losses due to cystic echinococcosis in India: Need for urgent action to control the disease.

PubMed

Singh, Balbir B; Dhand, Navneet K; Ghatak, Sandeep; Gill, Jatinder P S

2014-01-01

Cystic ehinococcosis (CE) caused by Echinococcus granulosus remains a neglected zoonotic disease despite its considerable human and animal health concerns. This is the first systematic analysis of the livestock and human related economic losses due to cystic echinococcosis in India. Data about human cases were obtained from a tertiary hospital. Human hydatidosis cases with and without surgical interventions were extrapolated to be 5647 and 17075 per year assuming a total human population of 1210193422 in India. Data about prevalence of hydatid cysts in important food producing animals were obtained from previously published abattoir based epidemiological surveys that reported a prevalence of 5.39% in cattle, 4.36% in buffaloes, 3.09% in pigs, 2.23% in sheep and 0.41% in goats. Animal population data were sourced from the latest census conducted by the Department of Animal Husbandry, Dairying and Fisheries, India. Other input parameters were obtained from published scientific literature. Probability distributions were included for many input values to account for variability and uncertainty. Sensitivity analyses were conducted to evaluate the effect of important parameters on the estimated economic losses. The analysis revealed a total annual median loss of Rs. 11.47 billion (approx. US $ 212.35 million). Cattle and buffalo industry accounted for most of the losses: 93.05% and 88.88% of the animal and total losses, respectively. Human hydatidosis related losses were estimated to be Rs. 472.72 million (approx. US $ 8.75 million) but are likely to be an under-estimate due to under-reporting of the disease in the country. The human losses more than quadrupled to Rs. 1953 million i.e. approx. US $ 36.17 million, when the prevalence of human undiagnosed cases was increased to 0.2% in the sensitivity analyses. The social loss and psychological distress were not taken into account for calculating human loss. The results highlight an urgent need for a science based policy

Independent evaluation of a canine Echinococcosis Control Programme in Hobukesar County, Xinjiang, China

PubMed Central

van Kesteren, Freya; Qi, Xinwei; Tao, Jiang; Feng, Xiaohui; Mastin, Alexander; Craig, Philip S.; Vuitton, Dominique A.; Duan, Xinyu; Chu, Xiangdong; Zhu, Jinlong; Wen, Hao

2015-01-01

The Xinjiang Uyghur Autonomous Region in northwest China is one of the world's most important foci for cystic echinococcosis. Domestic dogs are the main source for human infection, and previous studies in Xinjiang have found a canine Echinococcus spp. coproELISA prevalence of between 36% and 41%. In 2010 the Chinese National Echinococcosis Control Programme was implemented in Xinjiang, and includes regular dosing of domestic dogs with praziquantel. Six communities in Hobukesar County, northwest Xinjiang were assessed in relation to the impact of this control programme through dog necropsies, dog Echinococcus spp. coproantigen surveys based on Lot Quality Assurance Sampling (LQAS) and dog owner questionnaires. We found that 42.1% of necropsied dogs were infected with Echinococcus granulosus, and coproELISA prevalences were between 15% and 70% in the communities. Although approximately half of all dog owners reported dosing their dogs within the 12 months prior to sampling, coproELISA prevalence remained high. Regular praziquantel dosing of owned dogs in remote and semi-nomadic communities such as those in Hobukesar County is logistically very difficult and additional measures should be considered to reduce canine echinococcosis. PMID:25661801

Independent evaluation of a canine Echinococcosis Control Programme in Hobukesar County, Xinjiang, China.

PubMed

van Kesteren, Freya; Qi, Xinwei; Tao, Jiang; Feng, Xiaohui; Mastin, Alexander; Craig, Philip S; Vuitton, Dominique A; Duan, Xinyu; Chu, Xiangdong; Zhu, Jinlong; Wen, Hao

2015-05-01

The Xinjiang Uyghur Autonomous Region in northwest China is one of the world's most important foci for cystic echinococcosis. Domestic dogs are the main source for human infection, and previous studies in Xinjiang have found a canine Echinococcus spp. coproELISA prevalence of between 36% and 41%. In 2010 the Chinese National Echinococcosis Control Programme was implemented in Xinjiang, and includes regular dosing of domestic dogs with praziquantel. Six communities in Hobukesar County, northwest Xinjiang were assessed in relation to the impact of this control programme through dog necropsies, dog Echinococcus spp. coproantigen surveys based on Lot Quality Assurance Sampling (LQAS) and dog owner questionnaires. We found that 42.1% of necropsied dogs were infected with Echinococcus granulosus, and coproELISA prevalences were between 15% and 70% in the communities. Although approximately half of all dog owners reported dosing their dogs within the 12 months prior to sampling, coproELISA prevalence remained high. Regular praziquantel dosing of owned dogs in remote and semi-nomadic communities such as those in Hobukesar County is logistically very difficult and additional measures should be considered to reduce canine echinococcosis. Copyright © 2015 The Authors. Published by Elsevier B.V. All rights reserved.

Cystic echinococcosis therapy: Albendazole-loaded lipid nanocapsules enhance the oral bioavailability and efficacy in experimentally infected mice.

PubMed

Pensel, Patricia E; Ullio Gamboa, Gabriela; Fabbri, Julia; Ceballos, Laura; Sanchez Bruni, Sergio; Alvarez, Luis I; Allemandi, Daniel; Benoit, Jean Pierre; Palma, Santiago D; Elissondo, María C

2015-12-01

Therapeutic failures attributed to medical management of cystic echinococcosis (CE) with albendazole (ABZ) have been primarily linked to the poor drug absorption rate resulting in low drug level in plasma and hydatid cysts. Lipid nanocapsules (LNCs) represent nanocarriers designed to encapsulate lipophilic drugs, such as ABZ. The goals of the current work were: (i) to characterize the plasma and cyst drug exposure after the administration of ABZ as ABZ-LNCs or ABZ suspension (ABZ-SUSP) in mice infected with Echinococcus granulosus, and ii) to compare the clinical efficacies of both ABZ formulations. Enhanced ABZ sulphoxide (ABZ-SO) concentration profiles were obtained in plasma and cysts from ABZ-LNC treated animals. ABZSO exposure (AUC0-LOQ) was significantly higher in plasma and cyst after the ABZ-LNC treatments, both orally and subcutaneously, compared to that observed after oral administration of ABZ-SUSP. Additionally, ABZSO concentrations measured in cysts from ABZ-LNC treated mice were 1.7-fold higher than those detected in plasma. This enhanced drug availability correlated with an increased efficacy against secondary CE in mice observed for the ABZ-LNCs, while ABZ-SUSP did not reach differences with the untreated control group. This new pharmacotechnically-based strategy could be a potential alternative to improve the treatment of human CE. Copyright © 2015 Elsevier B.V. All rights reserved.

Progress in the pharmacological treatment of human cystic and alveolar echinococcosis: Compounds and therapeutic targets

PubMed Central

Siles-Lucas, Mar; Casulli, Adriano; Cirilli, Roberto

2018-01-01

Human cystic and alveolar echinococcosis are helmintic zoonotic diseases caused by infections with the larval stages of the cestode parasites Echinococcus granulosus and E. multilocularis, respectively. Both diseases are progressive and chronic, and often fatal if left unattended for E. multilocularis. As a treatment approach, chemotherapy against these orphan and neglected diseases has been available for more than 40 years. However, drug options were limited to the benzimidazoles albendazole and mebendazole, the only chemical compounds currently licensed for treatment in humans. To compensate this therapeutic shortfall, new treatment alternatives are urgently needed, including the identification, development, and assessment of novel compound classes and drug targets. Here is presented a thorough overview of the range of compounds that have been tested against E. granulosus and E. multilocularis in recent years, including in vitro and in vivo data on their mode of action, dosage, administration regimen, therapeutic outcomes, and associated clinical symptoms. Drugs covered included albendazole, mebendazole, and other members of the benzimidazole family and their derivatives, including improved formulations and combined therapies with other biocidal agents. Chemically synthetized molecules previously known to be effective against other infectious and non-infectious conditions such as anti-virals, antibiotics, anti-parasites, anti-mycotics, and anti-neoplastics are addressed. In view of their increasing relevance, natural occurring compounds derived from plant and fungal extracts are also discussed. Special attention has been paid to the recent application of genomic science on drug discovery and clinical medicine, particularly through the identification of small inhibitor molecules tackling key metabolic enzymes or signalling pathways. PMID:29677189

Investigation of the Applicability of a Rapid Diagnosis Test in the Diagnosis of Cystic Echinococcosis.

PubMed

Ertuğ, Sema; Çalışkan, Serçin Özlem; Malatyalı, Erdoğan; Ertabaklar, Hatice

2018-05-21

Echinococcus granulosus, the etiological agent of cystic echinococcosis (CE) in humans and livestock, is a widely distributed zoonotic pathogen tapeworm. The infection is transmitted to humans by the ingestion of E. granulosus eggs released in the feces of definitive hosts such as dogs. The larval stage of the parasite develops a slowly enlarging cyst in the visceral organs, particularly in the liver and/or lung. The aim of the present study was to evaluate the diagnostic value of an immunochromatographic test (ICT) for CE. A total of 50 sera from surgically and/or pathologically confirmed patients with CE were included in the study as the study group; the control group comprised patients who tested negative for enzyme-linked immunosorbent assay (ELISA). Sera were selected from the collection at Adnan Menderes University, Faculty of Medicine, Parasitology Laboratory, by simple random sampling. The collection included sera obtained between 2010 and 2014; antibody titers of each serum sample were determined using in-house ELISA, before storage at -20°C. The presence of E. granulosus antibody in the sera was determined using a commercially available ICT (VIRAPID® HYDATIDOSIS) kit method. In the study group (E. granulosus-confirmed cases), two (4%) of the 50 sera were negative and 48 (96%) were positive with ICT. In the control group (ELISA-negative), all were negative with ICT. The rapid diagnostic test has been evaluated as a practical, easy-to-use method for detecting CE, and it can be used as a screening test in routine diagnosis and research.

Veterinary public health activities at FAO: echinococcosis/hydatid disease.

PubMed

Eddi, C; de Balogh, K; Lubroth, J; Amanfu, W; Speedy, A; Battaglia, D

2004-12-01

Cystic hydatidosis is a zoonotic disease that remain as a significant cause of human morbidity and mortality in many parts of the world. The disease has veterinary public health implications. FAO is involved with some activities in the control of echinococcosis/hydatid disease: within the Animal Production and Health Division the Veterinary Public Health (VHP) Programme is constituted by members of the different Services (Animal Health, Animal Production, and Livestock Policy) within the Division. FAO regular programme has also established a global network of professionals directly involved in VPH. Furthermore FAO's Technical Cooperation Projects (TCP) is a tool to assist member countries in responding to urgent and unforeseen demands.

Cystic echinococcosis in children and adults: a seven-year comparative study in western Romania.

PubMed

Vlad, Daliborca Cristina; Neghina, Adriana Maria; Dumitrascu, Victor; Marincu, Iosif; Neghina, Raul; Calma, Crenguta Livia

2013-02-01

This study aims to compare the epidemiological, clinical, and laboratory aspects of cystic echinococcosis (CE) in children with those of adults from western Romania, and to determine the specifics of this disease in the younger population. The medical records of the patients diagnosed with CE and hospitalized during 2004-2010 in four western Romanian counties were retrospectively investigated. Overall, 82 children (mean age, 10.8 years) and 369 adults (mean age, 48 years) were diagnosed with CE during the study period. The yearly detection rate was 3.1/100,000 children and 3.4/100,000 adults. The incidence rate registered a peak in children aged 5-14 years (4.4/100,000) and adults aged 60-69 years (5/100,000). Female to male ratio was 1.6:1 in pediatric cases and 1.2:1 in adults. CE was almost twice more frequent in rural areas (4.4/100,000 children and 4.9/100,000 adults) as compared to urban regions (2.3/100,000 children and 2.6/100,000 adults). Hepatic cysts were found more frequently than pulmonary cysts, at a ratio of 3.6:1 in children and 12.8:1 in adults. Most of the cases had no complications (91.5% of children and 87% of adults). Children had higher eosinophil count (7%) and erythrocyte sedimentation rate (ESR) (29.7 mm/h) as compared with adults (eosinophil count, 5.3%, p<0.001; ESR, 22.4 mm/h, p=0.001). The active transmission of the parasite in western Romania is demonstrated by the high incidence of CE in children. Long-term prevention of this disease may be reached through joined legislative efforts focused on the registration of dogs, home slaughter of sheep and swine, veterinary control in abattoirs, destruction of infected materials, and restriction of dogs' access to vegetable gardens and children's parks.

Comparison of the Diagnostic Accuracy of Three Rapid Tests for the Serodiagnosis of Hepatic Cystic Echinococcosis in Humans

PubMed Central

Tamarozzi, Francesca; Covini, Ilaria; Mariconti, Mara; Narra, Roberta; Tinelli, Carmine; De Silvestri, Annalisa; Manzoni, Federica; Casulli, Adriano; Ito, Akira; Neumayr, Andreas; Brunetti, Enrico

2016-01-01

Background The diagnosis of cystic echinococcosis (CE) is based primarily on imaging, in particular with ultrasound for abdominal CE, complemented by serology when imaging results are unclear. In rural endemic areas, where expertise in ultrasound may be scant and conventional serology techniques are unavailable due to lack of laboratory equipment, Rapid Diagnostic Tests (RDTs) are appealing. Methodology/Principal Findings We evaluated the diagnostic accuracy of 3 commercial RDTs for the diagnosis of hepatic CE. Sera from 59 patients with single hepatic CE cysts in well-defined ultrasound stages (gold standard) and 25 patients with non-parasitic cysts were analyzed by RDTs VIRapid HYDATIDOSIS (Vircell, Spain), Echinococcus DIGFA (Unibiotest, China), ADAMU-CE (ICST, Japan), and by RIDASCREEN Echinococcus IgG ELISA (R-Biopharm, Germany). Sensitivity, specificity and ROC curves were compared with McNemar and t-test. For VIRapid and DIGFA, correlation between semiquantitative results and ELISA OD values were evaluated by Spearman’s coefficient. Reproducibility was assessed on 16 randomly selected sera with Cohen’s Kappa coefficient. Sensitivity and Specificity of VIRapid (74%, 96%) and ADAMU-CE (57%, 100%) did not differ from ELISA (69%, 96%) while DIGFA (72%, 72%) did (p = 0.045). ADAMU-CE was significantly less sensitive in the diagnosis of active cysts (p = 0.019) while DIGFA was significantly less specific (p = 0.014) compared to ELISA. All tests were poorly sensitive in diagnosing inactive cysts (33.3% ELISA and ADAMU-CE, 42.8% DIGFA, 47.6% VIRapid). The reproducibility of all RDTs was good-very good. Band intensity of VIRapid and DIGFA correlated with ELISA OD values (r = 0.76 and r = 0.79 respectively, p<0.001). Conclusions/Significance RDTs may be useful in resource-poor settings to complement ultrasound diagnosis of CE in uncertain cases. VIRapid test appears to perform best among the examined kits, but all tests are poorly sensitive in the presence of

Cystic echinococcosis in children - the seventeen-year experience of two large medical centers in Serbia.

PubMed

Djuricic, Slavisa M; Grebeldinger, Slobodan; Kafka, Dejan I; Djan, Igor; Vukadin, Miroslav; Vasiljevic, Zorica V

2010-06-01

Cystic echinococcosis (CE) is a public health problem in countries having such endemic areas. Epidemiological studies of CE, especially pediatric, are rare. The aim of this study was to evaluate epidemiological and clinical characteristics of CE in children in Serbia. Data were obtained retrospectively from the case records of patients under the age of 18 years admitted for surgical treatment of CE at two large pediatric medical institutions in the period 1990-2006. Patients' age, number of cysts and their anatomic location were evaluated in relation to differences by patients' gender and socio-geographic status (urban or rural origin). The study included 149 children with 272 hydatid cysts. The mean age of patients was 10.1+/-3.8 years. There were no significant differences in the number of patients in relation to gender and urban:rural origin. There were no significant differences in patients' age at the time of surgery or the number of cysts per patient when patients' gender or socio-geographic status was evaluated. The anatomic location of cysts was as follows: liver (N=165; 60.7%), lungs (N=82; 30.1%), and other locations (N=25; 9.2%). Multiple cysts, and combined liver/lung involvement were identified in 34.2% (N=51), and 6.0% (N=9) of patients, respectively. Hepatic cysts were significantly more common in girls than in boys. There were no significant differences in anatomic location of cysts between socio-geographic groups. The large number of infected children during a long period of investigation indicates an active transmission of disease and a lack of program for control and prevention of CE in Serbia.

Review of risk factors for human echinococcosis prevalence on the Qinghai-Tibet Plateau, China: a prospective for control options

PubMed Central

2014-01-01

Objective Echinococcosis is a major parasitic zoonosis of public health importance in western China. In 2004, the Chinese Ministry of Health estimated that 380,000 people had the disease in the region. The Qinghai-Tibet Plateau is highly co-endemic with both alveolar echinococcosis (AE) and cystic echinococcosis (CE). In the past years, the Chinese government has been increasing the financial support to control the diseases in this region. Therefore, it is very important to identify the significant risk factors of the diseases by reviewing studies done in the region in the past decade to help policymakers design appropriate control strategies. Review Selection criteria for which literature to review were firstly defined. Medline, CNKI (China National Knowledge Infrastructure), and Google Scholar were systematically searched for literature published between January 2000 and July 2011. Significant risk factors found by single factor and/or multiple factors analysis were listed, counted, and summarized. Literature was examined to check the comparability of the data; age and sex specific prevalence with same data structures were merged and used for further analysis. A variety of assumed social, economical, behavioral, and ecological risk factors were studied on the Plateau. Those most at risk were Tibetan herdsmen, the old and female in particular. By analyzing merged comparable data, it was found that females had a significant higher prevalence, and a positive linearity relationship existed between echinococcosis prevalence and increasing age. In terms of behavioral risk factors, playing with dogs was mostly correlated with CE and/or AE prevalence. In terms of hygiene, employing ground water as the drinking water source was significantly correlated with CE and AE prevalence. For definitive hosts, dog related factors were most frequently identified with prevalence of CE or/and AE; fox was a potential risk factor for AE prevalence only. Overgrazing and deforestation

Review of risk factors for human echinococcosis prevalence on the Qinghai-Tibet Plateau, China: a prospective for control options.

PubMed

Wang, Qian; Huang, Yan; Huang, Liang; Yu, Wenjie; He, Wei; Zhong, Bo; Li, Wei; Zeng, Xiangman; Vuitton, Dominique A; Giraudoux, Patrick; Craig, Philip S; Wu, Weiping

2014-01-29

Echinococcosis is a major parasitic zoonosis of public health importance in western China. In 2004, the Chinese Ministry of Health estimated that 380,000 people had the disease in the region. The Qinghai-Tibet Plateau is highly co-endemic with both alveolar echinococcosis (AE) and cystic echinococcosis (CE). In the past years, the Chinese government has been increasing the financial support to control the diseases in this region. Therefore, it is very important to identify the significant risk factors of the diseases by reviewing studies done in the region in the past decade to help policymakers design appropriate control strategies. Selection criteria for which literature to review were firstly defined. Medline, CNKI (China National Knowledge Infrastructure), and Google Scholar were systematically searched for literature published between January 2000 and July 2011. Significant risk factors found by single factor and/or multiple factors analysis were listed, counted, and summarized. Literature was examined to check the comparability of the data; age and sex specific prevalence with same data structures were merged and used for further analysis.A variety of assumed social, economical, behavioral, and ecological risk factors were studied on the Plateau. Those most at risk were Tibetan herdsmen, the old and female in particular. By analyzing merged comparable data, it was found that females had a significant higher prevalence, and a positive linearity relationship existed between echinococcosis prevalence and increasing age. In terms of behavioral risk factors, playing with dogs was mostly correlated with CE and/or AE prevalence. In terms of hygiene, employing ground water as the drinking water source was significantly correlated with CE and AE prevalence. For definitive hosts, dog related factors were most frequently identified with prevalence of CE or/and AE; fox was a potential risk factor for AE prevalence only. Overgrazing and deforestation were significant for

Biological, Epidemiological, and Clinical Aspects of Echinococcosis, a Zoonosis of Increasing Concern

PubMed Central

Eckert, Johannes; Deplazes, Peter

2004-01-01

Echinococcosis in humans is a zoonotic infection caused by larval stages (metacestodes) of cestode species of the genus Echinococcus. Cystic echinococcosis (CE) is caused by Echinococcus granulosus, alveolar echinococcosis (AE) is caused by E. multilocularis, and polycystic forms are caused by either E. vogeli or E. oligarthrus. In untreated cases, AE has a high mortality rate. Although control is essentially feasible, CE remains a considerable health problem in many regions of the northern and southern hemispheres. AE is restricted to the northern hemisphere regions of North America and Eurasia. Recent studies have shown that E. multilocularis, the causative agent of AE, is more widely distributed than previously thought. There are also some hints of an increasing significance of polycystic forms of the disease, which are restricted to Central and South America. Various aspects of human echinococcosis are discussed in this review, including data on the infectivity of genetic variants of E. granulosus to humans, the increasing invasion of cities in Europe and Japan by red foxes, the main definitive hosts of E. multilocularis, and the first demonstration of urban cycles of the parasite. Examples of emergence or reemergence of CE are presented, and the question of potential spreading of E. multilocularis is critically assessed. Furthermore, information is presented on new and improved tools for diagnosing the infection in final hosts (dogs, foxes, and cats) by coproantigen or DNA detection and the application of molecular techniques to epidemiological studies. In the clinical field, the available methods for diagnosing human CE and AE are described and the treatment options are summarized. The development of new chemotherapeutic options for all forms of human echinococcosis remains an urgent requirement. A new option for the control of E. granulosus in the intermediate host population (mainly sheep and cattle) is vaccination. Attempts are made to reduce the

Echinococcosis

MedlinePlus

... is a microscopic tapeworm that is found in dogs, sheep, other livestock, and wild animals. The tapeworm ... humans are in close contact with livestock or dogs. In the United States, echinococcosis mostly is found ...

Prevalence and Risk Factors for Human Cystic Echinococcosis in the Cusco Region of the Peruvian Highlands Diagnosed Using Focused Abdominal Ultrasound

PubMed Central

Tamarozzi, Francesca; Hou, Amy; Morales, Maria Luisa; Giordani, Maria Teresa; Vilca, Freddy; Mozo, Karen; Bascope, Ruben; White, A. Clinton; Brunetti, Enrico; Chen, Lin; Cabada, Miguel M.

2017-01-01

Latin America is among the highly endemic regions for cystic echinococcosis (CE). In Peru, an estimated 1,139 disability-adjusted life years are lost annually from surgical treatment of CE. This is comparable with the combined total for Argentina, Brazil, Uruguay, and Chile. The prevalence of human infection has been investigated in the central Peruvian Andes, but there are no community-based screening data from other regions of Peru. We carried out a population survey in January 2015 using abdominal ultrasound to estimate the prevalence of abdominal CE in the Canas and Canchis provinces, in the Cusco region of Peru. Among 1,351 subjects screened, 41 (3%) had CE. There was significant variation between communities with similar socioeconomic features in a small geographical area. A history of CE was reported by 4.1% of the screened subjects, among whom 30.3% still had CE on ultrasound. Among patients reporting previous CE treatment, 14.9% had CE in active stages. Limited education, community of residence, and knowing people with CE in the community were associated with CE. These results demonstrate a significant burden of CE in the region and suggest the need for further investigations, control activities, and optimization of clinical management for CE in this area. PMID:28719254

Recurrent pulmonary embolism due to echinococcosis secondary to hepatic surgery for hydatid cysts.

PubMed

Damiani, Mario Francesco; Carratù, Pierluigi; Tatò, Ilaria; Vizzino, Heleanna; Florio, Carlo; Resta, Onofrio

2012-01-01

We describe the case of a 53-year-old man with recurrent pulmonary embolism due to intra-arterial cysts from Echinococcus. Both the patient's medical history and the computed tomographic (CT) scan abnormalities led to the diagnosis. The CT scan, performed during hospitalization in our ward, showed cystic masses in the left main pulmonary artery and in the descending branch of the right pulmonary artery. Within cystic masses, thin septa were visible, giving a chambered appearance, which was suggestive of a group of daughter cysts. In the past, our patient underwent multiple operations for recurring echinococcal cysts of the liver. After the last intervention, 4 years earlier, his postoperative course was complicated by pulmonary embolism: a CT scan showed a filling defect in the descending branch of the right pulmonary artery, which was caused by the same cystic mass as 4 years later, although smaller. This mass, not properly treated, increased in diameter. Moreover, after 4 years, there has been a new episode of embolism, which involved the left main pulmonary artery. This is the first case in which there are repeated episodes of pulmonary embolism echinococcosis after hepatic surgery for removal of hydatid cysts.

Emergence of polycystic neotropical echinococcosis.

PubMed

Tappe, Dennis; Stich, August; Frosch, Matthias

2008-02-01

Echinococcosis is a parasitic zoonosis of increasing concern. In 1903, the first cases of human polycystic echinococcosis, a disease resembling alveolar echinococcosis, emerged in Argentina. One of the parasites responsible, Echinococcus oligarthrus, had been discovered in its adult strobilar stage before 1850. However, >100 years passed from the first description of the adult parasite to the recognition that this species is responsible for some cases of human neotropical polycystic echinococcosis and the elucidation of the parasite's life cycle. A second South American species, E. vogeli, was described in 1972. Obtaining recognition of the 2 species and establishing their connection to human disease were complicated because the life cycle of tapeworms is complex and comprises different developmental stages in diverse host species. To date, at least 106 human cases have been reported from 12 South and Central American countries.

Emergence of Polycystic Neotropical Echinococcosis

PubMed Central

Stich, August; Frosch, Matthias

2008-01-01

Echinococcosis is a parasitic zoonosis of increasing concern. In 1903, the first cases of human polycystic echinococcosis, a disease resembling alveolar echinococcosis, emerged in Argentina. One of the parasites responsible, Echinococcus oligarthrus, had been discovered in its adult strobilar stage before 1850. However, >100 years passed from the first description of the adult parasite to the recognition that this species is responsible for some cases of human neotropical polycystic echinococcosis and the elucidation of the parasite’s life cycle. A second South American species, E. vogeli, was described in 1972. Obtaining recognition of the 2 species and establishing their connection to human disease were complicated because the life cycle of tapeworms is complex and comprises different developmental stages in diverse host species. To date, at least 106 human cases have been reported from 12 South and Central American countries. PMID:18258123

Human cystic echinococcosis in Morocco: Ultrasound screening in the Mid Atlas through an Italian-Moroccan partnership.

PubMed

Chebli, Houda; Laamrani El Idrissi, Abderrhamane; Benazzouz, Mustapha; Lmimouni, Badre Eddine; Nhammi, Haddou; Elabandouni, Mourad; Youbi, Mohammed; Afifi, Rajaa; Tahiri, Sara; Essayd El Feydi, Abdellah; Settaf, Adbellatif; Tinelli, Carmine; De Silvestri, Annalisa; Bouhout, Souad; Abela-Ridder, Bernadette; Magnino, Simone; Brunetti, Enrico; Filice, Carlo; Tamarozzi, Francesca

2017-03-01

Cystic echinococcosis (CE) is a neglected parasitic zoonosis with considerable socioeconomic impact on affected pastoral communities. CE is endemic throughout the Mediterranean, including Morocco, where the Mid Atlas is the most prevalent area for both human and animal infection. The highest hospital annual incidence of human CE is recorded in the provinces of Ifrane and El Hajeb. However, hospital-based statistics likely underestimate the real prevalence of infection, as a proportion of cases never reach medical attention or official records. In 2012, a project on clinical management of CE in Morocco was launched with the aims of estimating the prevalence of human abdominal CE in selected rural communes of the above mentioned provinces using ultrasound (US) screening and training local physicians to implement US-based focused assessment and rational clinical management of CE according to the WHO-IWGE Expert Consensus. A total of 5367 people received abdominal US during four campaigns in April-May 2014. During the campaigns, 24 local general practitioners received >24 hours of hands-on training and 143 health education sessions were organized for local communities. We found an overall CE prevalence of 1.9%, with significantly higher values in the rural communes of Ifrane than El Hajeb (2.6% vs 1.3%; p<0.001). CE cysts were predominantly in inactive stage, especially in older age groups. However, active cysts were present also in adults, indicating acquisition of infection at all ages. Province of residence was the only risk factor consistently associated with CE infection. Our results show a high prevalence and on-going, likely environmental transmission of CE in the investigated provinces of Morocco, supporting the implementation of control activities in the area by national health authorities and encouraging the acceptance and divulgation of diagnosis and treatment algorithms based on imaging for CE at both national and local level.

Epidemiology and control of echinococcosis in central Asia, with particular reference to the People's Republic of China.

PubMed

Zhang, Wenbao; Zhang, Zhuangzhi; Wu, Weiping; Shi, Baoxin; Li, Jun; Zhou, Xiaonong; Wen, Hao; McManus, Donald P

2015-01-01

At least 270 million people (58% of the total population) are at risk of cystic echinococcosis (CE) in Central Asia including areas of Mongolia, Kazakhstan, Kyrgyzstan, Tajikistan, Turkmenistan, Uzbekistan, Afghanistan, Iran, Pakistan and western China. The annual surgical incidence rate in Uzbekistan and Tadjikistan has been estimated to be as high as 25-27 cases/100,000 with the highest prevalence reaching 10% (range from 0.8 to 11.9%) in some Tibetan communities in western China. Echinococcus transmission in the region is largely associated with social factors including limited community knowledge of echinococcosis, small-scale household animal production, home killing of livestock, and the feeding of dogs with uncooked offal. Alveolar echinococcosis (AE) is also endemic in Central Asia and is recognized as a major problem in some Tibetan communities with up to 6% of villagers infected in some villages. In western China, 5-30% of the population are seropositive against E. granulosus antigens, indicating that a large number of individuals have been exposed to the parasite. Although echinococcosis control programs have been initiated in some countries in Central Asia, control efforts are generally fragmented and uncoordinated. Monthly deworming of dogs with praziquantel (PZQ), as a key measure to control the Echinococcus parasites, has been used in western China. However, the approach has proven difficult in local semi-nomadic communities. Additional control measures including health education, domestic livestock animal treatment/vaccination and dog vaccination are needed in CE-endemic areas to accelerate progress. Copyright © 2014. Published by Elsevier B.V.

A community-based study to examine the epidemiology of human cystic echinococcosis in Rio Negro Province, Argentina.

PubMed

Bingham, Glenda M; Budke, Christine M; Larrieu, Edmundo; Del Carpio, Mario; Mujica, Guillermo; Slater, Margaret R; Moguillansky, Sergio

2014-08-01

Although cystic echinococcosis (CE) is an important public health problem in Rio Negro Province, current epidemiological data for CE, in this region of Argentina, are not available. Therefore, a community-based study, which incorporated diagnostic imaging and a questionnaire, was conducted in Ingeniero Jacobacci, a small town in southern Rio Negro Province. This study sought to assess the prevalence of human CE, in the study population, and to evaluate epidemiologic factors associated with CE transmission within the study area. Of the 560 individuals who volunteered to participate in the study, 189 (34%) were children and 371 (66%) were adults. All study participants were screened for CE using abdominal ultrasound scanning, with CE-positive or suspect individuals also receiving thoracic radiographs. The overall prevalence of CE was 7.1% (40/560), with 1.6% (3/189) of children, and 10% (37/371) of adults diagnosed as CE-positive. Although 92.5% (37/40) of the CE-positive individuals had only hepatic lesions, two participants had both hepatic and pulmonary lesions, and one participant had a single renal lesion. Approximately 92% (340/371) of the adult study participants completed the questionnaire, which was used to identify factors associated with an increased risk for human infection. Age, level of education, dog ownership, and contact with sheep were found to be significantly associated with CE status. This study demonstrated that CE continues to be highly endemic in this region of Rio Negro Province, Argentina. In addition, community-based ultrasound screening surveys are a noninvasive, effective approach to case detection at the community level. Copyright © 2014 Elsevier B.V. All rights reserved.

Comparison of the serological tests ICT and ELISA for the diagnosis of alveolar echinococcosis in France☆

PubMed Central

Knapp, Jenny; Sako, Yasuhito; Grenouillet, Frédéric; Bresson-Hadni, Solange; Richou, Carine; Gbaguidi-Haore, Houssein; Ito, Akira; Millon, Laurence

2014-01-01

Serological diagnosis of alveolar echinococcosis (AE) is a key element for efficient patient treatment management. A rapid immunochromatography test kit (ICT) using the recombinant Em18 antigen (rEm18) was recently developed. The aim of our study was to assess this test on a panel of sera from French patients with alveolar echinococcosis and control patients. In a blind test, a total of 112 serum samples were tested including samples of AE (n = 30), cystic echinococcosis [CE] (n = 15), and polycystic echinococcosis [PE] (n = 1). For the comparison, 66 sera from patients with hepatocarcinoma, fascioliasis, toxocariasis, Caroli’s disease, or autoimmune chronic active hepatitis were used. The diagnostic test sets we used were the rEm18-ICT and two validated ELISAs with rEm18 and Em2-Em18 antigens, respectively. For the ICT, 27/30 sera from AE patients, 4/15 sera from CE patients and the PE patient serum were positive. One serum from the control panel (toxocariasis) was positive for the ICT. The rEm18-ICT sensitivity (90.0%) and specificity (92.7%) for detection of Em18-specific antibodies confirmed it as a relevant tool for AE diagnosis. The rEm18-ELISA had a sensitivity of 86.7% and specificity of 91.5%, and the Em2-Em18-ELISA had a sensitivity of 96.7% and specificity of 87.8%. However, when AE patient sera are recorded as weak in intensity with the ICT, we recommend a double reading and use of a reference sample if the ICT is used for patient follow-up. PMID:25058754

Comparison of the serological tests ICT and ELISA for the diagnosis of alveolar echinococcosis in France.

PubMed

Knapp, Jenny; Sako, Yasuhito; Grenouillet, Frédéric; Bresson-Hadni, Solange; Richou, Carine; Gbaguidi-Haore, Houssein; Ito, Akira; Millon, Laurence

2014-01-01

Serological diagnosis of alveolar echinococcosis (AE) is a key element for efficient patient treatment management. A rapid immunochromatography test kit (ICT) using the recombinant Em18 antigen (rEm18) was recently developed. The aim of our study was to assess this test on a panel of sera from French patients with alveolar echinococcosis and control patients. In a blind test, a total of 112 serum samples were tested including samples of AE (n = 30), cystic echinococcosis [CE] (n = 15), and polycystic echinococcosis [PE] (n = 1). For the comparison, 66 sera from patients with hepatocarcinoma, fascioliasis, toxocariasis, Caroli's disease, or autoimmune chronic active hepatitis were used. The diagnostic test sets we used were the rEm18-ICT and two validated ELISAs with rEm18 and Em2-Em18 antigens, respectively. For the ICT, 27/30 sera from AE patients, 4/15 sera from CE patients and the PE patient serum were positive. One serum from the control panel (toxocariasis) was positive for the ICT. The rEm18-ICT sensitivity (90.0%) and specificity (92.7%) for detection of Em18-specific antibodies confirmed it as a relevant tool for AE diagnosis. The rEm18-ELISA had a sensitivity of 86.7% and specificity of 91.5%, and the Em2-Em18-ELISA had a sensitivity of 96.7% and specificity of 87.8%. However, when AE patient sera are recorded as weak in intensity with the ICT, we recommend a double reading and use of a reference sample if the ICT is used for patient follow-up. © J. Knapp et al., published by EDP Sciences, 2014.

Abattoir based surveillance of cystic echinococcosis (CE) in the Sultanate of Oman during 2010-2013.

PubMed

Al Kitani, Fadya Abdullah; Al Riyami, Shumoos; Al Yahyai, Sabra; Al Rawahi, Abdulmajeed Hamood; Al Maawali, Mahir; Al Awahi, Abdulmajeed Hamood; Al Aawali, Mahir; Hussain, Muhammad Hammad

2015-07-30

A passive surveillance was conducted in 8 selected abattoirs belonging to 8 governorates of Oman. Routine slaughtering of 282.020 animals was observed during 2010-2013 by duty veterinarians and suspected samples (n=1440) for cystic echinococcosis (CE) from camels (n=292), cattle (n=303), goats (n=682) and sheep (n=163) were collected. In total, 587 (40.8%) samples were found positive for CE and the highest percentage (64.2%) of positive samples belonged to Dhofar (Salalah) as compared to other governorates, p<0.01. The infection rate of CE was calculated as 5.3, 0.6, 0.07 and 0.03 in camel, cattle, sheep and goats respectively. Fertile cysts were found in 30.7% of positive samples and camels (52%) were found as major source of these followed by cattle (14.1%) and goats (9.7%). Percentage of positive to submitted samples was higher in females (67.9%) than males (29.8%). The fertility of cyst increased with age and the highest percentage of fertile cysts were collected from animals above 5 years of age (54.4%) followed by between 3 and 5 years (36.7%) and below 3 years of age (8.9%). For different breeds, 478 (62.7%) samples from local breeds of livestock were positive for CE as compared to 108 (15.9%) from imported animals (OR 8.92), p<0.01. The highest percent of positive samples was observed in cysts from lungs (54.4%) followed by those from liver (25.7%), liver and lungs (14.1%) and others (3.8%). Molecular investigations indicated that G1 and G6/7 were the two strains of CE circulating in camels and cattle while G1 was the only strain found in samples from small ruminants. Results indicated that CE is endemic in the livestock of Oman and further molecular epidemiological work in different host species is required for the conceptualization of a comprehensive control program. Copyright © 2015 Elsevier B.V. All rights reserved.

Cystic echinococcosis in Poland: genetic variability and the first record of Echinococcus granulosus sensu stricto (G1 genotype) in the country.

PubMed

Sałamatin, Rusłan; Kowal, Jerzy; Nosal, Paweł; Kornaś, Sławomir; Cielecka, Danuta; Jańczak, Dawid; Patkowski, Waldemar; Gawor, Jakub; Kornyushin, Vadim; Golab, Elzbieta; Šnábel, Viliam

2017-11-01

Cystic echinococcosis is one of the most important zoonotic diseases affecting humans and livestock worldwide, and is endemic in Poland. A set of six isolates on larval stages of Echinococcus granulosus sensu lato tapeworms collected from three humans, two pigs and one sheep from Polish foci of CE was examined by DNA sequencing of two mitochondrial genes (cox1, rrnS). The results demonstrated the presence of E. canadensis and E. granulosus sensu stricto in the investigated hydatid cysts. The former species was found in all five isolates from pigs and humans derived from central Poland. In a sheep hydatid cyst originating from Lesser Poland Voivodeship in southern Poland, E. granulosus s. s. (G1 genotype) was identified. This is the first report of an unambiguously autochthonous infection with E. granulosus s. s. in Poland. The global distribution and host affiliations of the commonly occurring G1 microvariant with nucleotide change 56C/T in cox1, detected here in Polish sheep, are discussed. The finding that sheep harboured E. granulosus s. s. may have important consequences for developing effective hydatid control programmes in Poland due to its longer maturation rate in dogs compared with E. canadensis G7. This may lead to greater expenditures for purchasing anthelmintics to provide an appropriate dosing regime in sheep-raising areas of the country.

Diagnosis of cystic fibrosis in a patient of Egyptian background.

PubMed

Yoshida, Rie; Ruge, Bobby

2016-10-20

Cystic fibrosis (CF) is the most common genetically inherited condition in European-derived populations. However, it is being increasingly recognised in other populations, including people of Asian, Black African and Caribbean descent. We present a case detailing the diagnosis of CF in a 12-year-old patient of Egyptian background who had been treated for difficult asthma. In doing so, we aim to highlight the importance of considering CF in all patients, regardless of ethnicity. 2016 BMJ Publishing Group Ltd.

Comparative Evaluation of Liposomal Albendazole and Tablet-Albendazole Against Hepatic Cystic Echinococcosis: A Non-Randomized Clinical Trial.

PubMed

Li, Haitao; Song, Tao; Shao, Yingmei; Aili, Tuergan; Ahan, Ayifuhan; Wen, Hao

2016-01-01

In this study, we aimed to compare the clinical efficacy of liposomal albendazole (L-ABZ) and tablet-albendazole (T-ABZ) for the treatment of human hepatic cystic echinococcosis (CE). Sixty patients with single cyst (CE1) or daughter cyst (CE2) were included in this study and were nonrandomly divided into the L-ABZ group (n = 30, 10  mg/kg per day, p.o., b.i.d.) and T-ABZ group (n = 30, 12-20 mg/kg per day, p.o., b.i.d.), respectively. The treatment duration lasted for 6 months, during which dynamic follow-up was carried out to evaluate the clinical efficacy through calculating the total effective rates (TERs). Measurement data and numerous data were analyzed by the chi-square test. Two-sided tests were performed for all the statistical tests. In our study, 2 patients were lost in the follow-up in the L-ABZ group. One patient was lost in the follow-up in the T-ABZ group, and 1 patient was withdrawal from the study due to receiving surgery. Significant difference was identified in the 3-month TERs of L-ABZ group and T-ABZ group (33.3% vs 76.7%, P < 0.05). Also, remarkable difference was noted in the 6-month TERs in the L-ABZ group and T-ABZ group (66.7% vs 93.3%, P = 0.01). No statistical difference was noticed in the incidence rate of adverse reactions in both groups (P > 0.05). Based on our study, both T-ABZ and L-ABZ are effective for treating human CE. The TER in the L-ABZ group is superior to that of T-ABZ.

Knowledge, attitudes, and practices regarding cystic echinococcosis and sheep herding in Peru: a mixed-methods approach.

PubMed

Merino, Veronika; Westgard, Christopher M; Bayer, Angela M; García, Patricia J

2017-07-06

The parasitic disease, cystic echinococcosis (CE), is prevalent in low-income, livestock-raising communities and 2000 new people will be diagnosed this year in South America alone. The disease usually passes from livestock to dogs to humans, making it a zoonotic disease and part of the One Health Initiative. Control of CE has been infamously difficult; no endemic areas of South America have succeeded in maintaining sustainable eradication of the parasite. For the current study, we aimed to gain a better understanding of the knowledge, attitudes, and practices of rural sheep farmers and other community leaders regarding their sheep herding practices and perspectives about a control program for CE. We also hope to identify potential barriers and opportunities that could occur in a control program. The authors conducted Knowledge, Attitude and Practices (KAP) surveys and semi-structured interviews in rural communities in the highlands of Peru. The KAP surveys were administered to 51 local shepherds, and the semi-structured interviews were administered to 40 individuals, including shepherds, community leaders, and health care providers. We found that the shepherds already deworm their sheep at a median of 2 times per year (N = 49, range 2-4) and have a mean willingness-to-pay of U.S. $ 0.60 for dog dewormer medication (N = 20, range = 0.00- $2.00 USD). We were not able to learn the deworming agent or agents that were being used, for neither sheep nor dogs. Additionally, 90% of shepherds slaughter their own sheep (N = 49). We also learned that the main barriers to an effective control program include: lack of education about the cause and control options for CE, accessibility to the distant communities and sparse grazing pastures, and a lack of economic incentive. Findings suggest it may be feasible to develop an effective CE control program which can be used to create an improved protocol to control CE in the region.

Ovine echinococcosis I. Immunological diagnosis by enzyme immunoassay.

PubMed

Gatti, Antonio; Alvarez, Angela Rosa; Araya, Daniel; Mancini, Sergio; Herrero, Eduardo; Santillan, Graciela; Larrieu, Edmundo

2007-01-31

Immunodiagnosis in sheep presents problems of sensitivity and specificity, limiting its applicability in surveillance systems. The objective of this study was to develop a sensitive, specific and accessible technique for diagnosing cystic echinococcosis in naturally infected sheep and to evaluate the validity of necropsy as a reference test. A total of 247 sheep were studied at slaughterhouses, confirming the parasitological diagnosis with histology. Serum was processed with enzyme immunoassay (EIA) using three antigen preparations: total hydatid liquid (LHT), purified fraction of LHT (S2B) and purified lipoprotein (B). Western Blot (WB) was used as a control. EIA proved effective for differentiating Echinococcus granulosus from larval stage of Taenia hydatigena and intestinal cestodes in all three antigen preparations. Serums from macroscopically negative sheep were reactive to EIA and positive with WB. In the whole flock, sensitivity was 89.2% for LHT, 80.0% for S2B and 86.4% for B. Sensitivity in lambs was 78.6% for LHT, 75.0% for S2B and 64.3% for B. Macroscopic diagnosis at the time of slaughter was found to have limitations as a reference test for immunodiagnosis of cystic equinococcosis in sheep, so it was necessary to include histology and WB as reference tests. LHT was the antigen preparation of greatest value and EIA proved to be a sensitive and specific technique, adequate for surveillance systems and for evaluating control programmes.

Non-surgical and non-chemical attempts to treat echinococcosis: do they work?

PubMed Central

Tamarozzi, Francesca; Vuitton, Lucine; Brunetti, Enrico; Vuitton, Dominique Angèle; Koch, Stéphane

2014-01-01

Cystic echinococcosis (CE) and alveolar echinococcosis (AE) are chronic, complex and neglected diseases. Their treatment depends on a number of factors related to the lesion, setting and patient. We performed a literature review of curative or palliative non-surgical, non-chemical interventions in CE and AE. In CE, some of these techniques, like radiofrequency thermal ablation (RFA), were shelved after initial attempts, while others, such as High-Intensity Focused Ultrasound, appear promising but are still in a pre-clinical phase. In AE, RFA has never been tested, however, radiotherapy or heavy-ion therapies have been attempted in experimental models. Still, application to humans is questionable. In CE, although prospective clinical studies are still lacking, therapeutic, non-surgical drainage techniques, such as PAIR (puncture, aspiration, injection, re-aspiration) and its derivatives, are now considered a useful option in selected cases. Finally, palliative, non-surgical drainage techniques such as US- or CT-guided percutaneous biliary drainage, centro-parasitic abscesses drainage, or vascular stenting were performed successfully. Recently, endoscopic retrograde cholangiopancreatography (ERCP)-associated techniques have become increasingly used to manage biliary fistulas in CE and biliary obstructions in AE. Development of pre-clinical animal models would allow testing for AE techniques developed for other indications, e.g. cancer. Prospective trials are required to determine the best use of PAIR, and associated procedures, and the indications and techniques of palliative drainage. PMID:25531730

Occurrence and financial loss assessment of cystic echinococcosis (hydatidosis) in cattle slaughtered at Wolayita Sodo municipal abattoir, Southern Ethiopia.

PubMed

Bekele, Jemere; Butako, Berhanu

2011-01-01

A cross-sectional study aimed at determining the prevalence and cyst characteristics and estimating the financial loss due to cystic echinococcosis (hydatidosis) in cattle slaughtered at Wolayita Sodo municipal abattoir was conducted from November 2009 to April 2010. Out of 546 cattle examined, 92 (16.85%) were found to harbor visible hydatid cysts. Significantly higher infection was detected in local (P < 0.05) than crossbred cattle. No significant variation was observed with regard to origin, sex, and body condition status of animals. Regarding organ distribution, infections of the lung, liver, spleen, and kidney were 57.78%, 35.46%, 8.75%, and 4.01%, respectively. Of the total 1,097 hydatid cysts counted, 952 (86.78%), 136 (12.4%), and eight (0.82%) were found to be small-sized, medium-sized, and large-sized, respectively. Likewise, out of 450 cysts assessed, 138 (30.67%) were fertile, 241 (53.56%) sterile, and 71 (15.78%) calcified. Of the 138 fertile cysts subjected for viability test, 13 (9.42%) were viable while 125 (90.57%) were nonviable. Moreover, assessment of annual economic loss due to bovine hydatidosis at Wolayita Sodo municipal abattoir from offal condemnation and carcass weight loss was estimated at 410,755.90 Ethiopian Birr (ETB; 30,202.64 US$; 1 US$ = 13.60 ETB). Despite the moderate magnitude of infection detected currently, there seems to be an existing socioeconomic situation favorable for hydatidosis, and hence, it remains one of the most important diseases warranting serious attention for prevention and control actions in Wolayita zone. Hence, establishment of well-equipped standardized abattoirs, creation of public awareness, and control of stray dogs are of paramount importance.

A retrospective analysis of human cystic echinococcosis in Hamedan province, an endemic region of Iran.

PubMed

Ahmadi, N A; Hamidi, M

2008-10-01

In a retrospective investigation, the medical files of the patients who, between 1992 and 2006, underwent surgery for the treatment of cystic echinococcosis (CE) in one of the three university hospitals in Hamedan province, in western Iran, were explored. Of the 179 patients involved, 44.1% were male and 55.9% female, giving a male/ female ratio of 0.79. The mean annual surgical incidence of CE in the catchment areas of the study hospitals was estimated to be 1.33 cases/100,000. Although most of Hamedan's population is urban (53.02% at the last census, in 1997), most (57.5%) of the patients lived in rural areas (P<0.05). Among the CE cases, housewives (47.3%), farmers (16.6%) and labourers (16.0%) were over-represented compared with the frequencies of these occupations in the general population of the region (P<0.05 for each). Most of the CE cases were aged 20-39 years (35.8%) or 40-59 years (29.0%) and most had liver cysts, either alone (60.9%) or with cysts in other sites (9.5%), although 19.6% of the cases appeared only to have lung cysts. Overall, 90.5% of the cases showed single-organ involvement. Liver involvement was 3.3-fold more common than lung involvement, the ratio between cases with liver cysts and those with lung cysts generally increasing with age (from 1.6 for the cases aged <20 years to 5.6 among the cases aged 40-59 years). The most common clinical symptoms were abdominal pain and/or a sensation of abdominal mass in those with hepatic CE, and chest pain, cough, dyspnoea and/or haemoptysis in those with pulmonary CE. Although 72% of the cases each had a single cyst, 19.6% had two cysts each and 8.4% each had at least three cysts. Only three (1.7%) of the cases had had previous surgery for CE. Diagnosis of CE in Hamedan province is largely dependent on ultrasonography and computed tomography. Together, these results confirm that CE is endemic in the province and indicate that certain occupations (housewife, farmer and labourer) and/or rural life are

Serological confirmatory testing of alveolar and cystic echinococcosis in clinical practice: results of a comparative study with commercialized and in-house assays.

PubMed

Reiter-Owona, Ingrid; Grüner, Beate; Frosch, Matthias; Hoerauf, Achim; Kern, Peter; Tappe, Dennis

2009-01-01

Sera of 50 patients with either cystic (CE) or alveolar echinococcosis (AE) in different clinical stages were examined for the presence of anti-Echinococcus-antibodies. Antibody-screening was performed with ELISA, IHA and IFAT, and confirmatory testing was done by the commercialized E. multilocularis-specific Em2plus-ELISA versus an in-house E. multilocularis-specific Em10-ELISA. Sera with discrepant confirmatory results were subjected to a commercial Echinococcus IgG Western blot (WB). In sera from patients with CE, the Em2plus-ELISA showed cross-reactions in 23.5%, whereas the Em10-ELISA did not exhibit any cross-reactivity. Cross-reactivity paralleled active infection with high antibody titers in the screening assays. In sera from patients with AE, confirmation by both ELISAs was achieved in 57.6%, mostly in patients with an advanced stage of the disease and high antibody titers in the screening assays. False-negative reactions of both ELISAs occurred in 30.3%, mostly in patients who had low antibody levels in the screening tests. The Em2plus-ELISA exhibited fewer false-negative reactions than the Em10-ELISA. The WB confirmed the positive results of either assay and was the assay with the highest reliability at different stages of CE and AE, followed by the Em2plus-ELISA for AE. High antibody titers in the screening assays will favour the detection of species-specific antibodies in either form.

Development and evaluation of real-time loop-mediated isothermal amplification assay for rapid detection of cystic echinococcosis.

PubMed

Ahmed, Mohamed E; Eldigail, Mawahib H; Elamin, Fatima M; Ali, Ibtisam A; Grobusch, Martin P; Aradaib, Imadeldin E

2016-09-13

Cystic echinococcosis (CE) or hydatidosis, caused by the larval stage of Echinococcus granulosus (EG)-complex, is a neglected parasitic disease of public health importance. The disease is endemic in many African and Mediterranean countries including the Sudan. The objective of the present study was to develop and evaluate a real-time loop-mediated isothermal amplification (LAMP) assay for simple and rapid detection of CE in humans and domestic live stock in Sudan. A set of six LAMP primers, designed from the mitochondrial NADH-1 gene of EG cattle strain of genotype 5 (G5), was used as a target for LAMP assay. The assay was performed at a constant temperature (63 °C), with a real-time follow-up using a LightCycler and fluorochrome dye. Following amplification cycles in a simple water bath, LAMP products were observed for color change by naked eye and were visualized under UV light source using agarose gel electrophoresis. The real-time LAMP assay identified a variety of hydatid cysts strains recovered in the Sudan, including Echinococcus canadenses (G6) and Echinococcus ortleppi (G5). Real-time LAMP positive results were detected by the presence of an amplification curve, whereas negative results were indicated by absence of fluorescence detection. Positive LAMP results appeared as a bluish-colored reaction as observed by naked eye, whereas negative LAMP results were observed as purple-colored reaction. The sensitivity studies indicated that the LAMP assay detected as little as a 10 fg of parasite DNA. There was 100 % agreement between results of the LAMP assay and our previously described nested PCR when testing 10-fold serial dilution of DNA extracted from EG-complex hydatid cyst. However, there was no cross-reactivity with other parasites including cysticercus bovis, Fasciola gigantica, and Schistosoma bovis and nucleic acid free samples. The developed LAMP assay would be expected to prove highly significant in epidemiological surveys of CE in developing

TLR2 and TLR4 expression in peripheral blood mononuclear cells of patients with chronic cystic echinococcosis and its relationship with IL-10.

PubMed

Shan, J-Y; Ji, W-Z; Li, H-T; Tuxun, T; Lin, R-Y; Wen, H

2011-12-01

This study aims at relating Toll-like receptors (TLR) and human systemic cytokines in patients with chronic cystic echinococcosis (CE). By real-time fluorescent quantitative reverse-transcription polymerase chain reaction, we measured the expression level of TLR2 and TLR4 mRNA in peripheral blood mononuclear cells (PBMCs), and using ELISA, we detected the cytokines IFN-γ, IL-12p70, IL-10, IL-4 and IL-17A from 34 chronic CE cases (four patients with biliary leakage; four patients with secondary location including three in lung and one in bone) and 22 healthy controls (HC). TLR2 and TLR4 mRNA expression were significantly higher in the CE group (P<0·05); levels of serum IL-10, IL-4 and IL-12p70 in patients with CE were significantly higher than those in controls (P<0·05). There were no differences in IFN-γ and IL-17A levels between the CE group and the HC group (P>0·05). In the patients with CE, positive correlations were noted between the expression of TLR2 and the serum level of IL-10, as well as between the expression of TLR4 and the serum level of IL-10. Our findings supported the hypothesis that during chronic CE infection, altered TLR expression might be involved in the cytokine modulation, which allowed the parasite to escape host immunosurveillance and promoted chronic infection. © 2011 Blackwell Publishing Ltd.

Cystic echinococcosis of the liver and lung treated by radiofrequency thermal ablation: an ex-vivo pilot experimental study in animal models.

PubMed

Lamonaca, Vincenzo; Virga, Antonino; Minervini, Marta Ida; Di Stefano, Roberta; Provenzani, Alessio; Tagliareni, Pietro; Fleres, Giovanna; Luca, Angelo; Vizzini, Giovanni; Palazzo, Ugo; Gridelli, Bruno

2009-07-14

To evaluate radiofrequency thermal ablation (RTA) for treatment of cystic echinococcosis in animal models (explanted organs). Infected livers and lungs from slaughtered animals, 10 bovine and two ovine, were collected. Cysts were photographed, and their volume, cyst content, germinal layer adhesion status, wall calcification and presence of daughter or adjacent cysts were evaluated by ultrasound. Some cysts were treated with RTA at 150 W, 80 degrees C, 7 min. Temperature was monitored inside and outside the cyst. A second needle was placed inside the cyst for pressure stabilization. After treatment, all cysts were sectioned and examined by histology. Cysts were defined as alive if a preserved germinal layer at histology was evident, and as successfully treated if the germinal layer was necrotic. The subjects of the study were 17 cysts (nine hepatic and eight pulmonary), who were treated with RTA. Pathology showed 100% success rate in both hepatic (9/9) and lung cysts (8/8); immediate volume reduction of at least 65%; layer of host tissue necrosis outside the cyst, with average extension of 0.64 cm for liver and 1.57 cm for lung; and endocyst attached to the pericystium both in hepatic and lung cysts with small and focal de novo endocyst detachment in just 3/9 hepatic cysts. RTA appears to be very effective in killing hydatid cysts of explanted liver and lung. Bile duct and bronchial wall necrosis, persistence of endocyst attached to pericystium, should help avoid or greatly decrease in vivo post-treatment fistula occurrence and consequent overlapping complications that are common after surgery or percutaneous aspiration, injection and reaspiration. In vivo studies are required to confirm and validate this new therapeutic approach.

Liver hydatid cyst leading to bilateral pulmonary artery embolism and bilateral multiple pulmonar echinococcosis via inferior vena cava: report of a case.

PubMed

Bayaroğullari, Hanifi; Davran, Ramazan; Cavuş, Yeliz; Yetim, Tülin Durgun; Evirgen, Ömer

2013-01-01

Hydatid disease (HD) is a worldwide parasitic disease. Echinococcosis may involve many organs but affect most commonly liver and lungs. The location of echinococcal cysts inside pulmonary artery is extremely rare. Radiologic findings range from purely cystic lesions to a completely solid appearance. Hydatid cysts (HC) can be solitary or multiple and varies size. Pulmonary artery embolism of HC can be symptomatic or asymptomatic. When symptomatic, we see the chest pain, dyspnea, cough, hemoptysis and sometimes acute cor pulmonale or sudden death secondary to massive giant pulmonary artery embolism of HC. Copyright © 2013 Elsevier Inc. All rights reserved.

Why more research needs to be done on echinococcosis in Pakistan.

PubMed

Ahmed, Haroon; Ali, Shahzad; Afzal, Muhammad Sohail; Khan, Abid Ali; Raza, Hamid; Shah, Zaheer Hussain; Simsek, Sami

2017-07-03

Echinococcosis has a worldwide geographical distribution with endemic foci on every inhabited continent. Due to the frequent outbreaks in different parts of Pakistan in the recent past, echinococcosis is being described as a neglected tropical disease and is considered one of the most neglected parasitic diseases in the country. In endemic regions, predominantly settings with limited resources, there are high numbers of echinococcosis patients, as these communities do not have access to appropriate treatment. In Pakistan, there are limited reports on echinococcosis. The disease is prevalent in human and livestock, but this has not been sufficiently explored yet. Pakistan is an agricultural country and due to the disease's zoonotic mode of transmission, there is a dire need of future research on it. The present paper is an effort to highlight the importance of echinococcosis in Pakistan. There is a dire need for future research on echinococcosis in Pakistan as very few investigations had been carried out on this topic thus far. The prevalence of the disease in neighbouring countries highlights that Pakistan might be at severe risk of this zoonotic infection and further supports the need for more research. In Pakistan, the majority of the population lives in rural areas with limited acess to proper hygienic/sanitary facilities. These conditions favour the outbreak of diseases such as echinococcosis. The limited available data could result in higher outbreaks in the future, and thus cause the already weak healthcare system to overburden. The country has a meagre annual budget for health, which is being spent on known infections such as polio, dengue fever and hepatic viral infections. A proper surveillance system for echinococcosis is required across the country as treatment is usually expensive, complicated and may require extensive surgery and/or prolonged drug therapy. Development of new/novel drugs and other treatment modalities receives very little, if any

Surgical treatment of hepatic echinococcosis in Prizren (Kosovo)

PubMed Central

Avdaj, Afrim; Namani, Sadie

2014-01-01

Management option of hepatic echinococcosis represents a major challenge for a surgeon. The aim of the study was to evaluate surgical treatment of patients with hepatic echinococcosis at the surgery department of the regional hospital in Prizren (Kosovo). The medical records of 22 patients operated for hepatic echinococcosis in our department during a four year study period (2009–2013) were retrospectively reviewed. Apart from the total of 5850 operated patients, 22 cases were diagnosed for liver echinococcosis (0.4%). The most affected age group was from 26 to 50 years (54.5%). Female gender, 16 cases (73%), and patients living in rural places, 14 patients (64%), dominated significantly. The most affected region was Municipality of Dragash. All patients underwent ultrasonography, 13 patients underwent CT scans and 5 patients MRI of abdomen. The mean preoperative ultrasonographic diameter of cysts was 9.5 cm and maximal 21 cm. Cysts were most often localized in right hepatic lobe (77%) and subcostal laparotomy was most commonly performed (82%). The performed surgical procedures were: endocystectomy and partial pericystectomy with omentoplication according to Papadimitris (73%), endocystectomy and capitonnage and endocystectomy with external drainage (14%). The laparoscopic approach was used only in one patient. In conclusion, hepatic echinococosis was not common among operated patients at our surgery department. Subcostal laparotomy with endocystectomy and partial pericystectomy with omentoplication according to Papadimitris was most commonly used. Exact distribution of echinococcosis is needed to be analyzed with a larger cohort study including all surgery units in the country and with a longer monitoring. PMID:25568800

30-yr course and favorable outcome of alveolar echinococcosis despite multiple metastatic organ involvement in a non-immune suppressed patient

PubMed Central

2013-01-01

We report the 30-yr history of a well-documented human case of alveolar echinococcosis, with a lung lesion at presentation followed by the discovery of a liver lesion, both removed by surgery. Subsequently, within the 13 years following diagnosis, metastases were disclosed in eye, brain and skull, as well as additional lung lesions. This patient had no immune suppression, and did not have the genetic background known to predispose to severe alveolar echinococcosis; it may thus be hypothesized that iterative multi-organ involvement was mostly due to the poor adherence to benzimidazole treatment for the first decade after diagnosis. Conversely, after a new alveolar echinococcosis recurrence was found in the right lung in 1994, the patient accepted to take albendazole continuously at the right dosage. After serology became negative and a fluoro-deoxy-glucose-Positron Emission Tomography performed in 2005 showed a total regression of the lesions in all organs, albendazole treatment could be definitively withdrawn. In 2011, the fluoro-deoxy-glucose-Positron Emission Tomography showed a total absence of parasitic metabolic activity and the patient had no clinical symptoms related to alveolar echinococcosis. The history of this patient suggests that multi-organ involvement and alveolar echinococcosis recurrence over time may occur in non-immune suppressed patients despite an apparently “radical” surgery. Metastatic dissemination might be favored by a poor adherence to chemotherapy. Combined surgery and continuous administration of albendazole at high dosage may allow alveolar echinococcosis patients to survive more than 30 years after diagnosis despite multi-organ involvement. PMID:23281596

Knowledge, attitude, and practices (KAP) and risk factors analysis related to cystic echinococcosis among residents in Tibetan communities, Xiahe County, Gansu Province, China.

PubMed

Li, Dan; Gao, Qi; Liu, Jian; Feng, Yu; Ning, Wenhua; Dong, Yanqing; Tao, Lixin; Li, Jingyi; Tian, Xiujun; Gu, Junchao; Xin, Deli

2015-07-01

Cystic echinococcosis (CE) is a global parasitic zoonosis caused by Echinococcus granulosus. The disease is highly endemic in western China, especially in Tibetan areas, because of poor economic development and hygiene conditions, limited community knowledge of CE, a large scale of dogs, and home slaughtering of livestock. Although many researchers have analyzed risk factors of CE transmission in Tibetan Plateau, there are rare reports of knowledge, attitude, and practice (KAP) of residents about CE in Tibetan communities. In our current study, community based cross-sectional study was conducted in three townships in Xiahe County, Gannan Tibetan Autonomous Prefectures of Gansu Province from May to September 2013. A total of 972 participants originating from Tibetan communities of 31 villages in the 3 townships were registered and data were collected using structured questionnaires. From the total of 972 study participants (457 males and 515 females), 65.9% heard of the disease CE. Most of them (96.1%) would like to accept CE inspection. About half of the peoples feed their dogs often and major of them do not play with the dogs. Risk factors included resident, knowing dog could be infected, knowing eating could be route of infection, oldest dog's age, usually feed your dog by self, feed dogs with internal organs. In general our findings showed that most of residents had positive attitude toward treatments of the disease, but their practice about disease prevention and control was low. Therefore, our study called for continued and strengthened education of changing the life style, especially the behaviors related to dogs. Copyright © 2015 The Authors. Published by Elsevier B.V. All rights reserved.

Sonographical and serological survey of human cystic echinococcosis and analysis of risk factors associated with seroconversion in rural communities of Kerman, Iran.

PubMed

Harandi, M F; Moazezi, S S; Saba, M; Grimm, F; Kamyabi, H; Sheikhzadeh, F; Sharifi, I; Deplazes, P

2011-12-01

Cystic echinococcosis (CE) caused by larval stage of Echinococcus granulosus, is an endemic zoonosis in Iran particularly in rural regions. The objective of this study was to determine the prevalence of CE among rural communities in Kerman using ultrasonography (US) and serology. Kerman Province, in southeastern Iran, is the largest province, with 2.9 million inhabitants. A sample of 1140 individuals (200 males and 940 females) was selected by randomized cluster sampling in 2006-2008. After acquiring informed consent for each participant a questionnaire was filled, complete abdominal US in supine position was carried out and 5 ml blood was collected for ELISA test. Two hydatid cases (0.2%) were detected by ultrasound. Serological results showed 7.3% seropositivity, and females (8.3%) were significantly more positive than males (2.1%). There were significant difference between CE seropositivity and sex, age and occupation. Residents of desert regions (Shahdad, Andouhjerd and Golbaf) were 2.5 times more likely to be seropositive than mountainous regions with better socioeconomic status (OR = 2.5; 95% CI = 1.09-5.95). Dog ownership does not appear to be a significant risk factor for CE in the region. Only about 10% of households own dogs, usually only one dog. However, the stray dog population of Kerman province is estimated at 145 000-480 000 (3.5-11.5 times the owned dog population). Infection in humans and animals would appear to come mostly from infected stray dogs. Management of stray dog population could make major progress in control of hydatid disease. In addition, proper washing of vegetables decreased probability of infection by 53% (OR = 0.47; 95% CI = 0.26-0.84). The serological study showed that many people, especially women, had been exposed to Echinococcus eggs and had seroconverted but were not infected. © 2011 Blackwell Verlag GmbH.

Cystic Echinococcosis in the Province of Álava, North Spain: The Monetary Burden of a Disease No Longer under Surveillance

PubMed Central

Carabin, Hélène; Balsera-Rodríguez, Francisco J.; Rebollar-Sáenz, José; Benner, Christine T.; Benito, Aitziber; Fernández-Crespo, Juan C.; Carmena, David

2014-01-01

Cystic echinococcosis (CE) is endemic in Spain but has been considered non-endemic in the province of Álava, Northern Spain, since 1997. However, Álava is surrounded by autonomous regions with some of the highest CE prevalence proportions in the nation, casting doubts about the current classification. The purpose of this study is to estimate the frequency of CE in humans and animals and to use this data to determine the societal cost incurred due to CE in the Álava population in 2005. We have identified epidemiological and clinical data from surveillance and hospital records, prevalence data in intermediate (sheep and cattle) host species from abattoir records, and economical data from national and regional official institutions. Direct costs (diagnosis, treatment, medical care in humans and condemnation of offal in livestock species) and indirect costs (productivity losses in humans and reduction in growth, fecundity and milk production in livestock) were modelled using the Latin hypercube method under five different scenarios reflecting different assumptions regarding the prevalence of asymptomatic cases and associated productivity losses in humans. A total of 13 human CE cases were reported in 2005. The median total cost (95% credible interval) of CE in humans and animals in Álava in 2005 was estimated to range between €61,864 (95%CI%: €47,304–€76,590) and €360,466 (95%CI: €76,424–€752,469), with human-associated losses ranging from 57% to 93% of the total losses, depending on the scenario used. Our data provide evidence that CE is still very well present in Álava and incurs important cost to the province every year. We expect this information to prove valuable for public health agencies and policy-makers, as it seems advisable to reinstate appropriate surveillance and monitoring systems and to implement effective control measures that avoid the spread and recrudescence of the disease. PMID:25102173

Comparative analysis of the diagnostic performance of crude sheep hydatid cyst fluid, purified antigen B and its subunit (12 Kda), assessed by ELISA, in the diagnosis of human cystic echinococcosis.

PubMed

Tawfeek, Gihan M; Elwakil, Hala S; El-Hoseiny, Laila; Thabet, Hala S; Sarhan, Rania M; Awad, Nabil S; Anwar, Wagida A

2011-02-01

The diagnosis of patients with cystic echinococcosis (CE) by means of serology has a limited support in clinical practice due to cross-reactivity with other helminthes leading to overestimation of the parasite's true prevalence. A wealth of reports on the diagnostic performance of antigen B (AgB) has been produced. This study was designed to comparatively assess the diagnostic efficacy of crude sheep hydatid cyst fluid (HCF), AgB and its subunit (12 KDa) to detect IgG or IgG4 antibodies in CE patients' sera using enzyme-linked immunosorbent assay (ELISA).The best diagnostic performance was obtained with anti-HCF IgG ELISA which gave 92.4% sensitivity and 92.6% specificity. Despite the low sensitivity of anti AgB IgG ELISA (84%), it gave the best specificity (94.4%) with less cross-reaction with sera of subjects infected with other parasites. In conclusion, it is recommended to use anti-HCF IgG ELISA for initial screening in large seroprevalence studies. Further analysis of positive serum samples with anti AgB IgG ELISA would allow the confirmation of true positives. Specific IgG4 ELISA may represent a complementary assay, useful as secondary confirmatory tests for patients with suspected CE and negative for total IgG ELISA.

Canine echinococcosis: genetic diversity of Echinococcus granulosus sensu stricto (s.s.) from definitive hosts.

PubMed

Boufana, B; Lett, W; Lahmar, S; Griffiths, A; Jenkins, D J; Buishi, I; Engliez, S A; Alrefadi, M A; Eljaki, A A; Elmestiri, F M; Reyes, M M; Pointing, S; Al-Hindi, A; Torgerson, P R; Okamoto, M; Craig, P S

2015-11-01

Canids, particularly dogs, constitute the major source of cystic echinococcosis (CE) infection to humans, with the majority of cases being caused by Echinococcus granulosus (G1 genotype). Canine echinococcosis is an asymptomatic disease caused by adult tapeworms of E. granulosus sensu lato (s.l.). Information on the population structure and genetic variation of adult E. granulosus is limited. Using sequenced data of the mitochondrial cytochrome c oxidase subunit 1 (cox1) we examined the genetic diversity and population structure of adult tapeworms of E. granulosus (G1 genotype) from canid definitive hosts originating from various geographical regions and compared it to that reported for the larval metacestode stage from sheep and human hosts. Echinococcus granulosus (s.s) was identified from adult tapeworm isolates from Kenya, Libya, Tunisia, Australia, China, Kazakhstan, United Kingdom and Peru, including the first known molecular confirmation from Gaza and the Falkland Islands. Haplotype analysis showed a star-shaped network with a centrally positioned common haplotype previously described for the metacestode stage from sheep and humans, and the neutrality indices indicated population expansion. Low Fst values suggested that populations of adult E. granulosus were not genetically differentiated. Haplotype and nucleotide diversities for E. granulosus isolates from sheep and human origin were twice as high as those reported from canid hosts. This may be related to self-fertilization of E. granulosus and/or to the longevity of the parasite in the respective intermediate and definitive hosts. Improved nuclear single loci are required to investigate the discrepancies in genetic variation seen in this study.

[Epidemiology and prevention of echinococcosis in France].

PubMed

Petavy, A F; Deblock, S; Walbaum, S

1990-01-21

Echinococcosis is a cosmopolitan parasitic disease caused by the larvae of two tapeworms: Echinococcus granulosus responsible for unilocular hydatidosis, and Echinococcus multilocularis responsible for alveolar hydatid disease. In both cases man is infected by ingesting eggs of these tapeworms belonging to the genus Cestoda. In France, the only animal that acts as definitive host and transmits the unilocular type of hydatid disease is the dog, while the alveolar type is transmitted by dogs, foxes and cats. Carnivorous pet animals must be taken into account in the prevention of echinococcosis, whether their role is direct or indirect (fouling plants).

Echinococcosis/hydatidosis: a severe threat in Mediterranean countries.

PubMed

Dakkak, A

2010-11-24

Echinococcosis/hydatidosis is one of the most important parasitic zoonotic diseases in the world. Both cystic hydatidosis (CE) caused by Echinococcus granulosus and alveolar echinococcosis (AE) caused by E. multilocularis have been reported in several countries of the Mediterranean region (MR). E. granulosus has always been present in the MR and is the most common species. This parasite depends on the dog-sheep cycle and is actively transmitted in all pastoral regions where sheep, cattle and camelids predominate. E. multilocularis occurs only sporadically in limited areas of France, Serbia and Montenegro, Turkey, Tunisia and Morocco. However recent evidence indicates that it is spreading into other regions of the Mediterranean. Due to the lack of well-documented data, and to the fact that CE is not a notifiable disease in the majority of M countries, the precise incidence and prevalence of CE in humans and animals are not known. Published data suggests that prevalence is rather high in North Africa, Turkey, Greece, and in several regions of Italy and Spain. CE is an increasing public health and socio-economic concern due to the considerable morbidity rates that give rise to high economic losses both in the public health sector and in the livestock industry. Hospitalisation for human CE lasts from 2 weeks to more than 1 month in case of surgery. A number of factors contribute to the increase of prevalence and to the spreading of CE in the MR. These include the diversity of livestock production systems (predominantly extensive, traditional animal husbandry), small, ill-equipped and unsupervised slaughter-houses, illegal and family slaughtering, low public awareness of hydatid diseases, and the high population of stray dogs. Cyprus is the only country where an eradication programme has been successfully implemented. There have been, however, important developments in the last decade in CE epidemiology, in the diagnosis of canine infection, in strain characterisation

Surgical Treatment of Cardiac Echinococcosis: Report of Nine Cases

PubMed Central

Papo, Isidor; Ginsberg, Ervin; Albreht, Miša; Martinović, Nebojša; Sokolić, Josip

1982-01-01

We reviewed the results in eight patients followed from 10 months to 22 years after one-stage surgery for removal of primary and secondary myocardial and pericardial hydatid cysts. The results were satisfactory in eight patients who survived the operation. One patient, however, with recurrent myocardial echinococcosis, pulmonary embolization, and sepis died at reoperation. The series was analyzed with reference to the surgical treatment and world-wide experience of complicated cardiac echinococcosis. Images PMID:15226804

Worldwide epidemiology of liver hydatidosis including the Mediterranean area

PubMed Central

Grosso, Giuseppe; Gruttadauria, Salvatore; Biondi, Antonio; Marventano, Stefano; Mistretta, Antonio

2012-01-01

The worldwide incidence and prevalence of cystic echinococcosis have fallen dramatically over the past several decades. Nonetheless, infection with Echinococcus granulosus (E. granulosus) remains a major public health issue in several countries and regions, even in places where it was previously at low levels, as a result of a reduction of control programmes due to economic problems and lack of resources. Geographic distribution differs by country and region depending on the presence in that country of large numbers of nomadic or semi-nomadic sheep and goat flocks that represent the intermediate host of the parasite, and their close contact with the final host, the dog, which mostly provides the transmission of infection to humans. The greatest prevalence of cystic echinococcosis in human and animal hosts is found in countries of the temperate zones, including several parts of Eurasia (the Mediterranean regions, southern and central parts of Russia, central Asia, China), Australia, some parts of America (especially South America) and north and east Africa. Echinococcosis is currently considered an endemic zoonotic disease in the Mediterranean region. The most frequent strain associated with human cystic echinococcosis appears to be the common sheep strain (G1). This strain appears to be widely distributed in all continents. The purpose of this review is to examine the distribution of E. granulosus and the epidemiology of a re-emerging disease such as cystic echinococcosis. PMID:22509074

Study of cystic echinococcosis in slaughtered animals in Al Baha region, Saudi Arabia: interaction between some biotic and abiotic factors.

PubMed

Ibrahim, Mohamed M

2010-01-01

The variation in cystic echinococcosis (CE) prevalence and mean intensity was studied in relation to site, season and host age and sex. A total of 12,911 slaughtered animals, 140 camels, 2668 cattle, 6525 sheep and 3578 goats were inspected for hydatid cysts in Al Baha region, Saudi Arabia, in three study areas during four seasons from June 2008 to May 2009. The prevalence of infection was 32.85%, 8.28%, 12.61% and 6.56% in camels, cattle, sheep and goats respectively. The prevalence of the parasite varied significantly in relation to site, season and host age classes and sex in most host species. Spring showed the highest prevalence in camels, cattle and sheep. A significant association was found among host age classes and likelihood of infection in all examined hosts and the oldest age class was significantly more likely to be infected. The main effects in parasite intensity were host sex and age in most examined host species. A positive correlation was found between intensity of CE and host age class in all animal species examined. The most commonly infected organs were liver and lungs which constituted 48.75% and 32.83% respectively, of the total infected organs. There was a significant difference among host species in fertile cysts (P<0.0001). The higher percentages of fertile cysts were in sheep (47.67%) and goats (23.99%) indicating that sheep and goats are the most important intermediate hosts for Echinococcus granulosus. Examined hydatid cysts of the liver had a higher fertility rate (38.79%) than those of the lungs (25.13%). Cysts size ranged from 1 to 8cm in diameter. The mean cyst diameter was found in the lungs higher than that in the liver in all hosts. The range in the number of cysts was 1-33 in infected animals. The mean number of cysts was higher in lungs than that in liver in all examined animals. The viability rate of protoscoleces of liver fertile cysts (62.20%) was significantly higher than that of lung cysts (52.73%). In conclusion, these

Evaluation of a newly designed sandwich enzyme linked immunosorbent assay for the detection of hydatid antigen in serum, urine and cyst fluid for diagnosis of cystic echinococcosis.

PubMed

Chaya, Dr; Parija, Subhash Chandra

2013-07-01

Cystic echinococcosis (CE) is a zoonotic disease of humans with variable clinical manifestations. Imaging and immunological methods are currently the mainstay of diagnosis of this disease. Although the immunological tests for detection of anti-echinococcal antibodies have several disadvantages, they are widely being used. Antigen is far more superior than antibody detection test as they can provide a specific parasitic diagnosis. A sandwich enzyme linked immunosorbent assay (ELISA) was designed using antibodies to 24 kDa urinary hydatid antigen for the detection of hydatid antigens in urine, serum and cyst fluid specimens. The performance of this novel test was compared with that of other hydatid antibody detection ELISA and enzyme immune transfer blot (EITB) using radiological and surgical confirmation as the gold standard. The antigen detection ELISA showed 100% sensitivity and specificity when tested with cyst fluid. On testing urine and serum, the antigen detection ELISA was found to be more specific than antibody detection ELISA. EITB was found to be the most sensitive and specific test. ELISA using polyclonal antibodies against 24 kDa urinary hydatid protein was moderately sensitive to detect hydatid antigen in serum and urine. Hence polyclonal antibodies to 24 kDa urinary hydatid antigen can be used as an alternative source of antibody to detect hydatid antigen in serum, urine and cyst fluid. In the present study, EITB was found to be highly specific test for detection of hydatid antibodiesin serum. 24 kDa protein was found to be specific and of diagnostic value in CE.

Evaluation of a newly designed sandwich enzyme linked immunosorbent assay for the detection of hydatid antigen in serum, urine and cyst fluid for diagnosis of cystic echinococcosis

PubMed Central

Chaya, DR; Parija, Subhash Chandra

2013-01-01

Introduction: Cystic echinococcosis (CE) is a zoonotic disease of humans with variable clinical manifestations. Imaging and immunological methods are currently the mainstay of diagnosis of this disease. Although the immunological tests for detection of anti-echinococcal antibodies have several disadvantages, they are widely being used. Antigen is far more superior than antibody detection test as they can provide a specific parasitic diagnosis. Materials and Methods: A sandwich enzyme linked immunosorbent assay (ELISA) was designed using antibodies to 24 kDa urinary hydatid antigen for the detection of hydatid antigens in urine, serum and cyst fluid specimens. The performance of this novel test was compared with that of other hydatid antibody detection ELISA and enzyme immune transfer blot (EITB) using radiological and surgical confirmation as the gold standard. Results: The antigen detection ELISA showed 100% sensitivity and specificity when tested with cyst fluid. On testing urine and serum, the antigen detection ELISA was found to be more specific than antibody detection ELISA. EITB was found to be the most sensitive and specific test. Conclusions: ELISA using polyclonal antibodies against 24 kDa urinary hydatid protein was moderately sensitive to detect hydatid antigen in serum and urine. Hence polyclonal antibodies to 24 kDa urinary hydatid antigen can be used as an alternative source of antibody to detect hydatid antigen in serum, urine and cyst fluid. In the present study, EITB was found to be highly specific test for detection of hydatid antibodiesin serum. 24 kDa protein was found to be specific and of diagnostic value in CE. PMID:24470996

Study on the echinococcosis blood serum detection based on Raman spectroscopy combined with neural network

NASA Astrophysics Data System (ADS)

Cheng, Jin-ying; Xu, Liang; Lü, Guo-dong; Tang, Jun; Mo, Jia-qing; Lü, Xiao-yi; Gao, Zhi-xian

2017-01-01

A Raman spectroscopy method combined with neural network is used for the invasive and rapid detection of echinococcosis. The Raman spectroscopy measurements are performed on two groups of blood serum samples, which are from 28 echinococcosis patients and 38 healthy persons, respectively. The normalized Raman reflection spectra show that the reflectivity of the echinococcosis blood serum is higher than that of the normal human blood serum in the wavelength ranges of 101—175 nm and 1 801—2 701 nm. Then the principal component analysis (PCA) and back propagation neural network (BPNN) model are used to obtain the diagnosis results. The diagnosis rates for healthy persons and echinococcosis persons are 93.333 3% and 90.909 1%, respectively, so the average final diagnosis rate is 92.121 2%. The results demonstrate that the Raman spectroscopy analysis of blood serum combined with PCA-BPNN has considerable potential for the non-invasive and rapid detection of echinococcosis.

Echinococcosis in Serbia: an issue for the 21st century?

PubMed

Bobić, Branko; Nikolić, Aleksandra; Radivojević, Sofija Katić; Klun, Ivana; Djurković-Djaković, Olgica

2012-11-01

Echinococcosis (hydatidosis) is traditionally endemic in Southeast Europe, Serbia included. In Serbia, echinococcosis is mandatory reportable, and this review analyzes the officially reported data as well as the research data published between 1998 and 2010. Official data on human and animal infections were obtained from the Institute of Public Health of Serbia (IPHS, 2010), and from the Ministry of Agriculture, Trade, Forestry and Water Management (MATFWM) and the Statistical Office, respectively. Published data were obtained by searching the Medline, Scopus, and Google databases using "echinococcosis," "hydatidosis," and "Serbia" as key words. In addition, the search included national journals and doctoral theses, as well as conference proceedings. Only Echinococcus granulosus has been reported in Serbia, with a total of 409 cases of human infection officially reported during the observed period as opposed to 820 cases described in clinical studies. No trend in the incidence of infection was shown among adults, but the number of cases in children continuously decreased over the period. Patients were more frequently female and from rural areas. Differences in the geographic distribution of cases were noted, with a lower incidence in the central part of country. Liver disease was by far the most common presentation, but cases of unusual cyst locations have been described. Among domestic animals, sheep were the most highly infected species. A decreasing incidence of echinococcosis in animals has been noted as of the 1970s. Echinococcosis continues to be endemic in Serbia in the 21st century, but despite predictions, neither official data nor those from clinical studies indicate its re-emergence. However, there is gross underreporting. Public health authorities should actively work to increase reporting, as only valid reported data provide an accurate basis for future control plans.

[Survey of echinococcosis in Hokkaido and measure against it].

PubMed

Minagawa, T

1997-11-01

The first patient with echinococcosis who had lived in Rebun island was discovered in 1937 at Otaru and 134 patients had been identified as Rebun cases till 1989. In the meantime, the eradication of echinococcosis succeeded with extermination of dogs and foxes from the island. On the other hand, the first patient of Kon-sen cases was found in 1965 and 208 patients have been treated so far. The patients had been restricted within the Kon-sen region until 1979, but thereafter the patients have been found sporadically all over Hokkaido. This distributional tendency of the patients coincides with the explosion of the infection with Echinococcus multilocularis in foxes in Hokkaido after 1980. Prevention measure against echinococcosis has been carried out by the Committee for the Disorder of Hokkaido, by which we have been successful. We succeed in the early diagnosis and treatment of the patients, but still have no decisive methods to eradicate E. multilocularis in Hokkaido. This is one of the important problems of zoonosis to solve for the next century of Hokkaido.

Early Peritoneal Immune Response during Echinococcus granulosus Establishment Displays a Biphasic Behavior

PubMed Central

Mourglia-Ettlin, Gustavo; Marqués, Juan Martín; Chabalgoity, José Alejandro; Dematteis, Sylvia

2011-01-01

Background Cystic echinococcosis is a worldwide distributed helminth zoonosis caused by the larval stage of Echinococcus granulosus. Human secondary cystic echinococcosis is caused by dissemination of protoscoleces after accidental rupture of fertile cysts and is due to protoscoleces ability to develop into new metacestodes. In the experimental model of secondary cystic echinococcosis mice react against protoscoleces producing inefficient immune responses, allowing parasites to develop into cysts. Although the chronic phase of infection has been analyzed in depth, early immune responses at the site of infection establishment, e.g., peritoneal cavity, have not been well studied. Because during early stages of infection parasites are thought to be more susceptible to immune attack, this work focused on the study of cellular and molecular events triggered early in the peritoneal cavity of infected mice. Principal Findings Data obtained showed disparate behaviors among subpopulations within the peritoneal lymphoid compartment. Regarding B cells, there is an active molecular process of plasma cell differentiation accompanied by significant local production of specific IgM and IgG2b antibodies. In addition, peritoneal NK cells showed a rapid increase with a significant percentage of activated cells. Peritoneal T cells showed a substantial increase, with predominance in CD4+ T lymphocytes. There was also a local increase in Treg cells. Finally, cytokine response showed local biphasic kinetics: an early predominant induction of Th1-type cytokines (IFN-γ, IL-2 and IL-15), followed by a shift toward a Th2-type profile (IL-4, IL-5, IL-6, IL-10 and IL-13). Conclusions Results reported here open new ways to investigate the involvement of immune effectors players in E. granulosus establishment, and also in the sequential promotion of Th1- toward Th2-type responses in experimental secondary cystic echinococcosis. These data would be relevant for designing rational therapies

Impact of "Grain to Green" Programme on echinococcosis infection in Ningxia Hui Autonomous Region of China.

PubMed

Cleary, Eimear; Barnes, Tamsin S; Xu, Yangyang; Zhao, Hengbo; Clements, Archie C A; Gray, Darren J; McManus, Donald P; Atkinson, Jo-An M; Williams, Gail M; Yang, YuRong

2014-10-15

Cystic echinococcosis (CE) is endemic among the human population of Xiji County, Ningxia Hui Autonomous Region, China, where the prevalence is estimated to be between 2.2% and 3.6%. Government-run sheep abattoirs in Xiji County have closed in recent years and, as a consequence, slaughter is carried out mostly at rural market places. The market place in Xinglong Township, Xiji County, is home to an increasing number of stray dogs and the lack of government control over slaughter practices potentially favours Echinococcus granulosus transmission. A survey of sheep, goats and cattle reared in Xiji County was conducted in Xinglong Market and Xinglong Township to determine prevalence and transmission dynamics of E. granulosus infection. The liver and lungs of all livestock aged one year and older were examined macroscopically post mortem; visual examination and palpation of organs determined overall prevalence of E. granulosus. Cysts consistent in appearance with E. granulosus were observed in 2/184 sheep (prevalence 1.0%) and 1/55 of the cattle examined (prevalence 1.8%); 0/13 goats were found to be infected. However, microscopic examination of these suspected cysts failed to confirm these samples as E. granulosus, giving a prevalence of confirmed infection of zero percent in all three species. The prevalence of liver fluke was 61.3% in sheep and 12.7% in cattle with a significant difference between males and females (p ≤ 0.001). Considering the high prevalence of echinococcosis in the local human population, the absence of CE observed among commercially slaughtered livestock was surprising. Several explanations for this discrepancy and their implications are proposed. Copyright © 2014 Elsevier B.V. All rights reserved.

The reconsideration of natural history of echinococcosis at Rebun Island.

PubMed

Minagawa, T

1999-03-01

It has been believed that the outbreak of echinococcosis at Rebun Island had ceased by 1970. The first patient was diagnosed in 1936 and 131 patients have been authorized as echinococcosis so far. The conference of measures against the outbreak had been organized in 1948 and started to eradicate Echinococcus multilocularis from the Island. Medical examination to detect the patients and the capture and autopsy of dogs and cats had been carried out hard till 1970. At that time, foxes imported from Simusiru Island in the middle Kuriles during the years 1924 to 1926 had already disappeared and it has seemed to be sure that stray dogs and cats might carry E. multilocularis and excrete infectious eggs in stead of foxes. Since we have had no real data concerning the natural history of patients with echinococcosis without any treatments, it can not be recognized the time of infection and the role of dogs or cats on the spread of echinococcosis at Rebun Island. From the new data, it is concluded that the active life cycle of E. multilocularis between foxes and vole might be closed by 1940, since the last patient infected with E. multilocularis was born in 1940 and died in 1945. Furthermore, it is estimated that more than 200 patients (3 to 4% of people at the island) might die from echinococcosis, because of the fact of the unusual increase of mortality of liver disorders and oldness observed during the years of 1940 to 1960. 81 patients with the high possibility of echinococcosis detected from 1937 to 1963 can be added to 131 authorized patients. Surprisingly, it is noticed that the standard deviations of ages of death of 94 patients born in Meiji era (1880-1912) and 59 in Taisho and Showa eras (1912-1940) are 63.16 +/- 11.68, and 34.32 +/- 11.87, respectively. It means that both old and young people might be infected simultaneously but for the long period. There was no difference between the susceptibility of young and old men to E. multilocularis. The numbers of male

Prevention of alveolar echinococcosis--ecosystem and risk management perspectives in Japan.

PubMed

Konno, Keita; Oku, Yuzaburo; Tamashiro, Hiko

2003-12-01

We focused on the epidemiology of alveolar echinococcosis especially in Japan and discussed control measures to prevent an epidemic. No effective control measures against alveolar echinococcosis have been identified thus far because it is difficult to fully understand the ecology of the parasite and its hosts, i.e. the precise infection route to humans. In Hokkaido, Japan, infection rates among red foxes have recently risen even in low endemic districts. Infection seems to be spreading not only among wild foxes but also among domestic dogs. Despite only sporadic reports of human cases in Japan, we predict that the incidence of alveolar echinococcosis will increase in the near future if no effective preventive measures are put in place. An Echinococcus multilocularis epidemic would have the potential to affect the economy of Hokkaido, due to its impact on the agricultural and tourist industries. Well-designed epidemiological surveys are, therefore, urgently required prior to large outbreaks, based on understanding of the ecosystem around E. multilocularis.

An abattoir-based study on the prevalence and economic losses due to cystic echinococcosis in slaughtered herbivores in Ahwaz, south-western Iran.

PubMed

Ahmadi, N A; Meshkehkar, M

2011-03-01

A 10-year (1998-2008) retrospective study was carried out to investigate the prevalence and long-term trend of hydatid disease in slaughtered herbivores in the large complex abattoir of Ahwaz (the capital of Khuzestan province, south-western Iran). A total of 3,583,417 animals including 2,815,982 sheep, 427,790 goats and 339,645 cattle were inspected macroscopically for hydatid cysts in the 10-year period, and overall 155,555 (4.24%) livers and 228,172 (6.37%) lungs were condemned. Cystic echinococcosis (CE) was responsible for 36.08% and 48.04% of total liver and lung condemnations, respectively. The prevalence of pulmonary hydatid disease in sheep, goats and cattle was 2.22, 5.43 and 6.99%, respectively; on the other hand, the prevalence of hepatic hydatid disease for those animals was 1.26, 2.57 and 2.80%, respectively. Data showed an overall downward long-term trend for CE in all livestock slaughtered during the study period (P < 0.01). Lung condemnation due to CE was significantly more common than liver condemnation for each animal separately (P < 0.001). The prevalence of liver and lung hydatidosis in sheep was significantly lower than that in other livestock (P < 0.001). The prevalence of hydatid disease recovered from the sheep, cattle and goats varied in different seasons, but there was no statistical difference between various seasons. The odds ratio of lung and liver condemnations due to hydatidosis showed a slightly different pattern in some years; however, the overall declining trend was still observed. The total annual economic loss incurred due to hydatidosis in all ruminants slaughtered at Ahwaz municipal abattoir was estimated to be US$459,659.6, based on the market prices in the year 2008. This number corresponds to a loss of US$300,620.4 for cattle, US$123,490.0 for sheep and US$35,549.2 for goats. The current results provide baseline data for the future monitoring of this potentially important disease in the region, and also suggest that a

[How can we prevent alveolar echinococcosis? Ecosystem and risk management viewpoints].

PubMed

Konno, Keita; Oku, Yuzaburo; Kamiya, Masao; Doi, Rikuo; Tamashiro, Hidehiko

2002-01-01

This article focuses on understanding epidemiological features of alveolar echinococcosis and discussing its prevention and control, especially from a viewpoint of the ecosystem and risk management. Publications on alveolar echinococcosis throughout the world were systematically reviewed with special reference to ecology, epidemiology and countermeasures. Alveolar echinococcosis, caused by accidental infection with larva of the parasite Echinococcus multilocularis is fatal to humans unless diagnosed at an early stage. No effective control measures have been identified so far because it is difficult to fully understand the ecology of the parasite and its intermediate and definitive hosts. It is also not easy to determine the precise infection route to humans mainly because of the long latent period. In Hokkaido, infection rates among red foxes have recently risen even in low endemic districts. Not only stray and domestic dogs but also some pigs in Hokkaido have been found to be infected. While the number of reported human cases is still small, around 10 cases per year, local residents seem to be threatened with the risk of infection. We predict that the incidence of alveolar echinococcosis among humans in Japan will increase in the near future if no effective preventive measures are conducted. In addition, Echinococcus multilocularis infection has the potential to affect the economy of Hokkaido because of its impact on the agricultural and tourist industries. Well-designed epidemiological surveys are therefore urgently required, in the context of ecosystem and risk management prior to large outbreaks. International collaboration is also desired.

[Evaluation of the radiological, biochemical and serological parameters of patients prediagnosed as cystic echinococcosis in Çorum, Turkey].

PubMed

Güreser, A Semra; Özcan, Oğuzhan; Özünel, Leyla; Boyacıoğlu, Zehra İlkay; Taylan Özkan, Ayşegül

2015-04-01

Cystic echinococcosis (CE) is a zoonosis caused by Echinococcus granulosus. It is difficult to diagnose CE by clinical symptoms alone, therefore, radiological and serological examinations should be conducted as well. The aims of this retrospective study were to evaluate the biochemical, hemogram, serological and radiological findings of patients prediagnosed as CE, and to survey epidemiological data to detect the status of the disease in our region. A total of 253 patients (148 female, 105 male) who were admitted to Hitit University Training and Research Hospital in Corum province (located in the central Black Sea Region of Turkey), between October 2009 to July 2013, were included in the study. Serum samples collected from the patients were analyzed by indirect hemagglutination (IHA) test, in the Microbiology Reference Laboratories of the Turkish Public Health Institute, and 1/160 and higher titers were considered positive. Twenty-three (15.5%) of female patients and nine (8.6%) of male patients, with a total of 32 (12.7%) were found to be seropositive. The difference between the gender was not statistically significant (X2= 2.72). The age range of the 32 seropositive patients was between 16-90 years (mean: 51), and of them 24 (75%) being over 40 years old was found as statistically significant (X2= 22.45). All of the seropositive patients presented radiological findings diagnosed with ultrasonography and computed tomography. Additionally, it was noticed that two patients (one male, one female) who were seronegative by IHA test, have passed a CE operation and the diagnosis was confirmed with pathological findings. Of the patients 43.8% were admitted to general surgery, followed by infectious diseases (21.9%), gastroenterology (21.9%) and other (12.5%) clinics. Radiological diagnosis showed that 31 (96.9%) of seropositive patients had CE in the liver, of them two (6.3%) also had lung involvement, while one patient (3.1%) had intraperitoneal involvement alone

Complete Resolution of an Alveolar Echinococcosis Liver Lesion Following Percutaneous Treatment

DOE Office of Scientific and Technical Information (OSTI.GOV)

Koroglu, Mert; Akhan, Okan; Gelen, Mustafa Tekinalp

Herein we present a 63-year-old male patient with a solid hepatic alveolar echinococcosis diagnosed by surgical biopsy. His liver lesion, which was infected, was drained by percutaneous catheterization. The lesion surprisingly disappeared completely after the treatment. The patient was followed-up without any symptoms for 20 months after the drainage. As alveolar echinococcosis of the liver behaves like a slow-growing liver cancer, the disappearance of our patient's lesion was a very unusual and rare outcome, which, to the best of our knowledge, has never been published in the literature.

CCR7(lo)PD-1(hi) CXCR5(+) CD4(+) T cells are positively correlated with levels of IL-21 in active and transitional cystic echinococcosis patients.

PubMed

Zhang, Fengbo; Pang, Nannan; Zhu, Yuejie; Zhou, Dexian; Zhao, Hui; Hu, Jinwei; Ma, Xiumin; Li, Jun; Wen, Hao; Samten, Buka; Fan, Haining; Ding, Jianbing

2015-10-26

In our study, we investigated whether circulating T follicular helper (Tfh) and the related cytokines are involved in human cystic echinococcosis (CE). A total of 64 patients with CE and 30 healthy controls were enrolled in this study. Percentages of CCR7(lo)PD-1(hi) cells within CXCR5(+) CD4(+) T cells (circulating Tfh cells) were detected by flow cytometry. Levels of IL-21 and IL-4 in peripheral blood were detected by cytometric bead array. The mRNA expression of IL-21, IL-4, Bcl-6, and Blimp-1 in peripheral blood mononuclear cells (PBMCs) were measured by real-time PCR. Levels of IgG1, IgG2, IgG3, and IgG4 in the patients' sera were measured using enzyme-linked immunosorbent assay. Percentages of circulating Tfh cells were significantly increased in the CE1, CE2, and CE3 groups (p < 0.05). The concentrations of IL-21 and IL-4 in the serum were significantly increased in CE1, CE2, and CE3 groups (p < 0.05). IL-21 was positively correlated with circulating Tfh cells in CE3 group (r = 0.779, p < 0.05). The mRNA levels of IL-21, IL-4, and Bcl-6 were increased in CE1, CE2, and CE3 groups. Levels of IgG1 and IgG4 in patients' sera were increased in CE1, CE2, and CE3 groups. Levels of IgG2 and IgG3 were increased in CE4-5 group. Additionally, after stimulation with hydatid fluid in vitro, the levels of circulating Tfh cells, IL-21 and IL-4 in PBMCs isolated from CE patients were significantly increased (p < 0.05). The levels of circulating Tfh and related cytokines were significantly increased in CE patients, suggesting that they are involved in human CE.

Cystic echinococcosis in marketed offal of sheep in Basrah, Iraq: Abattoir-based survey and a probabilistic model estimation of the direct economic losses due to hydatid cyst.

PubMed

Abdulhameed, Mohanad F; Habib, Ihab; Al-Azizz, Suzan A; Robertson, Ian

2018-02-01

Cystic echinococcosis (CE) is a highly endemic parasitic zoonosis in Iraq with substantial impacts on livestock productivity and human health. The objectives of this study were to study the abattoir-based occurrence of CE in marketed offal of sheep in Basrah province, Iraq, and to estimate, using a probabilistic modelling approach, the direct economic losses due to hydatid cysts. Based on detailed visual meat inspection, results from an active abattoir survey in this study revealed detection of hydatid cysts in 7.3% (95% CI: 5.4; 9.6) of 631 examined sheep carcasses. Post-mortem lesions of hydatid cyst were concurrently present in livers and lungs of more than half (54.3% (25/46)) of the positive sheep. Direct economic losses due to hydatid cysts in marketed offal were estimated using data from government reports, the one abattoir survey completed in this study, and expert opinions of local veterinarians and butchers. A Monte-Carlo simulation model was developed in a spreadsheet utilizing Latin Hypercube sampling to account for uncertainty in the input parameters. The model estimated that the average annual economic losses associated with hydatid cysts in the liver and lungs of sheep marketed for human consumption in Basrah to be US$72,470 (90% Confidence Interval (CI); ±11,302). The mean proportion of annual losses in meat products value (carcasses and offal) due to hydatid cysts in the liver and lungs of sheep marketed in Basrah province was estimated as 0.42% (90% CI; ±0.21). These estimates suggest that CE is responsible for considerable livestock-associated monetary losses in the south of Iraq. These findings can be used to inform different regional CE control program options in Iraq.

[Analysis of human cyst echinococcosis in a northern Chilean region from 2008 to 2012].

PubMed

Soto-Aguilar, Andrea; Junod, Tania; Campillay, Mauricio; Acosta-Jamett, Gerardo; Landaeta-Aqueveque, Carlos

2017-05-01

The Coquimbo Region in Chile has the highest incidence of hydatidosis in central and northern Chile. To analyze the incidence of human cystic echinococcosis (CE) in the Coquimbo Region between 2008 and 2012. Official records of reported cases were analyzed. The association of the location, age and sex with the presence/absence of CE was analyzed by means of logistic regressions. The incidence was reported as cases per 100,000 inhabitants. The mean annual incidence was 2.6 cases per 100,000 inhabitants, which ranged from 3.1 (2010) to 1.9 (2012) cases. The districts with higher incidence were Punitaqui (21.2 cases), Paihuano (9 cases), Rio Hurtado (8 cases), Canela (8.1 cases), Monte Patria (7.9 cases), Vicuña (6.9 cases) and Combarbalá (6.9 cases). The incidence in these locations was significantly higher than elsewhere in the region. Males had a significantly higher incidence than females (3.3 and 1.8 respectively). Incidence increased significantly with age, notably increasing after 45 years of age. Rural locations had the highest incidences of CE in the Coquimbo Region. The disease was reported more commonly in adults and mainly in men.

Polycystic echinococcosis in Colombia: the larval cestodes in infected rodents.

PubMed

Morales, G A; Guzman, V H; Wells, E A; Angel, D

1979-07-01

Described are the characteristics of the polycystic larval cestodes found in an endemic area of echinococcosis in the Easter Plains of Colombia and the tissue reaction evoked in infected rodents. Of 848 free-ranging animals examined, polycystic hydatids were found in 44/93 Cuniculus paca and 1/369 Proechimys sp. None of 20 Dasyprocta fuliginosa examined was infected, but hunters provided a heart with hydatid cysts and information about two additional animals with infected livers. Recognition of an endemic area of polycystic echinococcosis provides a means to investigate the life cycle of the parasites and to study the histogenesis of the larval cestodes in susceptible laboratory animals.

Time Prediction Models for Echinococcosis Based on Gray System Theory and Epidemic Dynamics.

PubMed

Zhang, Liping; Wang, Li; Zheng, Yanling; Wang, Kai; Zhang, Xueliang; Zheng, Yujian

2017-03-04

Echinococcosis, which can seriously harm human health and animal husbandry production, has become an endemic in the Xinjiang Uygur Autonomous Region of China. In order to explore an effective human Echinococcosis forecasting model in Xinjiang, three grey models, namely, the traditional grey GM(1,1) model, the Grey-Periodic Extensional Combinatorial Model (PECGM(1,1)), and the Modified Grey Model using Fourier Series (FGM(1,1)), in addition to a multiplicative seasonal ARIMA(1,0,1)(1,1,0)₄ model, are applied in this study for short-term predictions. The accuracy of the different grey models is also investigated. The simulation results show that the FGM(1,1) model has a higher performance ability, not only for model fitting, but also for forecasting. Furthermore, considering the stability and the modeling precision in the long run, a dynamic epidemic prediction model based on the transmission mechanism of Echinococcosis is also established for long-term predictions. Results demonstrate that the dynamic epidemic prediction model is capable of identifying the future tendency. The number of human Echinococcosis cases will increase steadily over the next 25 years, reaching a peak of about 1250 cases, before eventually witnessing a slow decline, until it finally ends.

Time Prediction Models for Echinococcosis Based on Gray System Theory and Epidemic Dynamics

PubMed Central

Zhang, Liping; Wang, Li; Zheng, Yanling; Wang, Kai; Zhang, Xueliang; Zheng, Yujian

2017-01-01

Echinococcosis, which can seriously harm human health and animal husbandry production, has become an endemic in the Xinjiang Uygur Autonomous Region of China. In order to explore an effective human Echinococcosis forecasting model in Xinjiang, three grey models, namely, the traditional grey GM(1,1) model, the Grey-Periodic Extensional Combinatorial Model (PECGM(1,1)), and the Modified Grey Model using Fourier Series (FGM(1,1)), in addition to a multiplicative seasonal ARIMA(1,0,1)(1,1,0)4 model, are applied in this study for short-term predictions. The accuracy of the different grey models is also investigated. The simulation results show that the FGM(1,1) model has a higher performance ability, not only for model fitting, but also for forecasting. Furthermore, considering the stability and the modeling precision in the long run, a dynamic epidemic prediction model based on the transmission mechanism of Echinococcosis is also established for long-term predictions. Results demonstrate that the dynamic epidemic prediction model is capable of identifying the future tendency. The number of human Echinococcosis cases will increase steadily over the next 25 years, reaching a peak of about 1250 cases, before eventually witnessing a slow decline, until it finally ends. PMID:28273856

Gamma-Ray Treatment of Echinococcus Protoscoleces prior to Implantation in Mice Reduces Echinococcosis.

PubMed

Yuan, Qing; Li, Bo; Jiang, Shiping; Zhao, Qiang; Duo, Ji; Huang, Xiang

2016-01-01

Echinococcosis is a serious parasitic disease caused by Echinococcus tapeworms. Protoscoleces are sometimes released during surgical treatment for hydatid cysts, causing the recurrence of echinococcosis. Protoscoleces may be susceptible to radiation therapy. In this study Echinococcus protoscoleces were cultured in vitro and then divided into four different γ-ray irradiation dose groups (10 Gy, 20 Gy, 40 Gy, and 80 Gy) and a blank group. The protoscoleces were then implanted into the abdominal cavity of mice. Four months later, we observed that the incidence and weight of cysts declined with the increase of irradiation dose. γ-ray irradiation can suppress the generation of Echinococcus originated from protoscolex, the reason of which is due to the damaging to the structure of Echinococcus. Irradiation may prevent echinococcosis recurrence after surgical removal of hydatid cysts.

Gamma-Ray Treatment of Echinococcus Protoscoleces prior to Implantation in Mice Reduces Echinococcosis

PubMed Central

Yuan, Qing; Jiang, Shiping; Zhao, Qiang; Duo, Ji; Huang, Xiang

2016-01-01

Echinococcosis is a serious parasitic disease caused by Echinococcus tapeworms. Protoscoleces are sometimes released during surgical treatment for hydatid cysts, causing the recurrence of echinococcosis. Protoscoleces may be susceptible to radiation therapy. In this study Echinococcus protoscoleces were cultured in vitro and then divided into four different γ-ray irradiation dose groups (10 Gy, 20 Gy, 40 Gy, and 80 Gy) and a blank group. The protoscoleces were then implanted into the abdominal cavity of mice. Four months later, we observed that the incidence and weight of cysts declined with the increase of irradiation dose. γ-ray irradiation can suppress the generation of Echinococcus originated from protoscolex, the reason of which is due to the damaging to the structure of Echinococcus. Irradiation may prevent echinococcosis recurrence after surgical removal of hydatid cysts. PMID:27610384

Polycystic echinococcosis in Pacas, Amazon region, Peru.

PubMed

Mayor, Pedro; Baquedano, Laura E; Sanchez, Elisabeth; Aramburu, Javier; Gomez-Puerta, Luis A; Mamani, Victor J; Gavidia, Cesar M

2015-03-01

In the Peruvian Amazon, paca meat is consumed by humans. To determine human risk for polycystic echinococcosis, we examined wild pacas from 2 villages; 15 (11.7%) of 128 were infected with Echinococcus vogeli tapeworms. High E. vogeli prevalence among pacas indicates potential risk for humans living in E. vogeli-contaminated areas.

Polycystic Echinococcosis in Pacas, Amazon Region, Peru

PubMed Central

Mayor, Pedro; Baquedano, Laura E.; Sanchez, Elisabeth; Aramburu, Javier; Gomez-Puerta, Luis A.; Mamani, Victor J.

2015-01-01

In the Peruvian Amazon, paca meat is consumed by humans. To determine human risk for polycystic echinococcosis, we examined wild pacas from 2 villages; 15 (11.7%) of 128 were infected with Echinococcus vogeli tapeworms. High E. vogeli prevalence among pacas indicates potential risk for humans living in E. vogeli–contaminated areas. PMID:25695937

[Study on the ARIMA model application to predict echinococcosis cases in China].

PubMed

En-Li, Tan; Zheng-Feng, Wang; Wen-Ce, Zhou; Shi-Zhu, Li; Yan, Lu; Lin, Ai; Yu-Chun, Cai; Xue-Jiao, Teng; Shun-Xian, Zhang; Zhi-Sheng, Dang; Chun-Li, Yang; Jia-Xu, Chen; Wei, Hu; Xiao-Nong, Zhou; Li-Guang, Tian

2018-02-26

To predict the monthly reported echinococcosis cases in China with the autoregressive integrated moving average (ARIMA) model, so as to provide a reference for prevention and control of echinococcosis. SPSS 24.0 software was used to construct the ARIMA models based on the monthly reported echinococcosis cases of time series from 2007 to 2015 and 2007 to 2014, respectively, and the accuracies of the two ARIMA models were compared. The model based on the data of the monthly reported cases of echinococcosis in China from 2007 to 2015 was ARIMA (1, 0, 0) (1, 1, 0) 12 , the relative error among reported cases and predicted cases was -13.97%, AR (1) = 0.367 ( t = 3.816, P < 0.001), SAR (1) = -0.328 ( t = -3.361, P = 0.001), and Ljung-Box Q = 14.119 ( df = 16, P = 0.590) . The model based on the data of the monthly reported cases of echinococcosis in China from 2007 to 2014 was ARIMA (1, 0, 0) (1, 0, 1) 12 , the relative error among reported cases and predicted cases was 0.56%, AR (1) = 0.413 ( t = 4.244, P < 0.001), SAR (1) = 0.809 ( t = 9.584, P < 0.001), SMA (1) = 0.356 ( t = 2.278, P = 0.025), and Ljung-Box Q = 18.924 ( df = 15, P = 0.217). The different time series may have different ARIMA models as for the same infectious diseases. It is needed to be further verified that the more data are accumulated, the shorter time of predication is, and the smaller the average of the relative error is. The establishment and prediction of an ARIMA model is a dynamic process that needs to be adjusted and optimized continuously according to the accumulated data, meantime, we should give full consideration to the intensity of the work related to infectious diseases reported (such as disease census and special investigation).

Cystic Duct Closure by Sealing With Bipolar Electrocoagulation

PubMed Central

Damgaard, B.; Jorgensen, L. N.; Larsen, S. S.; Kristiansen, V. B.

2010-01-01

Background: Cystic duct leakage after cholecystectomy is not uncommon and is a potentially serious complication. The aim of this study was to assess a bipolar sealing system (LigaSure®) for closure of the cystic duct. Methods: The records from consecutive laparoscopic cholecystectomies performed in 2 hospitals with closure of the cystic duct with LigaSure after informed consent were recorded and complications and morbidity registered. The records were compared with those of patients undergoing laparoscopic cholecystectomy with closure of the cystic duct with clips during the same period. Results: During the study period, 218 laparoscopic cholecystectomies were performed; 102 of these were performed with the LigaSure. One patient was excluded due to violation of the protocol. We experienced no cases of cystic duct leakage, but in one patient, bile leakage from the gallbladder bed was observed probably due to a small aberrant duct. Conclusion: The LigaSure system was safe and effective for closure and division of the cystic duct in laparoscopic cholecystectomy. PMID:20412641

[Transmission dynamic model for echinococcosis granulosus: establishment and application].

PubMed

Yang, Shi-Jie; Wu, Wei-Ping

2009-06-01

A dynamic model of disease can be used to quantitatively describe the pattern and characteristics of disease transmission, predict the disease status and evaluate the efficacy of control strategy. This review summarizes the basic transmission dynamic models of echinococcosis granulosus and their application.

Computed Tomography Perfusion Imaging for the Diagnosis of Hepatic Alveolar Echinococcosis

PubMed Central

Sade, Recep; Kantarci, Mecit; Genc, Berhan; Ogul, Hayri; Gundogdu, Betul; Yilmaz, Omer

2018-01-01

Objective: Alveolar echinococcosis (AE) is a rare life-threatening parasitic infection. Computed tomography perfusion (CTP) imaging has the potential to provide both quantitative and qualitative information about the tissue perfusion characteristics. The purpose of this study was the examination of the characteristic features and feasibility of CTP in AE liver lesions. Material and Methods: CTP scanning was performed in 25 patients who had a total of 35 lesions identified as AE of the liver. Blood flow (BF), blood volume (BV), portal venous perfusion (PVP), arterial liver perfusion (ALP), and hepatic perfusion indexes (HPI) were computed for background liver parenchyma and each AE lesion. Results: Significant differences were detected between perfusion values of the AE lesions and background liver tissue. The BV, BF, ALP, and PVP values for all components of the AE liver lesions were significantly lower than the normal liver parenchyma (p<0.01). Conclusions: We suggest that perfusion imaging can be used in AE of the liver. Thus, the quantitative knowledge of perfusion parameters are obtained via CT perfusion imaging. PMID:29531482

Taeniasis, cysticercosis and echinococcosis in Thailand.

PubMed

Waikagul, Jitra; Dekumyoy, Paron; Anantaphruti, Malinee T

2006-01-01

Taeniasis is one of the major food-borne parasitic zoonoses in Thailand. During the years 1957-1997, the prevalence was low in most parts of the country. Recent (2000-2005) country prevalence was lower than 1%. A high prevalence (5.9%) was found among 1450 villagers from 30 villages in the North, and among 1233 stool samples from 19 provinces in the Northeast (2.8%). Taenia saginata was the dominant species. Cysticercosis in Thailand is somewhat under-reported/recorded. During the period 1965-2005, diagnosis was based on techniques other than serodiagnosis, giving a total of cysticercosis cases of less than 500. However, an immunoblot technique using delipidized cyst antigen showed 314 positive cases out of 754 samples tested in 2000-2005. Reports of neurocysticercosis appeared more often than cutaneous cysticercosis. A total of 24 cases of echinococcosis, mostly hydatid cysts (only 2 cases of alveolar cysts), were recorded during 1936-2005. These records included 3 cases of foreigners seeking surgery in hospitals in Bangkok. Most Thai patients were migrant workers from the Middle East, and only a few cases were indigenous. The prevalence of cysticercosis and echinococcosis is increasing resulting from sensitive modern diagnostic tests. Taeniasis will persist in Thailand as the consumption of raw/half-cooked meat dishes is still a normal practice for Thai people.

Immunodiagnosis of Echinococcus Infections: Confirmatory Testing and Species Differentiation by a New Commercial Western Blot

PubMed Central

Liance, Martine; Janin, Veronique; Bresson-Hadni, Solange; Vuitton, Dominique-Angele; Houin, Rene; Piarroux, Renaud

2000-01-01

The Echinococcus Western Blot IgG (LDBIO Diagnostics, Lyon, France), using a whole larval antigen from Echinococcus multilocularis, was evaluated for serodiagnosis and differentiation between two human parasitic infections of worldwide importance: cystic echinococcosis, due to Echinococcus granulosus, and alveolar echinococcosis, due to E. multilocularis. Fifty and 61 serum samples from patients with cystic and alveolar echinococcosis, respectively, were used for assessing diagnostic sensitivity. The sensitivity of the assay was compared with those of screening tests used for these applications. Sera used for assessing cross-reactivities were from 154 patients with other diseases, either parasitic or not. The assay allowed the detection of serum immunoglobulin G antibodies in 97% of Echinococcus-infected patients. It had a higher sensitivity than screening assays for the detection for each echinococcosis. The assay allowed us to correctly distinguish between E. granulosus- and E. multilocularis-infected patients in 76% of cases. It did not allow us to distinguish active from inactive forms of both echinococcoses. The occurrence of cross-reactivities with neurocysticercosis indicates the necessity for retesting sera with species-specific antigens, for rare patients with neurologic disorders. This study shows the usefulness of the commercially available Echinococcus Western Blot IgG for the serological confirmation of human echinococcosis. PMID:11015390

Multisystem involvement of alveolar echinococcosis in a child.

PubMed

Kantarci, Mecit; Bayraktutan, Ummugulsum; Pirimoglu, Berhan; Ogul, Hayri; Oral, Akgun; Eren, Suat; Gundogdu, Betul

2014-11-13

Alveolar echinococcosis (AE) is a chronic progressive infestation inducing a slowly progressing, life-threatening tumor-like growth in the liver. It may spread to other organs by regional extension or hematogenous or lymphatic metastasis. Herein, we report a fifteen-year-old patient diagnosed with AE of the liver and simultaneous lung and brain metastasis with a literature review.

[Relevance of parasitological examinations for the clinical course, epidemiology and prevention of alveolar echinococcosis - experiences of more than two decades in Austria].

PubMed

Auer, Herbert

2006-01-01

Alveolar echinococcosis has been known in Austria since 1854. At the beginning of the 20th century the Austrian researcher on echinococcosis, Adolf Posselt, has realized already the enormous medical relevance of serological tests for the diagnosis of alveolar echinococcosis as well as for preventive purposes. Since the beginning of the 1980ies a long-term study about the epidemiology and serodiagnosis of alveolar echinococcosis has started in the Department of Medical Parasitology of the Clinical Institute of Hygiene and Medical Microbiology of the University of Vienna. Today a highly sensitive enzyme-linked immunosorbent assay as well as a highly specific westernblot system are available for the serological diagnosis of human alveolar echinococcosis cases, for the serological monitoring of the disease as well as for seroepidemiological and preventive medical studies. In addition, molecular biological tools for the detection of E. multilocularis (as well as E. granulosus strain) specific DNA in surgical resected and bioptic materials complete our diagnostic spectrum of methods.

Evaluation of the impact of 2 years of a dosing intervention on canine echinococcosis in the Alay Valley, Kyrgyzstan.

PubMed

VAN Kesteren, F; Mastin, A; Torgerson, P R; Mytynova, Bermet; Craig, P S

2017-09-01

Echinococcosis is a re-emerging zoonotic disease in Kyrgyzstan. In 2012, an echinococcosis control scheme was started that included dosing owned dogs in the Alay Valley, Kyrgyzstan with praziquantel. Control programmes require large investments of money and resources; as such it is important to evaluate how well these are meeting their targets. However, problems associated with echinococcosis control schemes include remoteness and semi-nomadic customs of affected communities, and lack of resources. These same problems apply to control scheme evaluations, and quick and easy assessment tools are highly desirable. Lot quality assurance sampling was used to assess the impact of approximately 2 years of echinococcosis control in the Alay valley. A pre-intervention coproELISA prevalence was established, and a 75% threshold for dosing compliance was set based on previous studies. Ten communities were visited in 2013 and 2014, with 18-21 dogs sampled per community, and questionnaires administered to dog owners. After 21 months of control efforts, 8/10 communities showed evidence of reaching the 75% praziquantel dosing target, although only 3/10 showed evidence of a reduction in coproELISA prevalence. This is understandable, since years of sustained control are required to effectively control echinococcosis, and efforts in the Alay valley should be and are being continued.

The Molecular Biology of Adenoid Cystic Carcinoma

PubMed Central

Liu, Jia; Shao, Chunbo; Tan, Marietta L.; Mu, David; Ferris, Robert L.; Ha, Patrick K.

2011-01-01

Background Adenoid cystic carcinoma (ACC) is an unusual salivary gland malignancy that remains poorly understood. Standard treatment, including surgery with postoperative radiation therapy have attained reasonable local control rates, but the propensity for distant metastases has limited any improvement in survival over time. Our understanding of the molecular mechanisms driving adenoid cystic carcinoma is quite rudimentary, due to the infrequent nature of its occurrence. Methods An extensive literature review was performed on salivary gland adenoid cystic carcinoma and basic science research findings. Results This review highlights many findings that are emerging about the carcinogenesis of ACC including cytogenetics, tumor suppressor genes, oncogenes, epigenetic alterations, mitochondrial alterations, and biomarker studies. Conclusions While there have been many discoveries, much still remains unknown about this rare malignancy. PMID:22006498

A Case Report of Cystic Pheochromocytoma.

PubMed

Junejo, Shoaib Z; Tuli, Sandeep; Heimann, David M; Sachmechi, Issac; Reich, David

2017-07-25

BACKGROUND Pheochromocytoma is a rare catecholamine-producing tumor with an estimated incidence of less than 0.1% in the global population. We present a case of cystic pheochromocytoma that was diagnosed as an incidental finding. The patient presented with abdominal pain and had a history of hypertension. CASE REPORT A 64-year-old man with hypertension presented with a clinical history of intermittent abdominal pain for one year. He denied sweating, palpitations, headache or back pain. He was found to have an elevated blood pressure of 170/90 and no palpable abdominal mass. Contrast-enhanced computed tomography (CT) imaging of the abdomen and pelvis were performed that showed cystic mass measuring 9 cm in diameter arising from the left adrenal gland with contrast-enhancing mural nodules. Magnetic resonance imaging (MRI) confirmed the cystic nature of the mass. Laboratory analysis showed an elevated plasma normetanephrine (NMN) of 1,087 pg/ml and metanephrine (MN) of 372 pg/ml; 24-hour urine showed elevated levels of NMN and MN, 3,002 mg/24 h and 1,596 mg/24 h, respectively. Given the laboratory and radiologic findings, a diagnosis of cystic pheochromocytoma was made. After controlling blood pressure with the alpha-blocker, doxazosin, the patient was hydrated and scheduled for an elective adrenalectomy. The histopathology of the excised adrenal gland was consistent with a cystic pheochromocytoma. CONCLUSIONS Cystic pheochromocytoma is a very rare tumor that may present without symptoms. The clinical course of cystic pheochromocytoma is similar to that of solid pheochromocytoma. Early surgical intervention is recommended, following blood pressure control with an alpha-blocker, and adequate hydration.

Peripheral calcifying cystic odontogenic tumour of the maxillary gingiva

PubMed Central

2012-01-01

Background Odontogenic tumors are lesions that are derived from remnants of the components of the developing tooth germ. The calcifying cystic odontogenic tumor or calcifying odontogenic cyst is a benign cystic neoplasm of odontogenic origin that is characterized by an ameloblastoma-like epithelium and ghost cells. Calcifying cystic odontogenic tumor may be centrally or peripherally located, and its ghost cells may exhibit calcification, as first described by Gorlin in 1962. Most peripheral calcifying cystic odontogenic tumors are located in the anterior gingiva of the mandible or maxilla. Case presentation Authors report a rare case of a peripheral calcifying cystic odontogenic tumor of the maxillary gingiva. A 39-year-old male patient presented with a fibrous mass on the attached buccal gingiva of the upper left cuspid teeth. It was 0.7-cm-diameter, painless and it was clinically diagnosed as a peripheral ossifying fibroma. After an excisional biopsy, the diagnosis was peripheric calcifying cystic odontogenic tumor. The patient was monitored for five years following the excision, and no recurrence was detected. Conclusions All biopsy material must be sent for histological examination. If the histological examination of gingival lesions with innocuous appearance is not performed, the frequency of peripheral calcifying cystic odontogenic tumor and other peripheral odontogenic tumors may be underestimated. PMID:22917449

Cystic fibrosis - resources

MedlinePlus

Resources - cystic fibrosis ... The following organizations are good resources for information on cystic fibrosis : Cystic Fibrosis Foundation -- www.cff.org March of Dimes -- www.marchofdimes.org/baby/cystic-fibrosis-and- ...

Polycystic echinococcosis in the state of Acre, Brazil: contribution to patient diagnosis, treatment and prognosis

PubMed Central

de Siqueira, Nilton Ghiotti; de Siqueira, Cláudia Maria Villar Maziero; Rodrigues-Silva, Rosângela; Soares, Manoel do Carmo P; Póvoa, Marinete Marins

2013-01-01

The lack of knowledge regarding polycystic hydatid disease results in delayed or even incorrect diagnosis. The lack of systematic information regarding treatment also makes it difficult to assess the results and prognosis in patients with peritoneal and hepatic lesions caused by Echinococcus vogeli. Here we describe the clinical features of patients, propose a radiological classification protocol and describe a therapeutic option for the treatment of hydatid disease that previously had only been used for cases of cystic echinococcosis (Echinococcus granulosus). A prospective cohort study was initiated in 1999 and by 2009 the study included 60 patients. These patients were classified according to the PNM classification (parasite lesion, neighbouring organ invasion and metastases) and placed in one of three therapeutic modalities: (i) chemotherapy with albendazole at a dose of 10 mg/kg/day, (ii) surgical removal of cysts or (iii) percutaneous puncture of the cysts via puncture, aspiration, injection and re-aspiration (PAIR). The results were stratified according to therapeutic outcome: "cure", "clinical improvement", "no improvement", "death" or "no information". The PNM classification was useful in indicating the appropriate therapy in cases of polycystic hydatid disease. In conclusion, surgical therapy produced the best clinical results of all the therapies studied based on "cure" and "clinical improvement" outcomes. The use of PAIR for treatment requires additional study. PMID:23903966

[Unilocular cyst hydatid cases diagnosed between 1998-2005 in the Parasitology Laboratory of Yüzüncü Yıl University Research and Training Hospital].

PubMed

Yılmaz, Hasan; Cengiz, Zeynep Taş; Ciçek, Mutalip

2013-01-01

This study was carried out to contribute to the information of the spread of disease in our country by determining the distribution of cystic echinococcosis (CE) on people in our region and to demonstrate the importance of it for our region. In this study, the blood serum samples of a total of 558 patients, which were sent to the Parasitology Laboratory from several outpatient clinics of the Yüzüncü Yıl University Research and Training Hospital between 01.09.1998-01.09.2005, were evaluated in terms of CE by using Indirect Hemaglutination Test (IHA) or ELISA methods. 25.6% of a total of 558 cystic echinococcosis suspected patients were found to be seropositive by IHA or ELISA methods and it was confirmed that the cysts obtained as a result of the positive patients' surgery were unilocular cysts. In our study, seropositivity was found in 25.7% of 303 female patients, 25.5% of 255 male patients; 33.3% of 48 pediatric 8-15 age group patients and 24.9% of 510 adult patients over the age of 15 years. Cystic echinococcosis continues to be a major public health problem in the Van province. Large-scale prevention and control programs should be implemented against this disease.

Prevalence of Serum IgG Antibodies to Cystic Echinococcus Antigen among Patients in an Uzbekistan Emergency Hospital.

PubMed

Park, Se Jin; Han, Sung Sik; Anvarov, Khikmat; Khajibaev, Abdukhakim; Choi, Min-Ho; Hong, Sung-Tae

2015-12-01

Cystic echinococcosis (CE) is one of the most widespread zoonotic helminthiases, which can last an asymptomatic infection for several years. The purpose of this study was to demonstrate serum antibody prevalence of CE among asymptomatic people in Uzbekistan using ELISA. A total of 2,547 serum samples were collected, 66 from confirmed CE patients and 2,481 of patients with other diseases than CE at a hospital in Tashkent, Uzbekistan. The serum samples were screened for CE specific IgG antibodies by ELISA using cystic fluid antigen obtained from sheep. The serum antibody positive rate was 89.4% (59/66) in CE and 3.6% (89/2,481) in other disease patients. The present ELISA recognized 89.4% sensitivity and 96.4% specificity. The ELISA absorbance of positive samples was distributed 0.271-0.971 for CE and 0.273-0.887 for other disease patients. The other disease patients with high absorbance over 0.3 were 50 (2.0%) who were presumed to be active CE patients. The patients in their 40s showed the highest positive rate of 5.2% (P=0.181), and women were 4.4% while men were 3.1% positive (P=0.136). The data confirmed that there are many asymptomatic patients of CE in Tashkent. It is indicated that CE is an endemic disease of public health importance in Uzbekistan.

Molecular discrimination of Echinococcus granulosus and Echinococcus multilocularis by sequencing and a new PCR-RFLP method with the potential use for other Echinococcus species.

PubMed

Şakalar, Çağrı; Kuk, Salih; Erensoy, Ahmet; Dağli, Adile Ferda; Özercan, İbrahim Hanifi; Çetınkaya, Ülfet; Yazar, Süleyman

2014-01-01

To develop a novel polymerase chain reaction-restriction fragment length polymorphism (PCR-RFLP) protocol using a new genomic marker sequence and a novel set of restriction enzymes in order to detect and discriminate 2 Echinococcus species, E. granulosus and E. multilocularis, found in formalin-fixed paraffin-embedded (FFPE) human tissues. DNA was isolated from 11 FFPE human tissue samples positive for cystic echinococcosis or alveolar echinococcosis. A mitochondrial genomic marker region was amplified and sequenced using a novel primer pair and a new PCR-RFLP protocol was developed for the detection and discrimination of E. granulosus and E. multilocularis using a set of restriction enzymes including AccI, MboI, MboII, and TsoI. The selected marker region was amplified using DNA isolated from FFPE human tissue samples positive for cystic echinococcosis or alveolar echinococcosis and the discrimination of E. granulosus and E. multilocularis was accomplished by use of the novel PCR-RFLP method. In this PCR-RFLP protocol, use of any single restriction enzyme is enough for the discrimination of E. granulosus and E. multilocularis. The PCR-RFLP protocol can be potentially used for the discrimination of 5 other Echinococcus species: E. oligarthus, E. shiquicus, E. ortleppi, E. canadensis, and E. vogeli.

Non-neoplastic cystic and cystic-like lesions of the pancreas: may mimic pancreatic cystic neoplasms.

PubMed

Goh, Brian K P; Tan, Yu-Meng; Chung, Yaw-Fui A; Chow, Pierce K H; Ong, Hock-Soo; Lim, Dennis T H; Wong, Wai-Keong; Ooi, London L P J

2006-05-01

Cystic lesions of the pancreas consist of a broad range of pathological entities. With the exception of the pancreatic pseudocyst, these are usually caused by pancreatic cystic neoplasms. Non-neoplastic pancreatic cystic and cystic-like lesions are extremely rare. In the present article, the surgical experience with these unusual entities over a 14-year period is reported. Between 1991 and 2004, all patients who underwent surgical exploration for a cystic lesion of the pancreas were retrospectively reviewed. Patients with a pancreatic pseudocyst were excluded. There were 106 patients of whom 8 (7.5%) had a final pathological diagnosis consistent with a non-neoplastic pancreatic cystic or cystic-like lesion, including 3 patients with a benign epithelial cyst, 2 with a pancreatic abscess (one tuberculous and one foreign body), 2 with mucous retention cysts and 1 with a mucinous non-neoplastic cyst. These eight patients are the focus of this study. There were six female and two male patients with a median age of 61.5 years (range, 41-71 years). All the patients were of Asian origin including seven Chinese and one Indian. Four of the patients were asymptomatic and their pancreatic cysts were discovered incidentally on radiological imaging for other indications. All the patients underwent preoperative radiological investigations, including ultrasonography, computed tomography or magnetic resonance imaging, which showed a cystic lesion of the pancreas. Three patients, all of whom were symptomatic, were diagnosed preoperatively with a malignant cystic neoplasm on the basis of radiological imaging. Two patients were eventually found to have a pancreatic abscess, one tuberculous and the other, secondary to foreign body perforation. The third patient was found on final histology to have chronic pancreatitis with retention cysts. The remaining five patients had a preoperative diagnosis of an indeterminate cyst; on pathological examination, they were found to have a benign

Network for Early Onset Cystic Kidney Diseases-A Comprehensive Multidisciplinary Approach to Hereditary Cystic Kidney Diseases in Childhood.

PubMed

König, Jens Christian; Titieni, Andrea; Konrad, Martin

2018-01-01

Hereditary cystic kidney diseases comprise a complex group of genetic disorders representing one of the most common causes of end-stage renal failure in childhood. The main representatives are autosomal recessive polycystic kidney disease, nephronophthisis, Bardet-Biedl syndrome, and hepatocyte nuclear factor-1beta nephropathy. Within the last years, genetic efforts have brought tremendous progress for the molecular understanding of hereditary cystic kidney diseases identifying more than 70 genes. Yet, genetic heterogeneity, phenotypic variability, a lack of reliable genotype-phenotype correlations and the absence of disease-specific biomarkers remain major challenges for physicians treating children with cystic kidney diseases. To tackle these challenges comprehensive scientific approaches are urgently needed that match the ongoing "revolution" in genetics and molecular biology with an improved efficacy of clinical data collection. Network for early onset cystic kidney diseases (NEOCYST) is a multidisciplinary, multicenter collaborative combining a detailed collection of clinical data with translational scientific approaches addressing the genetic, molecular, and functional background of hereditary cystic kidney diseases. Consisting of seven work packages, including an international registry as well as a biobank, NEOCYST is not only dedicated to current scientific questions, but also provides a platform for longitudinal clinical surveillance and provides precious sources for high-quality research projects and future clinical trials. Funded by the German Federal Government, the NEOCYST collaborative started in February 2016. Here, we would like to introduce the rationale, design, and objectives of the network followed by a short overview on the current state of progress.

[The clinical, serological and morphological analysis of 91 patients with cystic echinococcosis following surgery].

PubMed

Mıman, Ozlem; Atambay, Metin; Aydin, Nasuhi Engin; Daldal, Nilgün

2010-01-01

Cystic echinococcocis (CE) is a zoonotic infection caused by metacestod form of Echinococcus granulosus. In this study, 91 patients with CE were revealed according to their gender, age, symptoms, cyst localization and size, and also to their preoperative serologic analysis results. These patients were recruited in Pathology Department of Inonu University, School of Medicine, Turgut Ozal Medical Center between January 2002 and December 2005. They had already undergone surgery following the preliminary diagnosis of CE and were diagnosed definitively after histopathological analysis. Thirty-seven of them were female (41%) and the rest were male (n=54, 59%). Mean age was 36.24 ± 20.35 (range: 4-80 years). Lung and liver were the most common localizations for CE cysts (n=67, 73.63%). Fifty patients' preoperative serological analysis (IHA) were already available (55%) and 46 of them (92%) were positive. Since histopathological definitive diagnosis was CE, false negative result rate was assessed as 8%. Intraabdominal CE cyst localization was found related with bigger cyst size and multiple organ involvement. Symptom duration was correlated with cyst size (p < 0.05; r < sup > 2 < /sup > =0.09). IHA results were failed to be found related with clinical features of the cysts.

Heart involvement in cystic fibrosis: A specific cystic fibrosis-related myocardial changes?

PubMed

Labombarda, Fabien; Saloux, Eric; Brouard, Jacques; Bergot, Emmanuel; Milliez, Paul

2016-09-01

Cystic fibrosis is a complex multi-systemic chronic disease characterized by progressive organ dysfunction with development of fibrosis, possibly affecting the heart. Over the last four decades pathological, experimental, and clinical evidence points towards the existence of a specific myocardial involvement in cystic fibrosis. Multi-modality cardiac imaging, especially recent echocardiographic techniques, evidenced diastolic and/or systolic ventricular dysfunction in cystic fibrosis leading to the concept of a cystic fibrosis-related cardiomyopathy. Hypoxemia and inflammation are among the most important factors for heart involvement in cystic fibrosis. Cystic Fibrosis Transmembrane Regulator was found to be involved in the regulation of cardiomyocyte contraction and may also account for cystic fibrosis-related myocardial dysfunction. This review, mainly focused on echocardiographic studies, seeks to synthesize the existing literature for and against the existence of heart involvement in cystic fibrosis, its mechanisms and prognostic implications. Careful investigation of the heart function may be helpful for risk stratification and therapeutic decisions in patients with cystic fibrosis. Copyright © 2016 Elsevier Ltd. All rights reserved.

Echinococcus equinus and Echinococcus granulosus sensu stricto from the United Kingdom: genetic diversity and haplotypic variation.

PubMed

Boufana, Belgees; Lett, Wai San; Lahmar, Samia; Buishi, Imad; Bodell, Anthony J; Varcasia, Antonio; Casulli, Adriano; Beeching, Nicholas J; Campbell, Fiona; Terlizzo, Monica; McManus, Donald P; Craig, Philip S

2015-02-01

Cystic echinococcosis is endemic in Europe including the United Kingdom. However, information on the molecular epidemiology of Echinococcus spp. from the United Kingdom is limited. Echinococcus isolates from intermediate and definitive animal hosts as well as from human cystic echinococcosis cases were analysed to determine species and genotypes within these hosts. Echinococcus equinus was identified from horse hydatid isolates, cysts retrieved from captive UK mammals and copro-DNA of foxhounds and farm dogs. Echinococcus granulosus sensu stricto (s.s.) was identified from hydatid cysts of sheep and cattle as well as in DNA extracted from farm dog and foxhound faecal samples, and from four human cystic echinococcosis isolates, including the first known molecular confirmation of E. granulosus s.s. infection in a Welsh sheep farmer. Low genetic variability for E. equinus from various hosts and from different geographical locations was detected using the mitochondrial cytochrome c oxidase subunit 1 gene (cox1), indicating the presence of a dominant haplotype (EQUK01). In contrast, greater haplotypic variation was observed for E. granulosus s.s. cox1 sequences. The haplotype network showed a star-shaped network with a centrally placed main haplotype (EgUK01) that had been reported from other world regions. Copyright © 2014 Australian Society for Parasitology Inc. Published by Elsevier Ltd. All rights reserved.

Comparisons of Serum Total IgE, IgG, and IgG1 Levels in Patients with and without Echinococcosis-Induced Anaphylactic Shock

PubMed Central

Li, Yimei; Zheng, Hong; Gu, Meilin; Cao, Xinghua; Wen, Hao; Liu, Zaoling; Liu, Tao

2012-01-01

We investigated serum total immunoglobulin E (IgE), IgG, and IgG1 levels in patients with and without echinococcosis-induced anaphylactic shock. This was a case-control study of 11 patients with echinococcosis-induced anaphylactic shock and 22 echinococcosis patients with cyst rupture but without anaphylactic shock. Blood was collected before surgery (T0), at the time of cyst rupture (T1), and shock (Tx), 1 h (T2), 1 day (T3), and 1 week (T4) after cyst rupture. Serum IgE, IgG, and IgG1 were determined by enzyme-linked immunosorbent assay. Serum IgE, IgG, and IgG1 levels were significantly higher in patients who developed anaphylactic shock at all time points. Increased pre-surgical IgG and IgG1 levels were identified to be a significant risk factors for developing anaphylactic shock. The results showed that a serum IgG concentration of 312.25 μg/mL could be used as a cut-off point to predict whether an echinococcosis patient would develop anaphylactic shock. PMID:22764299

Network for Early Onset Cystic Kidney Diseases—A Comprehensive Multidisciplinary Approach to Hereditary Cystic Kidney Diseases in Childhood

PubMed Central

König, Jens Christian; Titieni, Andrea; Konrad, Martin; Bergmann, C.

2018-01-01

Hereditary cystic kidney diseases comprise a complex group of genetic disorders representing one of the most common causes of end-stage renal failure in childhood. The main representatives are autosomal recessive polycystic kidney disease, nephronophthisis, Bardet–Biedl syndrome, and hepatocyte nuclear factor-1beta nephropathy. Within the last years, genetic efforts have brought tremendous progress for the molecular understanding of hereditary cystic kidney diseases identifying more than 70 genes. Yet, genetic heterogeneity, phenotypic variability, a lack of reliable genotype–phenotype correlations and the absence of disease-specific biomarkers remain major challenges for physicians treating children with cystic kidney diseases. To tackle these challenges comprehensive scientific approaches are urgently needed that match the ongoing “revolution” in genetics and molecular biology with an improved efficacy of clinical data collection. Network for early onset cystic kidney diseases (NEOCYST) is a multidisciplinary, multicenter collaborative combining a detailed collection of clinical data with translational scientific approaches addressing the genetic, molecular, and functional background of hereditary cystic kidney diseases. Consisting of seven work packages, including an international registry as well as a biobank, NEOCYST is not only dedicated to current scientific questions, but also provides a platform for longitudinal clinical surveillance and provides precious sources for high-quality research projects and future clinical trials. Funded by the German Federal Government, the NEOCYST collaborative started in February 2016. Here, we would like to introduce the rationale, design, and objectives of the network followed by a short overview on the current state of progress. PMID:29497606

Epidemiology and clinical features of cystic hydatidosis in Western Sicily: a ten-year review.

PubMed

Cappello, Elisa; Cacopardo, Bruno; Caltabiano, Eleonora; Li Volsi, Sigismindo; Chiara, Rosa; Sapienza, Mauro; Nigro, Luciano

2013-12-28

To assess retrospectively the epidemiological and clinical aspects of cystic echinococcosis (CE) and to evaluate follow-up and response to treatment in patients affected by CE. From January 2000 to December 2010, all patients affected by CE at the Infectious Diseases Units of the University of Catania and of Basilotta Hospital in Nicosia-Enna, were enrolled as participants in the study. Epidemiological, clinical and laboratory data were collected for each patient. Diagnosis of CE was performed using clinical imaging and laboratory parameters. Response to treatment was categorized as follows: "cure" as the disappearance or complete calcification of cyst/s; "improvement" as a reduction in the diameter and/or number of existing cysts; and "impairment" as an increase in the diameter and/or number of existing cyst/s and the onset of relapses (i.e., the onset of new cyst/s and an increase in the diameter of previously existing cyst/s and/or complications. Immunoglobulin E (IgE) titers and eosinophil percentages were evaluated at diagnosis, at six months after the initiation of treatment and again in the case of relapse. Hyper-eosinophilia was defined as an eosinophil percentage of ≥ 6%. Thirty-two patients were diagnosed with CE in our Unit during the research period, with a male-female ratio of 2:1. At the time of diagnosis, 40% of patients presented a single CE cyst. Sixty percent showed multi-organ involvement. The liver-lung localization ratio was 2:1. Patients below the age of 50 at diagnosis were more likely to have multiple cysts (73.7% vs 35.5%, P < 0.05). Regarding treatment, 30 patients were treated medically and 16 surgically. Fourteen patients were treated both medically and surgically. Relapses were seen to be less frequent in patients treated with albendazole before and after surgery. Complete cure or an improvement was achieved in 23 patients. Impairment was observed in one patient. Two patients showed no improvement. Relapses were more frequent in those

Mucinous cystic neoplasms of the mesentery: a case report and review of the literature

PubMed Central

Metaxas, Georgios; Tangalos, Athanasios; Pappa, Polyxeni; Papageorgiou, Irene

2009-01-01

Background Mucinous cystic neoplasms arise in the ovary and various extra-ovarian sites. While their pathogenesis remains conjectural, their similarities suggest a common pathway of development. There have been rare reports involving the mesentery as a primary tumour site. Case presentation A cystic mass of uncertain origin was demonstrated radiologically in a 22 year old female with chronic abdominal pain. At laparotomy, the mass was fixed within the colonic mesentery. Histology demonstrated a benign mucinous cystadenoma. Methods and results We review the literature on mucinous cystic neoplasms of the mesentery and report on the pathogenesis, biologic behavior, diagnosis and treatment of similar extra-ovarian tumors. We propose an updated classification of mesenteric cysts and cystic tumors. Conclusion Mucinous cystic neoplasms of the mesentery present almost exclusively in women and must be considered in the differential diagnosis of mesenteric tumors. Only full histological examination of a mucinous cystic neoplasm can exclude a borderline or malignant component. An updated classification of mesenteric cysts and cystic tumors is proposed. PMID:19454018

Neotropical echinococcosis caused by Echinococcus vogeli in a 6-year-old child: the second case report in humans in French Guiana.

PubMed

Debourgogne, Anne; Blanchet, Denis; Fior, Angela; Umhang, Gérald; Simon, Stéphane; Aznar, Christine

2017-02-01

Human polycystic echinococcosis is a parasitic infection caused by the larval stage of Echinococcus vogeli which occurs in rural areas of Central and South America. Abdominal echinococcosis caused by E. vogeli is reported for the first time in a child, a 6-year-old boy in French Guiana. The diagnosis was made by histological and molecular techniques. In tropical regions, this neglected disease must be considered even in children.

Suppression of E. multilocularis Hydatid Cysts after Ionizing Radiation Exposure

PubMed Central

Zhou, Rong; Zhang, Hong

2013-01-01

Background Heavy-ion therapy has an advantage over conventional radiotherapy due to its superb biological effectiveness and dose conformity in cancer therapy. It could be a potential alternate approach for hydatid cyst treatment. However, there is no information currently available on the cellular and molecular basis for heavy-ion irradiation induced cell death in cystic echinococcosis. Methododology/Principal Findings LD50 was scored by protoscolex death. Cellular and ultrastructural changes within the parasite were studied by light and electron microscopy, mitochondrial DNA (mtDNA) damage and copy number were measured by QPCR, and apoptosis was determined by caspase 3 expression and caspase 3 activity. Ionizing radiation induced sparse cytoplasm, disorganized and clumped organelles, large vacuoles and devoid of villi. The initial mtDNA damage caused by ionizing radiation increased in a dose-dependent manner. The kinetic of DNA repair was slower after carbon-ion radiation than that after X-rays radiation. High dose carbon-ion radiation caused irreversible mtDNA degradation. Cysts apoptosis was pronounced after radiation. Carbon-ion radiation was more effective to suppress hydatid cysts than X-rays. Conclusions These studies provide a framework to the evaluation of attenuation effect of heavy-ion radiation on cystic echinococcosis in vitro. Carbon-ion radiation is more effective to suppress E. multilocularis than X-rays. PMID:24205427

[Evaluation of the Cystic Eccinococcosis Cases Diagnosed in Dr. Lütfi Kırdar Kartal Education and Research Hospital Pathology Laboratory Between 2007 and 2013].

PubMed

Selek, Ayşegül; Selek, Mehmet Burak; Karadayı, Nimet

2015-06-01

The aim of the study was a retrospective evaluation of 299 cases that were histopathologically diagnosed as cystic echinococcosis (CE) in the pathology laboratory of our hospital in a seven years period. All specimens sent to the laboratory were examined microscopically following macroscopic and hematoxylin eosin (H-E) staining. 299 cases were reviewed according to age, gender and organ affected by the cyst, more than one specimen of the same organ was evaluated once. Of the 299 cases, 44.5% (133) were male whereas 55.5% (166) of them were female. Additionally, %5 (15) of the cases were between 0-15 ages, %31.8 (95) of them were between 16-30 ages, %29.4 (88) of them were between 31-45 ages, % 24.4 (73) of them were between 46-60 ages and %9.4 (28) of them were older than 61. Cysts were mostly localized in liver, lungs and peritoneal cavity, %71.9 (215), %11.4 (34) and %4.7 (14) respectively. Demographic features of our cases were mostly in line with the previous literature. Because of the presence of atypical localized cases, during pathologic evaluation of all surgical cystic specimens, elements of this parasite should be searched and evaluated carefully.

A Systematic Review of the Literature on Cystic Echinococcosis Frequency Worldwide and Its Associated Clinical Manifestations

PubMed Central

Budke, Christine M.; Carabin, Hélène; Ndimubanzi, Patrick C.; Nguyen, Hai; Rainwater, Elizabeth; Dickey, Mary; Bhattarai, Rachana; Zeziulin, Oleksandr; Qian, Men-Bao

2013-01-01

A systematic literature review of cystic echinoccocosis (CE) frequency and symptoms was conducted. Studies without denominators, original data, or using one serological test were excluded. Random-effect log-binomial models were run for CE frequency and proportion of reported symptoms where appropriate. A total of 45 and 25 articles on CE frequency and symptoms met all inclusion criteria. Prevalence of CE ranged from 1% to 7% in community-based studies and incidence rates ranged from 0 to 32 cases per 100,000 in hospital-based studies. The CE prevalence was higher in females (Prevalence Proportion Ratio: 1.35 [95% Bayesian Credible Interval: 1.16–1.53]) and increased with age. The most common manifestations of hepatic and pulmonary CE were abdominal pain (57.3% [95% confidence interval [CI]: 37.3–76.1%]) and cough (51.3% [95% CI: 35.7–66.7%]), respectively. The results are limited by the small number of unbiased studies. Nonetheless, the age/gender prevalence differences could be used to inform future models of CE burden. PMID:23546806

A systematic review of the literature on cystic echinococcosis frequency worldwide and its associated clinical manifestations.

PubMed

Budke, Christine M; Carabin, Hélène; Ndimubanzi, Patrick C; Nguyen, Hai; Rainwater, Elizabeth; Dickey, Mary; Bhattarai, Rachana; Zeziulin, Oleksandr; Qian, Men-Bao

2013-06-01

A systematic literature review of cystic echinoccocosis (CE) frequency and symptoms was conducted. Studies without denominators, original data, or using one serological test were excluded. Random-effect log-binomial models were run for CE frequency and proportion of reported symptoms where appropriate. A total of 45 and 25 articles on CE frequency and symptoms met all inclusion criteria. Prevalence of CE ranged from 1% to 7% in community-based studies and incidence rates ranged from 0 to 32 cases per 100,000 in hospital-based studies. The CE prevalence was higher in females (Prevalence Proportion Ratio: 1.35 [95% Bayesian Credible Interval: 1.16-1.53]) and increased with age. The most common manifestations of hepatic and pulmonary CE were abdominal pain (57.3% [95% confidence interval [CI]: 37.3-76.1%]) and cough (51.3% [95% CI: 35.7-66.7%]), respectively. The results are limited by the small number of unbiased studies. Nonetheless, the age/gender prevalence differences could be used to inform future models of CE burden.

Dexamethasone attenuates echinococcosis-induced allergic reactions via regulatory T cells in mice.

PubMed

Zhang, Qin; Ye, Jianrong; Zheng, Hong

2016-03-11

Cystic echinococcosis (CE), caused by infection with Echinococcus granulosus larvae, is a potentially life-threatening disease in humans. Anaphylactic shock caused by CE is very dangerous, and is highly prevalent during surgery. Dexamethasone (DEX) is used clinically before operations to prevent allergic reactions; Regulatory T cells (Treg cells) are believed to be associated with negative immune response, which play an important role in alleviating allergic reactions. However, the association of Treg cells with DEX remains unknown. In this study, C57BL/6 mice were divided into uninfected group, untreated group and DEX group which were inoculated with protoscoleces from E. granulosus and sensitized using a cyst fluid suspension to induce anaphylactic shock. In addition, the mice in DEX group were treated with 10 mg/kg DEX by intraperitoneal injection 30 min before being sensitized. It was found that 93.75 % of all sensitized mice experienced allergic symptoms. The levels of IgE, IgE/IgG, and IgE/IgG1 were significantly higher in both untreated group and DEX group. The proportion of CD4 + CD25 + FOXP3 + Treg cells relative to CD4+ Treg cells, and the levels of interleukin-10 (IL-10) and tumor growth factor-β (TGF-β1) were significantly higher in DEX group. The level of IL-13 was significantly higher in the sensitized mice than in the other groups. These cells may play a key role in alleviating the immune response in CE-induced anaphylactic shock. The protective effect of DEX may be due to Treg cell upregulating IL-10 and TGF-β1 levels, and inhibiting helper T cell 2 cytokines.

Evaluation of Oxfendazole, Praziquantel and Albendazole against Cystic Echinococcosis: A Randomized Clinical Trial in Naturally Infected Sheep

PubMed Central

Gavidia, Cesar M.; Gonzalez, Armando E.; Barron, Eduardo A.; Ninaquispe, Berenice; Llamosas, Monica; Verastegui, Manuela R.; Robinson, Colin; Gilman, Robert H.

2010-01-01

Background Cystic Echinococosis (CE) is a zoonotic disease caused by larval stage Echinococcus granulosus. We determined the effects of high dose of Oxfendazole (OXF), combination Oxfendazole/Praziquantel (PZQ), and combination Albendazole (ABZ)/Praziquantel against CE in sheep. Methodology/Principal Findings A randomized placebo-controlled trial was carried out on 118 randomly selected ewes. They were randomly assigned to one of the following groups: 1) placebo; 2) OXF 60 mg/Kg of body weight (BW) weekly for four weeks; 3) ABZ 30 mg/Kg BW + PZQ 40 mg/Kg BW weekly for 6 weeks, and 4) OXF 30 mg/Kg BW+ PZQ 40 mg/Kg BW biweekly for 3 administrations (6 weeks). Percent protoscolex (PSC) viability was evaluated using a 0.1% aqueous eosin vital stain for each cyst. “Noninfective” sheep were those that had no viable PSCs; “low-medium infective” were those that had 1% to 60% PSC viability; and “high infective” were those with more than 60% PSC viability. We evaluated 92 of the 118 sheep. ABZ/PZQ led the lowest PSC viability for lung cysts (12.7%), while OXF/PZQ did so for liver cysts (13.5%). The percentage of either “noninfective” or “low-medium infective” sheep was 90%, 93.8% and 88.9% for OXF, ABZ/PZQ and OXF/PZQ group as compared to 50% “noninfective” or “low-medium infective” for placebo. After performing all necropsies, CE prevalence in the flock of sheep was 95.7% (88/92) with a total number of 1094 cysts (12.4 cysts/animal). On average, the two-drug-combination groups resulted pulmonary cysts that were 6 mm smaller and hepatic cysts that were 4.2 mm smaller than placebo (p<0.05). Conclusions/Significance We demonstrate that Oxfendazole at 60 mg, combination Oxfendazole/Praziquantel and combination Albendazole/Praziquantel are successful schemas that can be added to control measures in animals and merits further study for the treatment of animal CE. Further investigations on different schedules of monotherapy or combined chemotherapy are

Progressive alveolar echinococcosis after discontinuation of anthelmintic therapy

PubMed Central

2013-01-01

We report a case of a female patient with alveolar echinococcosis (AE) who presented with progressive pulmonary and hepatic lesions and had a fatal outcome. AE affecting the liver, the lungs and the brain had been diagnosed 20 years ago and treated successfully with albendazole and stereotactic gamma knife therapy. Due to severe hair loss albendazole was stopped 14 years before presentation. Lesions had remained stable in imaging studies for at least 11 years, but then had started to progress. Lifelong anthelmintic maintenance therapy and regular follow-up may therefore be crucial in order to prevent such a dramatic clinical course. PMID:24289745

Cystic fibrosis.

PubMed

Elborn, J Stuart

2016-11-19

Cystic fibrosis is a common life-limiting autosomal recessive genetic disorder, with highest prevalence in Europe, North America, and Australia. The disease is caused by mutation of a gene that encodes a chloride-conducting transmembrane channel called the cystic fibrosis transmembrane conductance regulator (CFTR), which regulates anion transport and mucociliary clearance in the airways. Functional failure of CFTR results in mucus retention and chronic infection and subsequently in local airway inflammation that is harmful to the lungs. CFTR dysfunction mainly affects epithelial cells, although there is evidence of a role in immune cells. Cystic fibrosis affects several body systems, and morbidity and mortality is mostly caused by bronchiectasis, small airways obstruction, and progressive respiratory impairment. Important comorbidities caused by epithelial cell dysfunction occur in the pancreas (malabsorption), liver (biliary cirrhosis), sweat glands (heat shock), and vas deferens (infertility). The development and delivery of drugs that improve the clearance of mucus from the lungs and treat the consequent infection, in combination with correction of pancreatic insufficiency and undernutrition by multidisciplinary teams, have resulted in remarkable improvements in quality of life and clinical outcomes in patients with cystic fibrosis, with median life expectancy now older than 40 years. Innovative and transformational therapies that target the basic defect in cystic fibrosis have recently been developed and are effective in improving lung function and reducing pulmonary exacerbations. Further small molecule and gene-based therapies are being developed to restore CFTR function; these therapies promise to be disease modifying and to improve the lives of people with cystic fibrosis. Copyright © 2016 Elsevier Ltd. All rights reserved.

Longitudinal cystic fibrosis care.

PubMed

Antunovic, S S; Lukac, M; Vujovic, D

2013-01-01

Cystic fibrosis is a complex disease entity that presents considerable lifelong challenges. Implementation of medical and surgical treatment options involves multisystem interventions to prevent and treat lung and gastrointestinal manifestations of cystic fibrosis and associated comorbidities. From birth through adulthood, cystic fibrosis care entails a longitudinal regimen aimed at achieving relief of disease symptoms and enhanced life expectancy. With increased knowledge of the molecular behavior of the cystic fibrosis transmembrane conductance regulator (CFTR) in health and disease, clinical practice has been enriched by the prospect of novel strategies, including mutation-specific drug and gene therapy targeting restoration of corrupted transepithelial ion transport. Emerging paradigms of comprehensive care increasingly enable personalized solutions to address the root cause of disease-transforming management options for individuals with cystic fibrosis.

Cystic fibrosis.

PubMed

O'Sullivan, Brian P; Freedman, Steven D

2009-05-30

Cystic fibrosis is the most common lethal genetic disease in white populations. The outlook for patients with the disease has improved steadily over many years, largely as a result of earlier diagnosis, more aggressive therapy, and provision of care in specialised centres. Researchers now have a more complete understanding of the molecular-biological defect that underlies cystic fibrosis, which is leading to new approaches to treatment. One of these treatments, hypertonic saline, is already in use, whereas others are in advanced stages of development. We review clinical care for cystic fibrosis and discuss recent advances in the understanding of its pathogenesis, implementation of screening of neonates, and development of therapies aimed at treating the basic defect.

Fine-needle aspiration study of cystic papillary thyroid carcinoma: Rare cytological findings

PubMed Central

Mokhtari, Maral; Kumar, Perikala Vijayananda; Hayati, Kamran

2016-01-01

Background: Cystic papillary thyroid carcinoma (CPTC) is a variant of papillary carcinoma that has many mimickers in cytological grounds. Aim: To study the cytomorphologic features of CPTC and compare them to those of other cystic thyroid lesions using fine-needle aspiration cytology (FNAC). We also aimed to identify the cytomorphologic features that distinguish CPTC from other cystic thyroid lesions. Materials and Methods: Seventy-three cases of CPTC were included in the study. The cytomorphologic features of these cases were analyzed. The FNA smears of other thyroid lesions with cystic changes (300 colloid goiters, 290 adenomatoid nodules, 11 follicular neoplasms, and 9 hurtle cell neoplasm) were also studied. Results: The smears in CPTC revealed isolated follicular cells, small groups of cells with scalloped margins, cell swirls, small clusters with a cartwheel pattern, papillary clusters, intranuclear inclusions, nuclear grooves, sticky colloid, intracellular colloids, psammoma bodies, multinucleated giant cells, and foamy and hemosiderin laden macrophages. Small groups of cells with scalloped borders, cellular swirls, and small clusters with a cartwheel pattern were seen in CPTC, but not in other cystic lesions. Interestingly, mesothelial-like cells and hemophagocytic cells were seen in five and three cases of CPTC, respectively, but not in other cystic lesions. Conclusion: Mesothelial-like cells and hemophagocytic cells were observed in five and three cases of CPTC, respectively. Similar finding have not been previously reported in the literature. PMID:27756982

Boiling sheep liver or lung for 30 minutes is necessary and sufficient to kill Echinococcus granulosus protoscoleces in hydatid cysts

PubMed Central

Li, Jun; Wu, Chuanchuan; Wang, Hui; Liu, Huanyuan; Vuitton, Dominique A.; Wen, Hao; Zhang, Wenbao

2014-01-01

Proper disposal of carcasses and offal after home slaughter is difficult in poor and remote communities and therefore dogs readily have access to hydatid cysts containing offal from livestock, thus completing the parasite cycle of Echinococcus granulosus and putting communities at risk of cystic echinococcosis. Boiling livers and lungs which contain hydatid cysts could be a simple, efficient and energy- and time-saving way to kill the infectious protoscoleces. The aim of this study was to provide precise practical recommendations to livestock owners. Our results show that boiling the whole sheep liver and/or lung, with single or multiple hydatid cysts, for 30 min is necessary and sufficient to kill E. granulosus protoscoleces in hydatid cysts. Advertising on this simple rule in at-risk communities would be an efficient and cheap complement to other veterinary public health operations to control cystic echinococcosis. PMID:25456565

Compare the Difference of B-cell Epitopes of EgAgB1 and EgAgB3 Proteins Selected through Bioinformatic Analysis

NASA Astrophysics Data System (ADS)

An, Mengting; Zhang, Fengbo; Zhu, Yuejie; Zhao, Xiao; Ding, Jianbing

2018-01-01

Cystic echinococcosis, as a zoonosis, seriously endangers humans and animals, so early diagnosis of this disease is particularly important. Therefore, this study is to predict B-cell epitopes of EgAgB1 and EgAgB3 proteins by bioinformatics software. B-cell epitopes of EgAgB1 and EgAgB3 proteins are predicted using DNAStar and IEDB software. The results suggest that there are two potential B-cell epitopes in EgAgB1, which located in the 8-15 and 31-37 amino acid residue segments. And two potential B-cell epitopes in EgAgB2, located in the 20∼27 and 47∼53 amino acid residue segments. This study predicted the B-cell epitopes of EgAgB1 and EgAgB3 proteins, which laid the foundation for the early diagnosis of Cystic echinococcosis.

Molecular Characterization of Echinococcus granulosus Cysts in North Indian Patients: Identification of G1, G3, G5 and G6 Genotypes

PubMed Central

Sharma, Monika; Sehgal, Rakesh; Fomda, Bashir Ahmad; Malhotra, Anil; Malla, Nancy

2013-01-01

Background Cystic echinococcosis (CE) caused by the Echinococcus granulosus, is a major public health problem worldwide, including India. The different genotypes of E. granulosus responsible for human hydatidosis have been reported from endemic areas throughout the world. However, the genetic characterization of E. granulosus infecting the human population in India is lacking. The aim of study was to ascertain the genotype(s) of the parasite responsible for human hydatidosis in North India. Methodology/Principal Findings To study the transmission patterns of E. granulosus, genotypic analysis was performed on hydatid cysts obtained from 32 cystic echinococcosis (CE) patients residing in 7 different states of North India. Mitochondrial cytochrome c oxidase subunit1 (cox1) sequencing was done for molecular identification of the isolates. Most of the CE patients (30/32) were found to be infected with hydatid cyst of either G3 (53.1%) or G1 (40.62%) genotype and one each of G5 (cattle strain) and G6 (camel strain) genotype. Conclusions/Significance These findings demonstrate the zoonotic potential of G1 (sheep strain) and G3 (buffalo strain) genotypes of E. granulosus as these emerged as predominant genotypes infecting the humans in India. In addition to this, the present study reports the first human CE case infected with G5 genotype (cattle strain) in an Asian country and presence of G6 genotype (camel strain) in India. The results may have important implications in the planning of control strategies for human hydatidosis. PMID:23785531

Phylogenetic systematics of the genus Echinococcus (Cestoda: Taeniidae).

PubMed

Nakao, Minoru; Lavikainen, Antti; Yanagida, Tetsuya; Ito, Akira

2013-11-01

Echinococcosis is a serious helminthic zoonosis in humans, livestock and wildlife. The pathogenic organisms are members of the genus Echinococcus (Cestoda: Taeniidae). Life cycles of Echinococcus spp. are consistently dependent on predator-prey association between two obligate mammalian hosts. Carnivores (canids and felids) serve as definitive hosts for adult tapeworms and their herbivore prey (ungulates, rodents and lagomorphs) as intermediate hosts for metacestode larvae. Humans are involved as an accidental host for metacestode infections. The metacestodes develop in various internal organs, particularly in liver and lungs. Each metacestode of Echinococcus spp. has an organotropism and a characteristic form known as an unilocular (cystic), alveolar or polycystic hydatid. Recent molecular phylogenetic studies have demonstrated that the type species, Echinococcus granulosus, causing cystic echinococcosis is a cryptic species complex. Therefore, the orthodox taxonomy of Echinococcus established from morphological criteria has been revised from the standpoint of phylogenetic systematics. Nine valid species including newly resurrected taxa are recognised as a result of the revision. This review summarises the recent advances in the phylogenetic systematics of Echinococcus, together with the historical backgrounds and molecular epidemiological aspects of each species. A new phylogenetic tree inferred from the mitochondrial genomes of all valid Echinococcus spp. is also presented. The taxonomic nomenclature for Echinococcus oligarthrus is shown to be incorrect and this name should be replaced with Echinococcus oligarthra. Copyright © 2013 Australian Society for Parasitology Inc. Published by Elsevier Ltd. All rights reserved.

A new data management system for the French National Registry of human alveolar echinococcosis cases.

PubMed

Charbonnier, Amandine; Knapp, Jenny; Demonmerot, Florent; Bresson-Hadni, Solange; Raoul, Francis; Grenouillet, Frédéric; Millon, Laurence; Vuitton, Dominique Angèle; Damy, Sylvie

2014-01-01

Alveolar echinococcosis (AE) is an endemic zoonosis in France due to the cestode Echinococcus multilocularis. The French National Reference Centre for Alveolar Echinococcosis (CNR-EA), connected to the FrancEchino network, is responsible for recording all AE cases diagnosed in France. Administrative, epidemiological and medical information on the French AE cases may currently be considered exhaustive only on the diagnosis time. To constitute a reference data set, an information system (IS) was developed thanks to a relational database management system (MySQL language). The current data set will evolve towards a dynamic surveillance system, including follow-up data (e.g. imaging, serology) and will be connected to environmental and parasitological data relative to E. multilocularis to better understand the pathogen transmission pathway. A particularly important goal is the possible interoperability of the IS with similar European and other databases abroad; this new IS could play a supporting role in the creation of new AE registries. © A. Charbonnier et al., published by EDP Sciences, 2014.

A new data management system for the French National Registry of human alveolar echinococcosis cases

PubMed Central

Charbonnier, Amandine; Knapp, Jenny; Demonmerot, Florent; Bresson-Hadni, Solange; Raoul, Francis; Grenouillet, Frédéric; Millon, Laurence; Vuitton, Dominique Angèle; Damy, Sylvie

2014-01-01

Alveolar echinococcosis (AE) is an endemic zoonosis in France due to the cestode Echinococcus multilocularis. The French National Reference Centre for Alveolar Echinococcosis (CNR-EA), connected to the FrancEchino network, is responsible for recording all AE cases diagnosed in France. Administrative, epidemiological and medical information on the French AE cases may currently be considered exhaustive only on the diagnosis time. To constitute a reference data set, an information system (IS) was developed thanks to a relational database management system (MySQL language). The current data set will evolve towards a dynamic surveillance system, including follow-up data (e.g. imaging, serology) and will be connected to environmental and parasitological data relative to E. multilocularis to better understand the pathogen transmission pathway. A particularly important goal is the possible interoperability of the IS with similar European and other databases abroad; this new IS could play a supporting role in the creation of new AE registries. PMID:25526544

Echinococcosis in wild carnivorous species: epidemiology, genotypic diversity, and implications for veterinary public health.

PubMed

Carmena, David; Cardona, Guillermo A

2014-05-28

Echinococcosis is a zoonosis caused by helminths of the genus Echinococcus. The infection, one of the 17 neglected tropical diseases listed by the World Health Organization, has a cosmopolitan distribution and can be transmitted through a variety of domestic, synanthropic, and sylvatic cycles. Wildlife has been increasingly regarded as a relevant source of infection to humans, as demonstrated by the fact that a significant proportion of human emerging infectious diseases have a wildlife origin. Based on available epidemiological and molecular evidence, of the nine Echinococcus species currently recognized as valid taxa, E. canadensis G8-G10, E. felidis, E. multilocularis, E. oligarthrus, E. shiquicus, and E. vogeli are primarily transmitted in the wild. E. canadensis G6-G7, E. equinus, E. granulosus s.s., and E. ortleppi are considered to be transmitted mainly through domestic cycles. We summarize here current knowledge on the global epidemiology, geographical distribution and genotype frequency of Echinococcus spp. in wild carnivorous species. Topics addressed include the significance of the wildlife/livestock/human interface, the sympatric occurrence of different Echinococcus species in a given epidemiological scenario, and the role of wildlife as natural reservoir of disease to human and domestic animal populations. We have also discussed the impact that human activity and intervention may cause in the transmission dynamics of echinococcosis, including the human population expansion an encroachment on shrinking natural habitats, the increasing urbanization of wildlife carnivorous species and the related establishment of synanthropic cycles of Echinococcus spp., the land use (e.g. deforestation and agricultural practices), and the unsupervised international trade and translocation of wildlife animals. Following the 'One Health' approach, we have also emphasized that successful veterinary public health interventions in the field of echinococcosis requires an

Molecular Diagnosis of Polycystic Echinococcosis Due to Echinococcus vogeli in a Paraguayan Immigrant in Argentina

PubMed Central

Frider, B.; Alvarez Rodriguez, J.; Amante, M.; Pestalardo, M. L.; Cazorla, A.; Bresson-Hadni, S.; Millon, L.

2013-01-01

Polycystic echinococcosis due to Echinococcus vogeli is a rare parasitic infection that occurs in rural areas of Central and South America. Only molecular identification performed on formalin-fixed paraffin-embedded liver tissue samples gave an unequivocal diagnosis of this disease in a Paraguayan immigrant in Argentina. PMID:23824768

Prevalence of zoonotic intestinal parasites in domestic and stray dogs in a rural area of Iran.

PubMed

Beiromvand, Molouk; Akhlaghi, Lame; Fattahi Massom, Seyed Hossein; Meamar, Ahmad Reza; Motevalian, Abbas; Oormazdi, Hormozd; Razmjou, Elham

2013-04-01

Certain zoonotic parasites are enteropathogens in dogs that cause serious human disease such as cystic echinococcosis, human alveolar echinococcosis, visceral larva migrans, and ocular larva migrans. This study investigated the prevalence of intestinal parasites in dogs in the Chenaran County, Razavi Khorasan Province, Iran. Sampling was carried out randomly in 17 villages from November 2009 to January 2010. Seventy-seven fecal samples from 28 domestic and 49 stray dogs were examined using sieving/flotation and modified Ziehl-Neelsen staining. Intestinal parasites were found in 51 of the 77 (66%) dogs most common being Toxascaris leonina (29%, 22/77), Toxocara spp. (25%, 19/77), Eimeria spp. (19%, 15/77), Taenia/Echinococcus spp. (18%, 14/77), Sarcocystis spp. (17%, 13/77), and Dicrocoelium dendriticum (14%, 11/77). Lower infection rates of parasites were observed for Trichuris vulpis (6%, 5/77), Cryptosporidium spp. (5%, 4/77), and Physaloptera spp. (3%, 2/77). Prevalence of infection by Dipylidium caninum, Capillaria spp., Cystoisospora spp., and hookworms was similar (1%, 1/77). This study is the first report of the prevalence of intestinal parasites of domestic and stray dogs in Chenaran County, Northeast Iran. The higher prevalence of zoonotic intestinal parasites such as Toxascaris leonina, Toxocara spp. and Taenia/Echinococcus spp. compared to other parasites indicates the need for control programs to minimize the risk of transmission of zoonotic disease, particularly cystic echinococcosis, alveolar echinococcosis, visceral larva migrans, and ocular larva migrans to people living in these areas. Copyright © 2012 Elsevier B.V. All rights reserved.

Cystic lesions of the pancreas

PubMed Central

Karoumpalis, Ioannis; Christodoulou, Dimitrios K.

2016-01-01

Different types of benign or malignant cystic lesions can be observed in the pancreas. Pancreatic cystic lesions are classified under pathology terms into simple retention cysts, pseudocysts and cystic neoplasms. Mucinous cystic neoplasm is a frequent type of cystic neoplasm and has a malignant potential. Serous cystadenoma follows in frequency and is usually benign. Intraductal papillary mucinous neoplasms are the most commonly resected cystic pancreatic neoplasms characterized by dilated segments of the main pancreatic duct and/or side branches, the wall of which is covered by mucus secreting cells. These neoplasms can occupy the pancreatic head or any part of the organ. Solid pseudopapillary tumor is rare, has a low tendency for malignancy, and is usually located in the pancreatic body or tail. Endoscopic ultrasound with the use of fine-needle aspiration and cytology permits discrimination of those lesions. In this review, the main characteristics of those lesions are presented, as well as recommendations regarding their follow up and management according to recent guidelines. PMID:27065727

Epidural Cystic Spinal Meningioma

PubMed Central

Zhang, Ji; Chen, Zheng-he; Wang, Zi-feng; Sun, Peng; Jin, Jie-tian; Zhang, Xiang-heng; Zhao, Yi-ying; Wang, Jian; Mou, Yong-gao; Chen, Zhong-ping

2016-01-01

Abstract Cystic spinal meningioma (CSM) is an uncommon meningioma variant. Extradural CSMs are particularly rare and difficult to distinguish from other intraaxial tumors. This study presents a case of a 36-year-old woman with intraspinal extradual CSM at the thoracolumbar spine. She experienced persistent weakness, progressive numbness, and sensory disturbance in the right lower limb. Magnetic resonance imaging (MRI) of the patient revealed an irregular cystic mass at the thoracic 11 to lumbar 3 levels dorsally. This case was misdiagnosed as other neoplasms prior to surgery because of the atypical radiographic features and location of the tumor. Extradural CSMs should be considered in the differential diagnosis of intraspinal extradural cystic neoplasms. Complete removal of cystic wall provides an optimal outcome, rendering the lesion curable. PMID:26986119

Boiling sheep liver or lung for 30 minutes is necessary and sufficient to kill Echinococcus granulosus protoscoleces in hydatid cysts.

PubMed

Li, Jun; Wu, Chuanchuan; Wang, Hui; Liu, Huanyuan; Vuitton, Dominique A; Wen, Hao; Zhang, Wenbao

2014-01-01

Proper disposal of carcasses and offal after home slaughter is difficult in poor and remote communities and therefore dogs readily have access to hydatid cysts containing offal from livestock, thus completing the parasite cycle of Echinococcus granulosus and putting communities at risk of cystic echinococcosis. Boiling livers and lungs which contain hydatid cysts could be a simple, efficient and energy- and time-saving way to kill the infectious protoscoleces. The aim of this study was to provide precise practical recommendations to livestock owners. Our results show that boiling the whole sheep liver and/or lung, with single or multiple hydatid cysts, for 30 min is necessary and sufficient to kill E. granulosus protoscoleces in hydatid cysts. Advertising on this simple rule in at-risk communities would be an efficient and cheap complement to other veterinary public health operations to control cystic echinococcosis. © J. Li et al., published by EDP Sciences, 2014.

In silico cloning and B/T cell epitope prediction of triosephosphate isomerase from Echinococcus granulosus.

PubMed

Wang, Fen; Ye, Bin

2016-10-01

Cystic echinococcosis is a worldwide zoonosis caused by Echinococcus granulosus. Because the methods of diagnosis and treatment for cystic echinococcosis were limited, it is still necessary to screen target proteins for the development of new anti-hydatidosis vaccine. In this study, the triosephosphate isomerase gene of E. granulosus was in silico cloned. The B cell and T cell epitopes were predicted by bioinformatics methods. The cDNA sequence of EgTIM was composition of 1094 base pairs, with an open reading frame of 753 base pairs. The deduced amino acid sequences were composed of 250 amino acids. Five cross-reactive epitopes, locating on 21aa-35aa, 43aa-57aa, 94aa-107aa, 115-129aa, and 164aa-183aa, could be expected to serve as candidate epitopes in the development of vaccine against E. granulosus. These results could provide bases for gene cloning, recombinant expression, and the designation of anti-hydatidosis vaccine.

Cystic Fibrosis and Pregnancy

MedlinePlus

... Global Map Premature Birth Report Cards Careers Archives Pregnancy Before or between pregnancies Nutrition, weight & fitness Prenatal ... complications > Cystic fibrosis and pregnancy Cystic fibrosis and pregnancy E-mail to a friend Please fill in ...

Pediatric Cystic Nephroma Is Morphologically, Immunohistochemically, and Genetically Distinct From Adult Cystic Nephroma.

PubMed

Li, Yunjie; Pawel, Bruce R; Hill, Dana A; Epstein, Jonathan I; Argani, Pedram

2017-04-01

The term cystic nephroma has traditionally been used to refer to 2 neoplasms, a lesion in adults that is now thought to be part of the spectrum of mixed epithelial stromal tumor (MEST) and a pediatric lesion that has been associated with mutations in the DICER1 gene. A direct detailed morphologic, immunohistochemical, and genetic comparison of these 2 lesions has not been performed. In this study, we compare the morphologic features, immunoreactivity for estrogen receptor and inhibin, and DICER1 genetic status of 12 adult cystic nephroma/MEST (median age 50.5 y, all females) and 7 pediatric cystic nephroma (median age 1.3 y, male:female=6:1). Both lesions (11 of 12 adult cases, 6 of 7 pediatric cases) frequently demonstrated subepithelial accentuation of stromal cellularity, though the increased cellularity frequently included inflammatory cells in the pediatric cases. All adult and pediatric cases labeled for estrogen receptor; however, whereas most (83%) of adult cases labeled for inhibin at least focally, no pediatric case labeled for inhibin. Most adult cases (58%) demonstrated wavy, ropy collagen in association with cellular stroma, whereas this was not found in pediatric cases. 86% of pediatric cases demonstrated DICER1 mutations, whereas only 1 of 10 adult cases demonstrated a DICER1 mutation. In summary, although cellular stroma and estrogen receptor immunoreactivity are commonly present in both adult and pediatric cystic nephroma, ropy collagen and inhibin immunoreactivity are far more common in adult cystic nephroma/MEST, whereas DICER1 mutations are far more prevalent in pediatric cystic nephroma. These results support the current World Health Organization Classification's separation of adult and pediatric cystic nephromas as distinct entities.

Investigating the genetic diversity of Echinococcus granulosus sensu stricto with new microsatellites.

PubMed

Umhang, Gérald; Grenouillet, Frédéric; Bastid, Vanessa; M'Rad, Selim; Valot, Benoît; Oudni-M'Rad, Myriam; Babba, Hamouda; Boué, Franck

2018-06-18

Cystic echinococcosis is a zoonotic disease with worldwide distribution caused by the larval stage of the Cestode parasite Echinococcus granulosus sensu lato. Due to the predominance or even the exclusive presence of E. granulosus sensu stricto (s.s.) among E. granulosus species in many areas, the genetic diversity needs to be further investigated at the species level to better understand the inter- and intra-focus epidemiological features. Short sequences of mitochondrial or nuclear genes generally lack or have limited discriminatory power, hindering the detection of polymorphisms to reflect geographically based peculiarities and/or any history of infection. A high discriminatory power can only be reached by sequencing complete or near complete mitogenomes or relatively long nuclear sequences, which is time-consuming and onerous. To overcome this issue, a systematic research for single-locus microsatellites was performed on the nuclear genome of E. granulosus s.s. in order to investigate its intra-species genetic diversity. Two microsatellites, EgSca6 and EgSca11, were selected and characterized. The test of a panel of 75 cystic echinococcosis samples revealed a very high discrimination index of 0.824 for EgSca6, 0.987 for EgSca11, and 0.994 when multiplexing both microsatellites. Testing cystic echinococcosis samples from both liver and lungs in five sheep revealed that these two microsatellites appear to be of particular interest for investigating genetic diversity at the intra-individual host level. As this method has many advantages compared to classical sequencing, the availability of other targets means that it is potentially possible to constitute a panel facilitating large-scale molecular epidemiology studies for E. granulosus s.l.

Review of "Echinococcus and Echinococcosis, Part A." edited by R. C. Andrew Thompson, Alan J. Lymbery and Peter Deplazes.

PubMed

Ito, Akira

2017-09-04

Thompson RCA, Deplazes P, Lymbery AJ, Editors. Echinococcus and Echinococcosis, Part A. Volume 95, Advances in Parasitology 95. Academic Press; 2017. 525 pages, ISBN 978-0-12-8114711 (hardcover); 9780128114728 (eBook).

Cystic Echinococcoses in Mongolia: Molecular Identification, Serology and Risk Factors

PubMed Central

Ito, Akira; Dorjsuren, Temuulen; Davaasuren, Anu; Yanagida, Tetsuya; Sako, Yasuhito; Nakaya, Kazuhiro; Nakao, Minoru; Bat-Ochir, Oyun-Erdene; Ayushkhuu, Tsendjav; Gonchigsengee, Nyamkhuu; Li, Tiaoying; Agvaandaram, Gurbadam; Davaajav, Abmed; Boldbaatar, Chinchuluun; Chuluunbaatar, Gantigmaa

2014-01-01

Background Cystic echinococcosis (CE) is a globally distributed cestode zoonosis that causes hepatic cysts. Although Echinococcus granulosus sensu stricto (s.s.) is the major causative agent of CE worldwide, recent molecular epidemiological studies have revealed that E. canadensis is common in countries where camels are present. One such country is Mongolia. Methodology/Principal Findings Forty-three human hepatic CE cases that were confirmed histopathologically at the National Center of Pathology (NCP) in Ulaanbaatar (UB) were identified by analysis of mitochondrial cox 1 gene as being caused by either E. canadensis (n = 31, 72.1%) or E. granulosus s.s. (n = 12, 27.9%). The majority of the E. canadensis cases were strain G6/7 (29/31, 93.5%). Twenty three haplotypes were identified. Sixteen of 39 CE cases with data on age, sex and province of residence were citizens of UB (41.0%), with 13 of the 16 cases from UB caused by E. canadensis (G6/7) (81.3%). Among these 13 cases, nine were children (69.2%). All pediatric cases (n  =  18) were due to E. canadensis with 17 of the 18 cases (94.4%) due to strain G6/7. Serum samples were available for 31 of the 43 CE cases, with 22 (71.0%) samples positive by ELISA to recombinant Antigen B8/1 (rAgB). Nine of 10 CE cases caused by E. granulosus s.s. (90.0%) and 13 of 20 CE cases by E. canadensis (G6/7) (65.0%) were seropositive. The one CE case caused by E. canadensis (G10) was seronegative. CE cases caused by E. granulosus s.s. showed higher absorbance values (median value 1.131) than those caused by E. canadensis (G6/7) (median value 0.106) (p  =  0.0137). Conclusion/Significance The main species/strains in the study population were E. canadenis and E. granulossus s.s. with E. canadensis the predominant species identified in children. The reason why E. canadensis appears to be so common in children is unknown. PMID:24945801

Confirmation of Echinococcus canadensis G8 and G10 in Idaho Gray Wolves ( Canis lupus) and Cervids.

PubMed

Cerda, Jacey Roche; Ballweber, Lora Rickard

2018-04-01

We confirm the presence of Echinococcus canadensis genotypes G8 and G10 in gray wolves ( Canis lupus) and cervids in Idaho, US. Our results demonstrated that cystic echinococcosis remains a potential public health issue, indicating the need for regular deworming of domestic dogs, who often act as potential bridge hosts.

Molecular Diagnosis of Cystic Fibrosis.

PubMed

Deignan, Joshua L; Grody, Wayne W

2016-01-01

This unit describes a recommended approach to identifying causal genetic variants in an individual suspected of having cystic fibrosis. An introduction to the genetics and clinical presentation of cystic fibrosis is initially presented, followed by a description of the two main strategies used in the molecular diagnosis of cystic fibrosis: (1) an initial targeted variant panel used to detect only the most common cystic fibrosis-causing variants in the CFTR gene, and (2) sequencing of the entire coding region of the CFTR gene to detect additional rare causal CFTR variants. Finally, the unit concludes with a discussion regarding the analytic and clinical validity of these approaches. Copyright © 2016 John Wiley & Sons, Inc.

Cystic neoplasms of the exocrine pancreas.

PubMed

Campbell, F; Azadeh, B

2008-04-01

The increasing use of radiological imaging has led to greater detection of small and asymptomatic cystic lesions of the pancreas. Most are resectable, but not all are neoplastic. This review provides an update on the histopathology, immunohistochemistry, molecular biology, pathogenesis and management of cystic neoplasms of the exocrine pancreas. These include the serous, the mucinous cystic, the intraductal papillary mucinous and the solid pseudopapillary neoplasms. Recently reported variants are described and very rare cystic variants of other pancreatic epithelial and mesenchymal neoplasms are briefly mentioned.

Justified Concern or Exaggerated Fear: The Risk of Anaphylaxis in Percutaneous Treatment of Cystic Echinococcosis—A Systematic Literature Review

PubMed Central

Neumayr, Andreas; Troia, Giuliana; de Bernardis, Chiara; Tamarozzi, Francesca; Goblirsch, Sam; Piccoli, Luca; Hatz, Christoph; Filice, Carlo; Brunetti, Enrico

2011-01-01

Percutaneous treatment (PT) emerged in the mid-1980s as an alternative to surgery for selected cases of abdominal cystic echinococcosis (CE). Despite its efficacy and widespread use, the puncture of echinococcal cysts is still far from being universally accepted. One of the main reasons for this reluctance is the perceived risk of anaphylaxis linked to PTs. To quantify the risk of anaphylactic reactions and lethal anaphylaxis with PT, we systematically searched MEDLINE for publications on PT of CE and reviewed the PT-related complications. After including 124 publications published between 1980 and 2010, we collected a total number of 5943 PT procedures on 5517 hepatic and non-hepatic echinococcal cysts. Overall, two cases of lethal anaphylaxis and 99 reversible anaphylactic reactions were reported. Lethal anaphylaxis occurred in 0.03% of PT procedures, corresponding to 0.04% of treated cysts, while reversible allergic reactions complicated 1.7% of PTs, corresponding to 1.8% of treated echinococcal cysts. Analysis of the literature shows that lethal anaphylaxis related to percutaneous treatment of CE is an extremely rare event and is observed no more frequently than drug-related anaphylactic side effects. PMID:21695106

Cost of care of patients with cystic fibrosis in The Netherlands in 1990-1.

PubMed Central

Wildhagen, M. F.; Verheij, J. B.; Verzijl, J. G.; Hilderink, H. B.; Kooij, L.; Tijmstra, T.; ten Kate, L. P.; Gerritsen, J.; Bakker, W.; Habbema, J. D.; Habbema, F.

1996-01-01

BACKGROUND: Research on the cost of care of patients with cystic fibrosis is scarce. The aim of this study was to estimate the costs using age-specific medical consumption from real patient data. METHODS: The age-specific medical consumption of patients with cystic fibrosis in The Netherlands in 1991 was estimated from a survey of medical records and a patient questionnaire. A distinction was made between costs of hospital care, hospital and non-hospital medication, and home care. Costs per year were obtained by multiplying the yearly amount of care and the costs per unit. RESULTS: On average the annual cost of a patient with cystic fibrosis in 1991 was 10,908 pounds (hospital care 42%, medication 37%, home care 20%). The cost of care of cystic fibrosis in The Netherlands, with approximately 1000 patients, is estimated at 10.9 million pounds per year, which is 0.07% of the total health care budget. The cost of care of a patient up to the age of 35 is estimated at 614,587 pounds. When year-to-year survival is taken into account and future costs are discounted to the year of birth with a yearly discount rate of 5%, the cost of care of a patient with cystic fibrosis is estimated at 164,365 pounds for 1991. This estimate will be used in a prospective evaluation of screening for cystic fibrosis carriers. CONCLUSIONS: The cost of care of patients with cystic fibrosis estimated by age-specific medical consumption of real patients is higher than that estimated by non-age-specific medical consumption and/or expert opinions. PMID:8779135

Voice Disorder in Cystic Fibrosis Patients

PubMed Central

Lourenço, Bruna Mendes; Costa, Kauê Machado; da Silva Filho, Manoel

2014-01-01

Cystic fibrosis is a common autosomal recessive disorder with drastic respiratory symptoms, including shortness of breath and chronic cough. While most of cystic fibrosis treatment is dedicated to mitigating the effects of respiratory dysfunction, the potential effects of this disease on vocal parameters have not been systematically studied. We hypothesized that cystic fibrosis patients, given their characteristic respiratory disorders, would also present dysphonic symptoms. Given that voice disorders can severely impair quality of life, the identification of a potential cystic fibrosis-related dysphonia could be of great value for the clinical evaluation and treatment of this disease. We tested our hypothesis by measuring vocal parameters, using both objective physical measures and the GRBAS subjective evaluation method, in male and female cystic fibrosis patients undergoing conventional treatment and compared them to age and sex matched controls. We found that cystic fibrosis patients had a significantly lower vocal intensity and harmonic to noise ratio, as well as increased levels of jitter and shimmer. In addition, cystic fibrosis patients also showed higher scores of roughness, breathiness and asthenia, as well as a significantly altered general grade of dysphonia. When we segregated the results according to sex, we observed that, as a group, only female cystic fibrosis patients had significantly lower values of harmonic to noise ratio and an abnormal general grade of dysphonia in relation to matched controls, suggesting that cystic fibrosis exerts a more pronounced effect on vocal parameters of women in relation to men. Overall, the dysphonic characteristics of CF patients can be explained by dysfunctions in vocal fold movement and partial upper airway obstruction, potentially caused by the accumulation of mucus and chronic cough characteristic of CF symptomatology. Our results show that CF patients exhibit significant dysphonia and suggest they may

Vitamin A supplementation for cystic fibrosis.

PubMed

Bonifant, Catherine M; Shevill, Elizabeth; Chang, Anne B

2014-05-14

People with cystic fibrosis and pancreatic insufficiency are at risk of fat soluble vitamin deficiency as these vitamins (A, D, E and K) are co-absorbed with fat. Thus, some cystic fibrosis centres routinely administer these vitamins as supplements but the centres vary in their approach of addressing the possible development of deficiencies in these vitamins. Vitamin A deficiency causes predominantly eye and skin problems while supplementation of vitamin A to excessive levels may cause harm to the respiratory and skeletal systems in children. Thus a systematic review on vitamin A supplementation in people with cystic fibrosis would help guide clinical practice. To determine if vitamin A supplementation in children and adults with cystic fibrosis:1. reduces the frequency of vitamin A deficiency disorders;2. improves general and respiratory health;3. increases the frequency of vitamin A toxicity. We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group Trials Register which comprises of references identified from comprehensive electronic database searches and handsearches of relevant journals and abstract books of conference proceedings.Date of the most recent search of the Group's Cystic Fibrosis Trials Register: 07 April 2014. All randomised or quasi-randomised controlled trials comparing all preparations of oral vitamin A used as a supplement compared to either no supplementation (or placebo) at any dose and for any duration, in children or adults with cystic fibrosis (defined by sweat tests or genetic testing) with and without pancreatic insufficiency. No relevant studies for inclusion were identified in the search. No studies were included in this review. As there were no randomised or quasi-randomised controlled trials identified, we cannot draw any conclusions on the benefits (or otherwise) of regular administration of vitamin A in people with cystic fibrosis. Until further data are available, country or region specific guidelines on the use of

Hepatic alveolar echinococcosis: correlative US and CT study

DOE Office of Scientific and Technical Information (OSTI.GOV)

Didier, D.; Weiler, S.; Rohmer, P.

1985-01-01

A total of 24 cases of hepatic alveolar echinococcosis (HAE) due to Echinococcus multilocularis was assessed by US and CT. The diagnosis was confirmed in all cases by immunologic and histologic study. Both US and CT patterns of HAE showed changes of liver morphology in both contour and size. Abnormal areas of parenchyma were nodular or in fields, irregular, heterogeneous, and basically echogenic. Clustered microcalcifications were encountered within the abnormal parenchymal fields in 50% of cases, and necrotized zones occurred in 40% of cases. Dilatation of intrahepatic bile ducts was commonly seen, especially on US; hilar involvement was frequent. Follow-upmore » by both techniques can display increases of primary lesions, occurrence of new foci, and local or regional extensions. Precise evaluations of the lesions arising from correlative use of US and CT permits adequate therapeutic management.« less

‘Teeth in the brain’ – a case of giant intracranial mature cystic teratoma

PubMed Central

O’Grady, John; Kobayter, Lina; Kaliaperumal, Chandrasekaran; O’Sullivan, Michael

2012-01-01

The authors describe a case of a giant intracranial mature cystic teratoma in a 16-year-old girl presenting acutely with a severe headache, vomiting and a complex generalised seizure with a background history of intermittent headaches for 3 years. CT and MRI brain demonstrated a ruptured large cystic teratoma encapsulating two large teeth within the diffusely dense fatty heterogeneous lesion. Surgical debulking of the cyst was performed and the calcific remnants were left behind owing to dense adhesion to the brain. The procedure was complicated by postoperative hydrocephalus and needed a ventricloperitoneal shunt. She is currently asymptomatic and undergoing rehabilitation. PMID:22707688

Associations between Academic Achievement and Psychosocial Variables in Adolescents with Cystic Fibrosis

ERIC Educational Resources Information Center

Grieve, Adam J.; Tluczek, Audrey; Racine-Gilles, Caroline N.; Laxova, Anita; Albers, Craig A.; Farrell, Philip M.

2011-01-01

Background: Cystic fibrosis (CF) is a chronic genetic disease that leads to the accumulation of thick mucus in multiple organ systems, leading to chronic lung infection and affecting the body's ability to absorb nutrients necessary for growth and development. This cross-sectional, correlational study examined the potential effects of CF on…

Pancreatic cystic tumors.

PubMed

Salvia, R; Festa, L; Butturini, G; Tonsi, A; Sartori, N; Biasutti, C; Capelli, P; Pederzoli, P

2004-04-01

Cystic tumors of the pancreas are less frequent than other tumors in neoplastic pancreatic pathology, but in recent years the literature has reported an increasing number. After the first report by Becourt in 1830, cystic tumors were classified into 2 different types by Compagno and Oertel in 1978: benign tumors with glycogen-rich cells and mucinous cystic neoplasms with overt and latent malignancy. The WHO classification of exocrine tumors of the pancreas, published in 1996, is based on the histopathological features of the epithelial wall, which are the main factor in differential diagnosis with cystic lesions of the pancreas. Thanks to the knowledge acquired up to now, a surgical procedure is not always required because the therapeutic choice is conditioned by the correct classification of this heterogeneous group of tumors. Clinical signs are not really useful in the clinical work up, most patients have no symptoms and when clinical signs are present, they may help us to pinpoint the organ of origin but never to identify the type of pathology. In the last few years, the great improvement in imaging has enabled us not only to discriminate cystic from solid lesions, but also to identify the features of the lesions and label them preoperatively. More invasive diagnostic procedures such as fine needle aspiration and intracystic fluid tumor marker level are not really useful because they are not sensitive and the cystic wall can show different degrees of dysplasia and de-epithelialization. These are the reasons for sending the entire specimen to pathology. Good cooperation between surgeons, pathologists, radiologists and gastroenterologists is mandatory to increase the chances of making a proper diagnosis. Therefore, we must analyze all the information we have, such as age, sex, clinical history, location of the tumor and radiological features, in order to avoid the mistake of treating a cystic neoplasm as a benign lesion or as a pseudocyst, as described in the

Variants in mannose‐binding lectin and tumour necrosis factor α affect survival in cystic fibrosis

PubMed Central

Buranawuti, Kitti; Boyle, Michael P; Cheng, Suzanne; Steiner, Lori L; McDougal, Kathryn; Fallin, M Daniele; Merlo, Christian; Zeitlin, Pamela L; Rosenstein, Beryl J; Mogayzel, Peter J; Wang, Xinjing; Cutting, Garry R

2007-01-01

Background Patients with cystic fibrosis with the same mutations in the cystic fibrosis transmembrane conductance regulator (CFTR) gene differ widely in survival suggesting other factors have a substantial role in mortality. Objective To determine if the genotype distribution of variants in three putative cystic fibrosis modifier genes (tumour necrosis factor α (TNFα), transforming growth factor β1 (TGFβ1) or mannose‐binding lectin (MBL2)) differed among patients with cystic fibrosis grouped according to age and survival status. Methods Genotypes of four variants (TNFα‐238, TNFα‐308, TGFβ1‐509 and MBL2 O) were determined in three groups of Caucasians from a single medical centre: 101 children with cystic fibrosis (aged <17 years; mean age 9.4 years), 115 adults with cystic fibrosis (aged ⩾17 years; mean age 30.8 years) and 38 non‐surviving adults with cystic fibrosis (21 deceased and 17 lung transplant after 17 years of age). Genotypes of 127 healthy Caucasians in the same geographical region were used as controls. Kaplan–Meier and Cox hazard regression were used to evaluate the genotype effect on cumulative survival. Results Genotype frequencies among adults and children with cystic fibrosis differed for TNFα‐238 (G/G vs G/A; p = 0.022) and MBL2 (A/A vs O/O; p = 0.016). When adults with cystic fibrosis were compared to non‐surviving adults with cystic fibrosis, genotype frequencies of both genes differed (TNFα‐238G/G vs G/A; p =  0.0015 and MBL2: A/A vs O/O; p = 0.009). The hazard ratio for TNFα‐238G/G vs G/A was 0.25 (95% CI 0.06 to 1.0, p = 0.04) and for MBL2 O/O vs A/A or A/O was 2.5 (95% CI 1.3 to 4.9, p = 0.007). Conclusions TNFα‐238 G/A and MBL2 O/O genotypes appear to be genetic modifiers of survival of cystic fibrosis. PMID:17158822

Cystic fibrous dysplasia in the long bone.

PubMed

Bahk, Won-Jong; Kang, Yong-Koo; Rhee, Seung-Koo; Chung, Yang-Guk; Lee, An-Hee; Bahk, Yong-Whee

2007-10-01

Prominent osteolysis associated with "ground glass" density of fibrous dysplasia may indicate cystic change or sarcomatous transformation. This complication has been reported only sporadically in the long bones. This article presents clinical, radiographic, and pathologic findings, and outcome of simple curettage and bone graft observed in a series of 8 patients with prominent cystic fibrous dysplasia of the long bone. Magnetic resonance imaging features provide a basis for separation of benign cystic change from malignant transformation. However, biopsy is necessary to distinguish nonspecific cystic degeneration from secondary aneurysmal bone cyst. Simple curettage with allo-chip-bone graft is an effective treatment for cystic fibrous dysplasia.

Gastrointestinal Manifestations of Cystic Fibrosis

PubMed Central

2016-01-01

Cystic fibrosis has historically been considered a pulmonary disease, but with the increasing life expectancy of these patients, gastrointestinal manifestations are becoming more important. Furthermore, nutritional status is closely linked to pulmonary function and, thus, overall mortality. This article discusses gastrointestinal manifestations (which involve nutritional, pancreatic, hepatobiliary, and, in particular, gastrointestinal tract issues) of cystic fibrosis as well as management of the disease. In addition, the article discusses studies that have been critical to our understanding of gastrointestinal manifestations of cystic fibrosis. PMID:27330503

Abnormal Ion Permeation through Cystic Fibrosis Respiratory Epithelium

NASA Astrophysics Data System (ADS)

Knowles, M. R.; Stutts, M. J.; Spock, A.; Fischer, N.; Gatzy, J. T.; Boucher, R. C.

1983-09-01

The epithelium of nasal tissue excised from subjects with cystic fibrosis exhibited higher voltage and lower conductance than tissue from control subjects. Basal sodium ion absorption by cystic fibrosis and normal nasal epithelia equaled the short-circuit current and was amiloride-sensitive. Amiloride induced chloride ion secretion in normal but not cystic fibrosis tissue and consequently was more effective in inhibiting the short-circuit current in cystic fibrosis epithelia. Chloride ion-free solution induced a smaller hyperpolarization of cystic fibrosis tissue. The increased voltage and amiloride efficacy in cystic fibrosis reflect absorption of sodium ions across an epithelium that is relatively impermeable to chloride ions.

First detection and molecular characterization of Echinococcus equinus in a mule in Turkey.

PubMed

Simsek, Sami; Cevik, Aydin

2014-10-01

Cystic echinococcosis is a zoonotic disease with a cosmopolital distribution. It is caused by the larval stages (metacestodes) of the parasite Echinococcus granulosus which infects different animal species. In this report, we present a case of E. granulosus infection in a mule and molecular characterization of the cyst. For this purpose parasite material was collected from the liver of a necropsied mule. DNA was isolated and PCR amplification of mitochondrial 12S rRNA as well as partial sequencing of mitochondrial cytochrome c oxidase subunit 1 (mt-CO1) genes were performed. Six unilocular cysts, filled with clear fluid were found in the liver and spleen. All cysts were found to be fertile. The 12S rRNA-PCR did not yield any band while mt-CO1-PCR yielded a 446 bp sized amplification product. Sequence corresponding to mt-CO1 gene was identical to a sequence reported for E. equinus (formerly G4) (Genbank accession number: KC953029). This is the first record of E. equinus as a cause of cystic echinococcosis in a mule in Turkey.

Genetics Home Reference: cystic fibrosis

MedlinePlus

... Foundation) Genetic Testing (1 link) Genetic Testing Registry: Cystic fibrosis Other Diagnosis and Management Resources (5 links) American Society for Reproductive Medicine: Male Infertility Baby's First Test GeneReview: Cystic Fibrosis and Congenital Absence of the Vas Deferens Genomics ...

Induction of ovarian cystic follicles in sheep.

PubMed

Christman, S A; Bailey, M T; Head, W A; Wheaton, J E

2000-10-01

Cystic follicles are a significant cause of infertility in women, dairy cattle and sheep. Sheep were used as a model to identify factors that may elicit formation of cystic follicles. Insulin resistance and elevated LH activity were tested in overweight ewes because of associations among these factors and the formation of cystic follicles. Sheep were synchronized using a progesterone-releasing pessary and insulin resistance was induced during the synchronization period through administration of bovine somatotropin. Following removal of pessaries follicular growth was stimulated by treatment with eCG or eCG and hCG (PG-600). Follicular growth was monitored via daily transrectal ultrasonography and blood samples were collected for hormonal analyses. Six of 18 ewes had a subnormal or absent preovulatory gonadotropin surge and developed cystic follicles. Neither insulin resistance nor elevated LH activity were associated with formation of cystic follicles. Ewes that developed cystic follicles were heavier (93 +/- 4 kg) than ewes that ovulated (81 +/- 3 kg; P = 0.02). Furthermore, following pessary removal and initiation of daily ultrasonography, ewes that developed cystic follicles lost body weight (-3 +/- 1%), while ovulatory ewes continued to gain body weight (1 +/- 1%; P = 0.005). It is speculated that in heavy ewes metabolic factors associated with acute body weight loss inhibit the positive feedback of estradiol and thereby suppress the preovulatory gonadotropin surge leading to formation of cystic follicles.

What's it Like to Have Cystic Fibrosis?

MedlinePlus

... deal with cystic fibrosis. What Is CF? Cystic fibrosis (CF) is a disease that causes the body to make thick, sticky mucus (say: ... special protein. This protein is defective in cystic fibrosis, producing the thick, sticky mucus that causes problems for people with CF. What Causes CF? ...

[Parasitological study of 78 cases of human cystic echinococcosis collected between 2005 to 2012 in Mustapha hospital center of Algiers].

PubMed

Zait, H; Boulahbel, M; Normand, A C; Zait, F; Achir, I; Guerchani, M K; Chaouche, H; Ladjadj, Y; Hamrioui, B

2014-12-01

secondary echinococcosis or the relapses postoperatives. Copyright © 2014 Elsevier Masson SAS. All rights reserved.

Early detection of cystic fibrosis lung disease: multiple‐breath washout versus raised volume tests

PubMed Central

Lum, Sooky; Gustafsson, Per; Ljungberg, Henrik; Hülskamp, Georg; Bush, Andrew; Carr, Siobhán B; Castle, Rosemary; Hoo, Ah‐fong; Price, John; Ranganathan, Sarath; Stroobant, John; Wade, Angie; Wallis, Colin; Wyatt, Hilary; Stocks, Janet

2007-01-01

Background Lung clearance index (LCI), a measure of ventilation inhomogeneity derived from the multiple‐breath inert gas washout (MBW) technique, has been shown to detect abnormal lung function more readily than spirometry in preschool children with cystic fibrosis, but whether this holds true during infancy is unknown. Objectives To compare the extent to which parameters derived from the MBW and the raised lung volume rapid thoraco–abdominal compression (RVRTC) techniques identify diminished airway function in infants with cystic fibrosis when compared with healthy controls. Methods Measurements were performed during quiet sleep, with the tidal breathing MBW technique being performed before the forced expiratory manoeuvres. Results Measurements were obtained in 39 infants with cystic fibrosis (mean (SD) age 41.4 (22.0) weeks) and 21 controls (37.0 (15.1) weeks). Infants with cystic fibrosis had a significantly higher respiratory rate (38 (10) vs 32 (5) bpm) and LCI (8.4 (1.5) vs 7.2 (0.3)), and significantly lower values for all forced expiratory flow‐volume parameters compared with controls. Girls with cystic fibrosis had significantly lower forced expiratory volume (FEV0.5 and FEF25–75 ) than boys (mean (95% CI girls–boys): –1.2 (–2.1 to −0.3) for FEV0.5 Z score; FEF25–75: –1.2 (–2.2 to −0.15)). When using both the MBW and RVRTC techniques, abnormalities were detected in 72% of the infants with cystic fibrosis, with abnormalities detected in 41% using both techniques and a further 15% by each of the two tests performed. Conclusions These findings support the view that inflammatory and/or structural changes in the airways of children with cystic fibrosis start early in life, and have important implications regarding early detection and interventions. Monitoring of early lung disease and functional status in infants and young children with cystic fibrosis may be enhanced by using both MBW and the RVRTC. PMID:17121870

Endoscopic treatment of intraventricular cystic tumors.

PubMed

Margetis, Konstantinos; Souweidane, Mark M

2013-02-01

Intraventricular cystic tumors constitute a surgical challenge, because of their deep location and the histologically benign nature of most of them. We aim to present concisely, yet comprehensively, the role of neuroendoscopy in the treatment of intraventricular cystic tumors. A literature review searching for applications of endoscopy in the treatment of intraventricular cystic tumors is presented. Our experience is added to the presented data. In controversial issues, a comparison is made with traditional treatment methods. Intraventricular endoscopy has been successfully used in the treatment of the whole range of intraventricular cystic tumors. The most common indication is the treatment of colloid cysts. In the treatment of colloid cysts, a comparison with microsurgical techniques showed that endoscopy is advantageous in regard to operative morbidity and postoperative shunt dependency but is associated with a slightly higher recurrence rate. Intraventricular endoscopy has emerged as a viable option in the treatment of intraventricular cystic tumors. Copyright © 2013 Elsevier Inc. All rights reserved.

Vitamin A supplementation for cystic fibrosis.

PubMed

Bonifant, Catherine M; Shevill, Elizabeth; Chang, Anne B

2012-08-15

People with cystic fibrosis and pancreatic insufficiency are at risk of fat soluble vitamin deficiency as these vitamins (A, D, E and K) are co-absorbed with fat. Thus, some cystic fibrosis centres routinely administer these vitamins as supplements but the centres vary in their approach of addressing the possible development of deficiencies in these vitamins. Vitamin A deficiency causes predominantly eye and skin problems while supplementation of vitamin A to excessive levels may cause harm to the respiratory and skeletal systems in children. Thus a systematic review on vitamin A supplementation in people with cystic fibrosis would help guide clinical practice. To determine if vitamin A supplementation in children and adults with CF: 1. reduces the frequency of vitamin A deficiency disorders; 2. improves general and respiratory health; 3. increases the frequency of vitamin A toxicity. We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group Trials Register which comprises of references identified from comprehensive electronic database searches and handsearches of relevant journals and abstract books of conference proceedings.Date of the most recent search of the Group's Cystic Fibrosis Trials Register: 23 May 2012. All randomised or quasi-randomised controlled trials comparing all preparations of oral vitamin A used as a supplement compared to either no supplementation (or placebo) at any dose and for any duration, in children or adults with cystic fibrosis (defined by sweat tests or genetic testing) with and without pancreatic insufficiency. No relevant studies for inclusion were identified in the search. No studies were included in this review. As there were no randomised or quasi-randomised controlled trials identified, we cannot draw any conclusions on the benefits (or otherwise) of regular administration of vitamin A in people with cystic fibrosis. Until further data are available, country or region specific guidelines on the use of vitamin A in

First molecular identification of Echinococcus vogeli and Echinococcus granulosus (sensu stricto) G1 revealed in feces of domestic dogs (Canis familiaris) from Acre, Brazil.

PubMed

das Neves, Leandro Batista; Teixeira, Paulo Eduardo Ferlini; Silva, Sidnei; de Oliveira, Fernanda Bittencourt; Garcia, Daniel Daipert; de Almeida, Fernanda Barbosa; Rodrigues-Silva, Rosângela; Machado-Silva, José Roberto

2017-01-14

Echinococcus granulosus (sensu lato) (s.l.) and Echinococcus vogeli are causative agents of chronic zoonotic diseases such as cystic and polycystic echinococcosis, respectively. In Brazil, polycystic echinococcosis has a restricted geographical distribution in the North Region, while cystic echinococcosis is observed in the South Region. Domestic dogs (Canis familiaris) fed with raw viscera represent a risk factor for E. granulosus (s.l.) infection in the South Region. Although this practice is frequent, it remains unclear whether domestic dogs are infected with E. vogeli in the state of Acre, located in the Amazon basin in the North Region of Brazil. The aim of this study was to investigate this gap in the polycystic echinococcosis epidemiology. Sixty-five fecal samples were collected from the ground in five municipalities (Sena Madureira, n = 14; Rio Branco, n = 06; Bujari, n = 06; Xapuri, n = 30; and Epitaciolândia, n = 09) located in the state of Acre, northern Brazil. The samples were screened for parasites by copro-PCR using the cox1 gene associated with automated sequencing. Echinococcus vogeli was molecularly confirmed in a sample from Sena Madureira and E. granulosus (sensu stricto) (s.s.) (G1) in a sample from Rio Branco. These findings indicate that molecular assays are useful in typing Echinococcus taxa from fecal samples of dogs in northern Brazil. The present study is the first molecular record of E. vogeli in domestic dogs found in the state of Acre, reinforcing their role as a source of infection for humans. Because E. granulosus (s.s.) (G1) was detected for the first time in the North Region, from the epidemiological standpoint this finding is highly relevant, because it expands the known geographical distribution, which was previously restricted to the South Region of Brazil.

Chloride impermeability in cystic fibrosis

NASA Astrophysics Data System (ADS)

Quinton, Paul M.

1983-02-01

Cystic fibrosis is the most common fatal genetic disease affecting Caucasians and is perhaps best characterized as an exocrinopathy involving a disturbance in fluid and electrolyte transport1. A high NaCl concentration in the sweat is characteristic of patients with this disease; the basic physiological reason for this abnormality is unknown. We have microperfused isolated sweat ducts from control subjects and cystic fibrosis patients, and report here results which suggest that abnormally low Cl- permeability in cystic fibrosis leads to poor reabsorption of NaCl in the sweat duct, and hence to a high concentration of NaCl in the sweat.

Cystic Fibrosis (CF) Respiratory Screen: Sputum

MedlinePlus

... for Educators Search English Español Cystic Fibrosis (CF) Respiratory Screen: Sputum KidsHealth / For Parents / Cystic Fibrosis (CF) Respiratory Screen: Sputum What's in this article? What It ...

CXCR4+ granulocytes reflect fungal cystic fibrosis lung disease.

PubMed

Carevic, Melanie; Singh, Anurag; Rieber, Nikolaus; Eickmeier, Olaf; Griese, Matthias; Hector, Andreas; Hartl, Dominik

2015-08-01

Cystic fibrosis airways are frequently colonised with fungi. However, the interaction of these fungi with immune cells and the clinical relevance in cystic fibrosis lung disease are incompletely understood.We characterised granulocytes in airway fluids and peripheral blood from cystic fibrosis patients with and without fungal colonisation, non-cystic fibrosis disease controls and healthy control subjects cross-sectionally and longitudinally and correlated these findings with lung function parameters.Cystic fibrosis patients with chronic fungal colonisation by Aspergillus fumigatus were characterised by an accumulation of a distinct granulocyte subset, expressing the HIV coreceptor CXCR4. Percentages of airway CXCR4(+) granulocytes correlated with lung disease severity in patients with cystic fibrosis.These studies demonstrate that chronic fungal colonisation with A. fumigatus in cystic fibrosis patients is associated with CXCR4(+) airway granulocytes, which may serve as a potential biomarker and therapeutic target in fungal cystic fibrosis lung disease. Copyright ©ERS 2015.

MYB expression and translocation in adenoid cystic carcinomas and other salivary gland tumors with clinicopathologic correlation

PubMed Central

Kong, Christina; Clarke, Nicole; Gilks, Thea; Lipsick, Joe; Cao, Hongbin; Kwok, Shirley; Montgomery, Kelli D.; Varma, Sushama; Le, Quynh-Thu

2011-01-01

Background Adenoid cystic carcinoma is a locally aggressive salivary gland neoplasm which has a poor long term prognosis. A chromosomal translocation involving the genes encoding the transcription factors MYB and NFIB has been recently discovered in these tumors. Methods MYB translocation and protein expression was studied in 37 adenoid cystic carcinomas, 112 other salivary gland neoplasms, and 409 non salivary gland neoplasms by FISH and immunohistochemistry. MYB translocation and expression status in adenoid cystic carcinoma was correlated with clinicopathologic features including outcome, with a median follow up of 77.1 months (range: 23.2–217.5) for living patients. Results A balanced translocation between MYB and NFIB is present in 49% of adenoid cystic carcinomas but is not identified in other salivary gland tumors or non-salivary gland neoplasms. There is no apparent translocation of MYB in 35% of the cases. Strong Myb immunostaining is very specific for adenoid cystic carcinomas but is only present in 65% of all cases. Interestingly, Myb immunostaining is confined to the basal cell component though the translocation is present in all the cells. Neoplasms with MYB translocation demonstrate a trend towards higher local relapse rates, but the results are not statistically significant with current case numbers. Conclusions MYB translocation and expression are useful diagnostic markers for a subset of adenoid cystic carcinomas. The presence of the translocation may be indicative of local aggressive behavior but a larger cohort may be required to demonstrate statistical significance. PMID:21164292

Impairment of Macrophage Presenting Ability and Viability by Echinococcus granulosus Antigens.

PubMed

Mejri, Naceur; Hassen, Imed Eddine; Knapp, Jenny; Saidi, Mouldi

2017-03-01

Despite advances toward an improved understanding of the evasive mechanisms leading to the establishment of cystic echinococcosis, the discovery of specific immunosuppressive mechanisms and related factors are of great interest in the development of an immunotherapeutic approach. To elucidate immunosuppressive effects of bioactive factors contained in chromatographic fractions from hydatid cystic fluid (HCF) of Echinococcus granulosus. Hydatid cystic fluid was fractionated by reverse phase chromatography. Non-specific Concanavalin A-driven proliferation of spleen cells was used to determine specific inhibitory fractions. Trypan blue exclusion test and flowcytometry analysis were performed to check whether highly inhibitory fractions of HCF have apoptotic effect on peritoneal macrophages. Western blot analysis was used to determine proteolytic effects of parasitic antigens on major histocompatibility complex (MHC) class II (I-a) contained in membrane proteins extract from macrophages. High concentrations of HCF and few of chromatographic fractions suppressed spleen cells proliferation. Fractions 7 and 35 were the highest inhibitory fractions. Specifically fraction 35 and to a lesser extent HCF induced apoptosis in peritoneal naive macrophages. However, HCF and the fraction 7 proteolytically altered the expression of MHC class II molecules on peritoneal macrophages. The proteolytic molecule was identified to be a serine protease. Macrophages taken at the chronic and end phase from cystic echinococcosis-infected mice were able to uptake and process C-Ovalbumine-FITC. These cells expressed a drastically reduced level of (I-a) molecules. Our study present new aspects of immune suppression function of E. granulosus. Further molecular characterization of apoptotic and proteolytic factors might be useful to develop immunotherapeutic procedure to break down their inhibitory effects.

Pelvic Hydatidosis Mimicking a Malignant Multicystic Ovarian Tumor

PubMed Central

Mushtaq, Deeba; Verma, Neetu; Mahajan, N. C.

2010-01-01

Echinococcosis is a multisystem disease and has propensity to involve any organ, an unusual anatomical site, and can mimic any disease process. Primary peritoneal echinococcosis is known to occur secondary to hepatic involvement but occasional cases of primary peritoneal hydatid disease including pelvic involvement have also been reported. We report here 1 such case of primary pelvic hydatidosis mimicking a malignant multicystic ovarian tumor where there was no evidence of involvement of the liver or spleen. Our patient, a 27-year-old female, was detected to have a large right cystic adnexal mass on per vaginal examination which was confirmed by ultrasonography. Her biochemical parameters were normal and CA-125 levels, though mildly raised, were below the cut off point. She underwent surgery and on exploratory laparotomy, another cystic mass was found attached to the mesentery of the small gut. The resected cysts were processed histopathologically. On cut sections both large cysts revealed numerous daughter cysts. Microscopic examination of fluid from the cysts revealed free scolices with hooklets and the cyst wall had a typical laminated membrane with inner germinal layer containing degenerated protoplasmic mass. The diagnosis of pelvic hydatid disease was confirmed and patient was managed accordingly. Hydatid disease must be considered while making the differential diagnosis of pelvic cystic masses, especially in endemic areas. PMID:20877508

Populations at Risk for Alveolar Echinococcosis, France

PubMed Central

Piarroux, Martine; Piarroux, Renaud; Knapp, Jenny; Bardonnet, Karine; Dumortier, Jérôme; Watelet, Jérôme; Gerard, Alain; Beytout, Jean; Abergel, Armand; Bresson-Hadni, Solange

2013-01-01

During 1982–2007, alveolar echinococcosis (AE) was diagnosed in 407 patients in France, a country previously known to register half of all European patients. To better define high-risk groups in France, we conducted a national registry-based study to identify areas where persons were at risk and spatial clusters of cases. We interviewed 180 AE patients about their way of life and compared responses to those of 517 controls. We found that almost all AE patients lived in 22 départements in eastern and central France (relative risk 78.63, 95% CI 52.84–117.02). Classification and regression tree analysis showed that the main risk factor was living in AE-endemic areas. There, most at-risk populations lived in rural settings (odds ratio [OR] 66.67, 95% CI 6.21–464.51 for farmers and OR 6.98, 95% CI 2.88–18.25 for other persons) or gardened in nonrural settings (OR 4.30, 95% CI 1.82–10.91). These findings can help sensitization campaigns focus on specific groups. PMID:23647623

Molecular diagnosis of cystic fibrosis.

PubMed

Shrimpton, Antony E

2002-05-01

A review of the current molecular diagnosis of cystic fibrosis including an introduction to cystic fibrosis, the gene function, the phenotypic variation, who should be screened for which mutation, newborn and couple screening, quality assurance, phenotype-genotype correlation, methods and method limitations, options, statements, recommendations, useful Websites and treatments.

Cystic precursors to invasive pancreatic cancer

PubMed Central

Matthaei, Hanno; Schulick, Richard D.; Hruban, Ralph H.; Maitra, Anirban

2011-01-01

Improvements in the sensitivity and quality of cross-sectional imaging have led to increasing numbers of patients being diagnosed with cystic lesions of the pancreas. In parallel, clinical, radiological, pathological and molecular studies have improved the systems for classifying these cysts. Patients with asymptomatic serous cystic neoplasms can be managed conservatively with regular monitoring; however, the clinical management of patients with intraductal papillary mucinous neoplasms and mucinous cystic neoplasms is far more challenging, as it is difficult to determine whether these lesions will progress to malignancy. Fortunately, prospective studies have helped to establish that proposed clinical and radiological criteria (the Sendai guidelines) can be used to guide the care of patients with cystic lesions of the pancreas. Despite this progress in imaging and clinical guidelines, sensitive and specific tests have not yet been developed that can reliably predict the histology and biological properties of a cystic lesion. Such biomarkers are urgently needed, as noninvasive precursors of pancreatic cancer are curable, while the vast majority of invasive pancreatic adenocarcinomas are not. PMID:21383670

Cystic fibrosis screening in assisted reproduction.

PubMed

Gazvani, Rafet; Lewis-Jones, Iwan

2006-06-01

The purpose of this review is to discuss the incidence of cystic fibrosis in the general population, in ethnically diverse populations and specifically in couples needing assisted reproduction caused by male factor subfertility. We review the current understanding of risks for reproductive couples and discuss ideal screening strategies. In ethnically diverse populations, a large difference in clinical sensitivity and birth prevalence exists between the broad racial/ethnic groups examined. Extensive data clearly demonstrate the cost-effectiveness of cystic fibrosis screening. Testing for cystic fibrosis gene mutations is reliable and, with a 26-mutation panel, nearly 90% of possible severe mutations can be detected. To halve the incidence of cystic fibrosis in the community, by offering genetic testing of the fetus if both partners are carrier positive, may also be possible. Recent guidelines suggest that all couples contemplating pregnancy should be informed of molecular screening for cystic fibrosis carrier status for purposes of genetic counselling. In ethnically diverse populations, ethnic-specific mutations should be included in the mutation panels.

Cystic renal tumors: new entities and novel concepts.

PubMed

Moch, Holger

2010-05-01

Cystic renal neoplasms and renal epithelial stromal tumors are diagnostically challenging and represent some novel tumor entities. In this article, clinical and pathologic features of established and novel entities are discussed. Predominantly cystic renal tumors include cystic nephroma/mixed epithelial and stromal tumor, synovial sarcoma, and multilocular cystic renal cell carcinoma. These entities are own tumor entities of the 2004 WHO classification of renal tumors. Tubulocystic carcinoma and acquired cystic disease-associated renal cell carcinoma are neoplasms with an intrinsically cystic growth pattern. Both tumor types should be included in a future WHO classification as novel entities owing to their characteristic features. Cysts and clear cell renal cell carcinoma frequently coexist within the kidneys of patients with von Hippel-Lindau disease. Sporadic clear cell renal cell carcinomas often contain cysts, usually as a minor component. Some clear cell renal cell carcinomas have prominent cysts, and multilocular cystic renal cell carcinoma is composed almost exclusively of cysts. Recent molecular findings suggest that clear cell renal cancer may develop through cyst-dependent and cyst-independent molecular pathways.

Cystic Echinococcal Liver Disease: New Insights into an Old Disease and an Algorithm for Therapy Planning

DOE Office of Scientific and Technical Information (OSTI.GOV)

Rozanes, Izzet, E-mail: rozanes@superonline.com; Gueven, Koray; Acunas, Buelent

2007-11-15

Human cystic echinococcosis (CE) continues to be a major health problem in developing countries. A review of current literature discloses four alternatives for the management of active CE, consisting of surgery, percutaneous treatment (PT), chemotherapy, and follow-up without intervention, but no clear guidelines for directing patients to the different management options. Palliation of symptoms or prevention of complications is the main rationale for the treatment of CE. Surgery has long been considered as the gold standard treatment. However, a meta-analysis comparing the clinical outcomes of patients treated with PT with those of a control group treated with surgery found PTmore » to be more effective, safer, and cheaper. Medical therapy is considered to be ineffective when the criterion of success is defined as the disappearance of the lesion. However, medical therapy seems to be effective when the goal of therapy is defined as the prevention of complications in asymptomatic patients. We propose an algorithm for therapy planning in CE where the first line of therapy for patients with active lesions is PT. Patients with lesions unsuitable for PT are directed to surgery if they are symptomatic, have complicated lesions or have lesions that are prone to rupture. Asymptomatic patients with uncomplicated lesions are directed to medical therapy. Medical therapy failures are redirected to surgery.« less

Cystic Fibrosis Associated with Worse Survival After Liver Transplantation.

PubMed

Black, Sylvester M; Woodley, Frederick W; Tumin, Dmitry; Mumtaz, Khalid; Whitson, Bryan A; Tobias, Joseph D; Hayes, Don

2016-04-01

Survival in cystic fibrosis patients after liver transplantation and liver-lung transplantation is not well studied. To discern survival rates after liver transplantation and liver-lung transplantation in patients with and without cystic fibrosis. The United Network for Organ Sharing database was queried from 1987 to 2013. Univariate Cox proportional hazards, multivariate Cox models, and propensity score matching were performed. Liver transplant and liver-lung transplant were performed in 212 and 53 patients with cystic fibrosis, respectively. Univariate Cox proportional hazards regression identified lower survival in cystic fibrosis after liver transplant compared to a reference non-cystic fibrosis liver transplant cohort (HR 1.248; 95 % CI 1.012, 1.541; p = 0.039). Supplementary analysis found graft survival was similar across the 3 recipient categories (log-rank test: χ(2) 2.68; p = 0.262). Multivariate Cox models identified increased mortality hazard among cystic fibrosis patients undergoing liver transplantation (HR 2.439; 95 % CI 1.709, 3.482; p < 0.001) and liver-lung transplantation (HR 2.753; 95 % CI 1.560, 4.861; p < 0.001). Propensity score matching of cystic fibrosis patients undergoing liver transplantation to non-cystic fibrosis controls identified a greater mortality hazard in the cystic fibrosis cohort using a Cox proportional hazards model stratified on matched pairs (HR 3.167; 95 % CI 1.265, 7.929, p = 0.014). Liver transplantation in cystic fibrosis is associated with poorer long-term patient survival compared to non-cystic fibrosis patients, although the difference is not due to graft survival.

Unilocular cystic lymphangioma of thigh-an extremely rare clinical entity.

PubMed

Thakur, Sudhir Kumar

2010-10-01

Majority of cystic lymphangiomas are multilocular and occur in neck and axilla. The cystic lymphangioma is also known as cystic hygroma. Unilocular cystic hygroma of neck and breast have been reported in literature and have been termed hydrocele of neck and breast respectively. However unilocular cystic hygroma of thigh is probably being reported for the first time.

Intestinal bile acid malabsorption in cystic fibrosis.

PubMed

O'Brien, S; Mulcahy, H; Fenlon, H; O'Broin, A; Casey, M; Burke, A; FitzGerald, M X; Hegarty, J E

1993-08-01

This study aimed at examining the mechanisms participating in excessive faecal bile acid loss in cystic fibrosis. The study was designed to define the relation between faecal fat and faecal bile acid loss in patients with and without cystic fibrosis related liver disease; to assess terminal ileal bile acid absorption by a seven day whole body retention of selenium labelled homotaurocholic acid (SeHCAT); and to determine if small intestinal bacterial overgrowth contributes to faecal bile acid loss. The study population comprised 40 patients (27 men; median age 18 years) with cystic fibrosis (n = 8) and without (n = 32) liver disease and eight control subjects. Faecal bile acid excretion was significantly higher in cystic fibrosis patients without liver disease compared with control subjects (mean (SEM) 21.5 (2.4) and 7.3 (1.2) micromoles/kg/24 hours respectively; p < 0.01) and patients with liver disease (7.9 (1.3) micromoles/kg/24 hours; p < 0.01). No correlation was found between faecal fat (g fat/24 hours) and faecal bile acid (micromoles 24 hours) excretion. Eight (33%) of cystic fibrosis patients had seven day SeHCAT retention < 10% (normal retention > 20%). SeHCAT retention in cystic fibrosis patients with liver disease was comparable with control subjects (30.0 (SEM) 8.3% v 36.8 (5.9)%; p = NS) while SeHCAT retention in cystic fibrosis patients who did not have liver disease was significantly reduced (19.9 (3.8); p < 0.05). Although evidence of small bowel bacterial overgrowth was present in 40% of patients no relation was found between breath hydrogen excretion, faecal fat, and faecal bile acid loss. The results are consistent with the presence of an abnormality in terminal ideal function in patients with cystic fibrosis who do not have liver disease and that a defect in the ileal absorption of bile acids may be a contributory factor to excessive faecal bile acid loss. Faecal bile acid loss in cystic fibrosis is unrelated to the presence of intraluminal fat

Intestinal bile acid malabsorption in cystic fibrosis.

PubMed Central

O'Brien, S; Mulcahy, H; Fenlon, H; O'Broin, A; Casey, M; Burke, A; FitzGerald, M X; Hegarty, J E

1993-01-01

This study aimed at examining the mechanisms participating in excessive faecal bile acid loss in cystic fibrosis. The study was designed to define the relation between faecal fat and faecal bile acid loss in patients with and without cystic fibrosis related liver disease; to assess terminal ileal bile acid absorption by a seven day whole body retention of selenium labelled homotaurocholic acid (SeHCAT); and to determine if small intestinal bacterial overgrowth contributes to faecal bile acid loss. The study population comprised 40 patients (27 men; median age 18 years) with cystic fibrosis (n = 8) and without (n = 32) liver disease and eight control subjects. Faecal bile acid excretion was significantly higher in cystic fibrosis patients without liver disease compared with control subjects (mean (SEM) 21.5 (2.4) and 7.3 (1.2) micromoles/kg/24 hours respectively; p < 0.01) and patients with liver disease (7.9 (1.3) micromoles/kg/24 hours; p < 0.01). No correlation was found between faecal fat (g fat/24 hours) and faecal bile acid (micromoles 24 hours) excretion. Eight (33%) of cystic fibrosis patients had seven day SeHCAT retention < 10% (normal retention > 20%). SeHCAT retention in cystic fibrosis patients with liver disease was comparable with control subjects (30.0 (SEM) 8.3% v 36.8 (5.9)%; p = NS) while SeHCAT retention in cystic fibrosis patients who did not have liver disease was significantly reduced (19.9 (3.8); p < 0.05). Although evidence of small bowel bacterial overgrowth was present in 40% of patients no relation was found between breath hydrogen excretion, faecal fat, and faecal bile acid loss. The results are consistent with the presence of an abnormality in terminal ideal function in patients with cystic fibrosis who do not have liver disease and that a defect in the ileal absorption of bile acids may be a contributory factor to excessive faecal bile acid loss. Faecal bile acid loss in cystic fibrosis is unrelated to the presence of intraluminal fat

Year to year change in FEV1 in patients with cystic fibrosis and different mutation classes.

PubMed

De Boeck, K; Zolin, A

2017-03-01

In patients with cystic fibrosis, most treatments addressing the underlying basic defect are mutation or mutation class specific. These treatments are disease modifying if they lower the year to year change in lung function. We therefore calculated the current loss of lung function, measured by year to year change in forced expired volume in 1s in 11,417 patients included in the European Cystic Fibrosis Society Patient Registry. Whereas patients with at least one mutation of class IV or V have on average a lower year to year change, we did not find a difference between patients with a stop codon mutation, homozygous for F508del or at least one class III mutation. These data are useful background information to discuss the impact of different disease modifying treatments. Copyright © 2016 European Cystic Fibrosis Society. Published by Elsevier B.V. All rights reserved.

Anatomical variations of the cystic artery.

PubMed

Mlakar, Bostjan; Gadzijev, Eldar M; Ravnik, Dean; Hribernik, Marija

2003-02-01

Thorough knowledge about the origin of the cystic artery is surgically important, especially when intraoperative or post-operative bleeding occurs in the gallbladder fossa. The arterial supply of the gallbladder was studied in 81 livers. The gallbladder was supplied by one cystic artery in 86% and by two arteries in 14% of cases. When a single artery was present, it originated from the right hepatic artery in 53% of livers. Other origins included the anterior or the posterior sectional hepatic artery, the replacing right hepatic artery, and in 5% of cases, segmental arteries for segments 4, 5, 6 and 8. When two cystic arteries supplied the gallbladder, both most commonly originated from the right hepatic artery (7% incidence). In 1% of cases, a subsegmental branch for segment 6 and a subsegmental branch for segment 5 respectively, originated from the cystic artery.

Longevity of Patients With Cystic Fibrosis in 2000 to 2010 and Beyond: Survival Analysis of the Cystic Fibrosis Foundation Patient Registry

PubMed Central

MacKenzie, Todd; Gifford, Alex H.; Sabadosa, Kathryn A.; Quinton, Hebe B.; Knapp, Emily A.; Goss, Christopher H.; Marshall, Bruce C.

2015-01-01

Background Advances in treatments for cystic fibrosis (CF) continue to extend survival. An updated estimate of survival is needed for better prognostication and to anticipate evolving adult care needs. Objective To characterize trends in CF survival between 2000 and 2010 and to project survival for children born and diagnosed with the disease in 2010. Design Registry-based study. Setting 110 Cystic Fibrosis Foundation–accredited care centers in the United States. Patients All patients represented in the Cystic Fibrosis Foundation Patient Registry (CFFPR) between 2000 and 2010. Measurements Survival was modeled with respect to age, age at diagnosis, gender, race or ethnicity, F508del mutation status, and symptoms at diagnosis. Results Between 2000 and 2010, the number of patients in the CFFPR increased from 21 000 to 26 000, median age increased from 14.3 to 16.7 years, and adjusted mortality decreased by 1.8% per year (95% CI, 0.5% to 2.7%). Males had a 19% (CI, 13% to 24%) lower adjusted risk for death than females. Median survival of children born and diagnosed with CF in 2010 is projected to be 37 years (CI, 35 to 39 years) for females and 40 years (CI, 39 to 42 years) for males if mortality remains at 2010 levels and more than 50 years if mortality continues to decrease at the rate observed between 2000 and 2010. Limitations The CFFPR does not include all patients with CF in the United States, and loss to follow-up and missing data were observed. Additional analyses to address these limitations suggest that the survival projections are conservative. Conclusion Children born and diagnosed with CF in the United States in 2010 are expected to live longer than those born earlier. This has important implications for prognostic discussions and suggests that the health care system should anticipate greater numbers of adults with CF. Primary Funding Source Cystic Fibrosis Foundation. PMID:25133359

Pregnancy and cystic fibrosis: Approach to contemporary management

PubMed Central

Tay, George; Callaway, Leonie; Bell, Scott C

2014-01-01

Over the previous 50 years survival of patients with cystic fibrosis has progressively increased. As a result of improvements in health care, increasing numbers of patients with cystic fibrosis are now considering starting families of their own. For the health care professionals who look after these patients, the assessment of the potential risks, and the process of guiding prospective parents through pregnancy and beyond can be both challenging and rewarding. To facilitate appropriate discussions about pregnancy, health care workers must have a detailed understanding of the various important issues that will ultimately need to be considered for any patient with cystic fibrosis considering parenthood. This review will address these issues. In particular, it will outline pregnancy outcomes for mothers with cystic fibrosis, issues that need to be taken into account when planning a pregnancy and the management of pregnancy for mothers with cystic fibrosis or mothers who have undergone organ transplantation as a result of cystic fibrosis. PMID:27512443

Efficacy of electrocoagulation in sealing the cystic artery and cystic duct occluded with only one absorbable clip during laparoscopic cholecystectomy.

PubMed

Yang, Chang-Ping; Cao, Jin-Lin; Yang, Ren-Rong; Guo, Hong-Rong; Li, Zhao-Hui; Guo, Hai-Ying; Shao, Yin-Can; Liu, Gui-Bao

2014-02-01

Even though laparoscopic cholecystectomy (LC) emerged over 20 years ago, controversies persist with regard to the best method to ligate the cystic duct and artery. We proposed to assess the effectiveness and safety of electrocoagulation to seal the cystic artery and cystic duct after their occlusion with only one absorbable clip. We retrospectively compared the clinical data for 635 patients undergoing LC using electrocoagulation to seal the cystic artery and cystic duct that were occluded with only one absorbable clip (Group 1) and 728 patients undergoing LC using titanium clips (Group 2). In parallel, 30 rabbits randomized into six groups underwent cholecystectomy. After cystic duct ligation with absorbable or titanium clips, the animals were sacrificed 1, 3, or 6 months later, and intraabdominal adhesions were assessed after celiotomy. The mean operative time was significantly shorter (41.6 versus 58.9 minutes, P<.01) in Group 1 than in Group 2. No cystic duct leaks occurred in any patients from Group 1, compared with seven leaks among the 728 (0.96%) patients from Group 2 (P<.05). The morbidity was significantly higher in Group 2 than in Group 1 (3.43% versus 1.58%). Mean intraoperative blood loss and hospitalization length were not significantly different between the two groups, and no deaths occurred in either group. In animal experiments, adhesion was tighter for absorbable than for titanium clips, but fibrous tissue encapsulation was thinner at the site of titanium clips. Electrocoagulation of the cystic artery and cystic duct that were occluded with only one absorbable clip is safe and effective during LC. This approach is associated with shortened operative times and reduced leakage, compared with the standard method using metal clips.

Diabetes in cystic fibrosis.

PubMed

Bridges, Nicola

2013-05-01

Cystic fibrosis related diabetes (CFRD) is a common complication of cystic fibrosis, caused by a fall in insulin secretion with age in individuals with pancreatic insufficiency. CFRD is associated with worse clinical status and increased mortality. Treatment of CFRD with insulin results in sustained improvements in lung function and nutrition. While clinical experience with insulin treatment in CF has increased, the selection of who to treat and glycaemic targets remain unclear. Copyright © 2013. Published by Elsevier Ltd.

Vitamin K supplementation for cystic fibrosis

PubMed Central

Jagannath, Vanitha A; Fedorowicz, Zbys; Thaker, Vidhu; Chang, Anne B

2015-01-01

Background Cystic fibrosis is a genetic disorder which can lead to multiorgan dysfunction. Malabsorption of fat and fat-soluble vitamins (A, D, E, K) may occur and can cause subclinical deficiencies of some of these vitamins. Vitamin K is known to play an important role in both blood coagulation and bone formation. Supplementation with vitamin K appears to be one way of addressing the deficiency, but there is very limited agreement on the appropriate dose and frequency of use of these supplements. Objectives To assess the effects of vitamin K supplementation in people with cystic fibrosis and to determine the optimal dose and route of administration of vitamin K for both routine and therapeutic use. Search methods We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group’s Trials Register comprising references identified from comprehensive electronic database searches and handsearches of relevant journals and abstract books of conference proceedings. Most recent search: 08 October 2014. Selection criteria Randomised and quasi-randomised controlled trials of all preparations of vitamin K used as a supplement compared to either no supplementation (or placebo) at any dose or route and for any duration, in children or adults diagnosed with cystic fibrosis (by sweat test or genetic testing). Data collection and analysis Two authors independently screened papers, extracted trial details and assessed their risk of bias. Main results Two trials (total of 32 participants) each lasting one month were included in the review and were assessed as having a moderate risk of bias. One was a dose-ranging parallel group trial in children (aged 8 to 18 years); and the other (with an older cohort) had a crossover design comparing supplements to no treatment, but no separate data were reported for the first intervention period. Neither of the trials addressed any of the primary outcomes (coagulation, bone formation and quality of life). Both trials reported the restoration

Singing for children and adults with cystic fibrosis.

PubMed

Irons, Jung Yoon; Kenny, Dianna Theadora; Chang, Anne B

2010-05-12

Cystic fibrosis is a genetically inherited, life-threatening condition that affects major organs. The management of cystic fibrosis involves a multi-faceted daily treatment regimen that includes airway clearance physiotherapy, taking pancreatic enzymes and other medications. Previous studies identified that compliance with this intensive treatment especially among adolescents with cystic fibrosis is poor. Because of both the nature and consequences of the illness and the relentless demands of treatments, many individuals with cystic fibrosis are likely to have a poor quality of life. Anecdotal evidence suggests that singing may provide rigorous exercises for the whole respiratory system as well as a means for emotional expression, which may enhance quality of life. To evaluate the effects of a singing intervention in addition to usual therapy on the quality of life, morbidity, respiratory muscle strength and pulmonary function of children and adults with cystic fibrosis. We searched the Group's Cystic Fibrosis Trials Register, the Cochrane Central Register of Controlled Trials, major allied complementary data bases, and clinical trial registers. Hand searching for relevant conference proceedings and journals was also carried out.Date of search of Trials Register: 02 September 2009.Date of additional searches: 17 September 2009. Randomised controlled trials in which singing (as an adjunctive intervention) is compared with either a sham intervention or no singing in people with cystic fibrosis. No trials were found that met the selection criteria. No meta-analysis could be performed. As no studies that met the criteria were found, this review is unable to support or refute the benefits of singing as a therapy for people with cystic fibrosis. Future randomised controlled trials are required to evaluate singing therapy for people with cystic fibrosis.

Diagnosing cystic fibrosis-related diabetes: current methods and challenges.

PubMed

Prentice, Bernadette; Hameed, Shihab; Verge, Charles F; Ooi, Chee Y; Jaffe, Adam; Widger, John

2016-07-01

Cystic fibrosis-related diabetes (CFRD) is the end-point of a spectrum of glucose abnormalities in cystic fibrosis that begins with early insulin deficiency and ultimately results in accelerated nutritional decline and loss of lung function. Current diagnostic and management regimens are unable to entirely reverse this clinical decline. This review summarises the current understanding of the pathophysiology of CFRD, the issues associated with using oral glucose tolerance tests in CF and the challenges faced in making the diagnosis of CFRD. Medline database searches were conducted using search terms "Cystic Fibrosis Related Diabetes", "Cystic Fibrosis" AND "glucose", "Cystic Fibrosis" AND "insulin", "Cystic Fibrosis" AND "Diabetes". Additionally, reference lists were studied. Expert commentary: Increasing evidence points to early glucose abnormalities being clinically relevant in cystic fibrosis and as such novel diagnostic methods such as continuous glucose monitoring or 30 minute sampled oral glucose tolerance test (OGTT) may play a key role in the future in the screening and diagnosis of early glucose abnormalities in CF.

Radiological analysis of cystic lesion in osteonecrosis of the femoral head.

PubMed

Gao, Fuqiang; Han, Jun; He, Zike; Li, Zirong

2018-04-27

Cystic lesions are a common complication in osteonecrosis of the femoral head (ONFH). This study will discuss the cause of cystic lesion formation and the feature of cystic lesion distribution in ONFH. According to the feature of cystic lesion in ONFH, we will discuss the possible mechanisms of cystic lesions and their  influence on collapse of the femoral head. We retrospectively gathered 102 ONFH patients (168 hips) from November in 2015 to August in 2016 on China-Japan Friendship Hospital. Three categories of patients' medical information were collected: demographic characteristics, bone cystic lesion location, and pathological finding on CT and MRI imaging (microfracture, collapse, crescent sign). On mid-coronal and mid-axial CT section, the femoral head was divided into four quadrants for locating the cystic lesion. And we classified the location relationship of cystic lesion and sclerosis rim as G1 type, G2 type, and G3 type on coronal CT section. A significant difference was found between ONFH group with cystic lesion and ONFH group without cystic lesion in terms of microfracture (P < 0.001), collapse (P < 0.001), and crescent sign (P < 0.001). Forty-four cystic lesions (70%) are located in anterior hip area and 19 cystic lesions (30%) are located in posterior hip area. There were 14, 24, and seven cystic lesions (31, 53, 16%) locating in lateral, central, and medial pillars of the femoral head. G2 type was the most common pattern of location relationship between cystic lesion and sclerosis rim. Cystic lesions are often found near sclerosis rim in ONFH. The femoral head with osteonecrosis complicating by cystic lesions is more likely to accompany microfracture, collapse, and crescent sign which indicate structural instability in the femoral head. Cystic lesion in ONFH plays an important role in aggravating the progression of femoral head collapse. The peak stress from sclerosis rim may be a main factor inducing the formation of cystic lesion in

Human Alveolar Echinococcosis in Poland: 1990–2011

PubMed Central

Nahorski, Wacław L.; Knap, Józef P.; Pawłowski, Zbigniew S.; Krawczyk, Marek; Polański, Jerzy; Stefaniak, Jerzy; Patkowski, Waldemar; Szostakowska, Beata; Pietkiewicz, Halina; Grzeszczuk, Anna; Felczak-Korzybska, Iwona; Gołąb, Elżbieta; Wnukowska, Natalia; Paul, Małgorzata; Kacprzak, Elżbieta; Sokolewicz-Bobrowska, Elżbieta; Niścigorska-Olsen, Jolanta; Czyrznikowska, Aleksandra; Chomicz, Lidia; Cielecka, Danuta; Myjak, Przemysław

2013-01-01

Background Alveolar echinococcosis (AE) caused by Echinococcus multilocularis infections is a dangerous old disease in the Northern Hemisphere. The aim of the paper was to collect and analyze data on human AE in Poland in the last two decades. Methodology/Principal Findings The sources of data were both the cases officially registered and detected by an active field and laboratory surveillance. The cases were verified by clinical, epidemiological, and laboratory criteria. Altogether 121 human cases of AE were detected. Among these 83 (68,6%) cases were classified as confirmed, 16 as probable and 22 as possible. During the two decades a continuous increase in detection rate was noticed. The cases were 6–82 years old at the time of diagnosis (mean - 47.7 years). Sex ratio M/F was 0.86/1.0. The AE was fatal in 23 (19%) patients (mean age at death - 54.1 years). Family agglomeration of AE was found in 4 foci, involving 9 patients. Seventy six of the cases were diagnosed in an advanced stage of disease. In all cases the liver was the primary location of AE. In 30 (24.8%) patients a spread to other organs was observed. Ninety four of the patients were treated with albendazole. In 73 (60%) patients a surgical operation was performed, including 15 liver transplantations. Conclusions/Significance The studies confirmed that AE is an emerging disease in Poland, which is the fourth country in Europe with over 120 cases detected. The results also indicate the need of a wider national programme for implementation of screening in the highest AE risk areas (north-eastern Poland) with an effort to increase the public awareness of the possibility of contracting E. multilocularis, and above all, training of the primary care physicians in the recognition of the risk of AE to allow for an early detection of this dangerous disease. PMID:23301116

Molecular genotyping of Echinococcus granulosus in the North of Iraq.

PubMed

Hammad, Salam Jumaah; Cavallero, Serena; Milardi, Giovanni Luigi; Gabrielli, Simona; D Amelio, Stefano; Al-Nasiri, Fatima Shihab

2018-01-15

Cystic echinococcosis/hydatidosis is an important cosmopolitan zoonotic disease that causes large economic losses and human suffering. The larval stages of Echinococcus granulosus are the etiological agents of cystic echinococcosis that showed different genotypes in different regions in the world. The present study was aimed at the detection of E. granulosus strains circulating in two cities from north of Iraq (Kirkuk and Sulaimania). A total of 47 specimens of hydatid cysts were collected from patients and from different domestic intermediate hosts including cattle, sheep, goat and buffalo from slaughterhouses. Molecular characterization was performed by direct sequencing of the mitochondrial DNA (mtDNA) genes coding for the cytochrome c oxidase I (cox1) and the small subunit ribosomal RNA (rrnS). The results showed a high prevalence for the sheep strain (G1), an isolated finding of the buffalo strain (G3) and the presence of seven and three different microvariants for cox1 and rrnS, respectively. This is the first contribution on molecular genotyping of E. granulosus in Iraq with the observation of genotypes other than G1. Copyright © 2017 Elsevier B.V. All rights reserved.

Chloride and potassium channels in cystic fibrosis airway epithelia

NASA Astrophysics Data System (ADS)

Welsh, Michael J.; Liedtke, Carole M.

1986-07-01

Cystic fibrosis, the most common lethal genetic disease in Caucasians, is characterized by a decreased permeability in sweat gland duct and airway epithelia. In sweat duct epithelium, a decreased Cl- permeability accounts for the abnormally increased salt content of sweat1. In airway epithelia a decreased Cl- permeability, and possibly increased sodium absorption, may account for the abnormal respiratory tract fluid2,3. The Cl- impermeability has been localized to the apical membrane of cystic fibrosis airway epithelial cells4. The finding that hormonally regulated Cl- channels make the apical membrane Cl- permeable in normal airway epithelial cells5 suggested abnormal Cl- channel function in cystic fibrosis. Here we report that excised, cell-free patches of membrane from cystic fibrosis epithelial cells contain Cl- channels that have the same conductive properties as Cl- channels from normal cells. However, Cl- channels from cystic fibrosis cells did not open when they were attached to the cell. These findings suggest defective regulation of Cl- channels in cystic fibrosis epithelia; to begin to address this issue, we performed two studies. First, we found that isoprenaline, which stimulates Cl- secretion, increases cellular levels of cyclic AMP in a similar manner in cystic fibrosis and non-cystic fibrosis epithelial cells. Second, we show that adrenergic agonists open calcium-activated potassium channels, indirectly suggesting that calcium-dependent stimulus-response coupling is intact in cystic fibrosis. These data suggest defective regulation of Cl- channels at a site distal to cAMP accumulation.

New animal models of cystic fibrosis: what are they teaching us?

PubMed Central

Keiser, Nicholas W.; Engelhardt, John F.

2013-01-01

Purpose of review Cystic fibrosis is the first human genetic disease to benefit from the directed engineering of three different species of animal models (mice, pigs, and ferrets). Recent studies on the cystic fibrosis pig and ferret models are providing new information about the pathophysiology of cystic fibrosis in various organ systems. Additionally, new conditional cystic fibrosis transmembrane conductance regulator (CFTR) knockout mice are teaching unexpected lessons about CFTR function in surprising cellular locations. Comparisons between these animal models and the human condition are key to dissecting the complexities of disease pathophysiology in cystic fibrosis. Recent findings Cystic fibrosis pigs and ferrets have provided new models to study the spontaneous development of disease in the lung and pancreas, two organs that are largely spared overt spontaneous disease in cystic fibrosis mice. New cystic fibrosis mouse models are now interrogating CFTR functions involved in growth and inflammation at an organ-based level using conditional knockout technology. Together, these models are providing new insights on the human condition. Summary Basic and clinical cystic fibrosis research will benefit greatly from the comparative pathophysiology of cystic fibrosis mice, pigs, and ferrets. Both similarities and differences between these three cystic fibrosis models will inform pathophysiologically important mechanisms of CFTR function in humans and aid in the development of both organ-specific and general therapies for cystic fibrosis. PMID:21857224

Enteral tube feeding for cystic fibrosis.

PubMed

Conway, S P; Morton, A; Wolfe, S

2008-04-16

Enteral tube feeding is routinely used in many cystic fibrosis centres when weight for height percentage is less than 85%, when there has been weight loss for longer than a two-month period or when there has been no weight gain for two to three months (under five years old) or for six months (over five years old). To examine the evidence that in people with cystic fibrosis supplemental enteral tube feeding improves nutritional status, respiratory function, and quality of life without significant adverse effects. We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group Trials Register which comprises references identified from comprehensive electronic database searches and handsearches of relevant journals and abstract books of conference proceedings. We also contacted the companies that market enteral feeds and reviewed their databases. Date of the most recent search of the Group's Cystic Fibrosis Trials Register: November 2007. All randomised controlled trials comparing supplemental enteral tube feeding for one month or longer with no specific intervention in people with cystic fibrosis. Thirteen trials were identified by the search; however, none were eligible for inclusion in this review. There are no trials included in this review. Supplemental enteral tube feeding is widely used throughout the world to improve nutritional status in people with cystic fibrosis. The methods mostly used, nasogastric or gastrostomy feeding, are invasive, expensive, and may have a negative effect on self-esteem and body image. Reported use of enteral tube feeding suggests that it results in nutritional and respiratory improvement and it is disappointing that their efficacy has not been fully assessed by randomised controlled trials. With the more frequent recommendations to use enteral tube feeding as an early rather than a late intervention, this systematic review identifies the need for a multicentre, randomised controlled trial assessing both efficacy and possible

Fatal Airway Obstruction in a Man With a Cystic Hygroma.

PubMed

Wygant, Cassandra Maria; Cohle, Stephen D

2018-05-03

We describe a 24-year-old man with a cystic hygroma of the left side of the lower neck that led to sudden death. Cystic hygroma (cystic lymphangioma) is a congenital malformation of the lymphatic system. The patient, who had a tracheostomy because of airway obstruction from the cystic hygroma, was found dead with his tracheostomy tube on the floor next to him. Complications of cystic hygroma include infiltration of the neck causing airway obstruction, dysphagia, pain, and obstructive sleep apnea.

Effect of amiloride and saline on nasal mucociliary clearance and potential difference in cystic fibrosis and normal subjects.

PubMed Central

Middleton, P. G.; Geddes, D. M.; Alton, E. W.

1993-01-01

BACKGROUND--Mucociliary clearance is an important component of pulmonary defence. Maximum clearance is thought to depend on an optimal depth of the sol layer, allowing the most efficient interaction between the cilia and the overlying mucus layer. Sodium absorption, the major ion transport in human airways, is thought to be important in the regulation of the depth of the sol layer. In the airways of patients with cystic fibrosis sodium absorption is increased and mucociliary clearance decreased. Amiloride, a sodium channel blocker, has been shown to improve pulmonary mucociliary clearance in patients with cystic fibrosis. However, its effects on nasal mucociliary clearance in either normal subjects or those with cystic fibrosis are unknown. A study was therefore performed to investigate whether nebulised amiloride improves nasal mucociliary clearance in normal or cystic fibrosis subjects. METHODS--Nasal mucociliary clearance was measured by the saccharin clearance technique in 12 normal subjects and 12 with cystic fibrosis. For the control study measurements were made on two consecutive days and the mean time for each subject averaged. For the drug study measurements were also made on two consecutive days, after administration of nasally nebulised amiloride or placebo (saline) in a double blind manner. Nasal potential difference was measured in eight patients with cystic fibrosis after the administration of amiloride or placebo to assess the efficacy of deposition and duration of action. RESULTS--Baseline values of mucociliary clearance were significantly faster in the normal subjects than in those with cystic fibrosis. In both groups mucociliary clearance was increased after both saline and amiloride, with no significant difference between either treatment. As previously reported, baseline nasal potential difference was significantly more negative in the subjects with cystic fibrosis. Amiloride significantly reduced the potential difference for at least 60 minutes

Acute Scedosporium apiospermum Endobronchial Infection in Cystic Fibrosis.

PubMed

Padoan, Rita; Poli, Piercarlo; Colombrita, Domenico; Borghi, Elisa; Timpano, Silviana; Berlucchi, Marco

2016-06-01

Fungi are known pathogens in cystic fibrosis patients. A boy with cystic fibrosis boy presented with acute respiratory distress. Bronchoscopy showed airways obstruction by mucus plugs and bronchial casts. Scedosporium apiospermum was identified as the only pathogen. Bronchoalveolar lavage successfully resolved the acute obstruction. Plastic bronchitis is a new clinical picture of acute Scedosporium endobronchial colonization in cystic fibrosis patients.

Cystic fibrosis-related diabetes: a distinct condition.

PubMed

Cano Megías, Marta; González Albarrán, Olga

2015-01-01

Cystic fibrosis is the most common fatal inherited autosomal recessive disease in Caucasians, affecting approximately one out of every 2,000 births. Survival of patients with cystic fibrosis has significantly improved due to advances in respiratory and nutritional care, and their current average life expectancy is 30-40 years. Development of cystic fibrosis-related diabetes is a comorbidity that increases with age and may reach a prevalence up to 50% in adults. Its development is associated to impaired lung function and nutritional status, and early diagnosis and treatment are therefore essential to improve quality of life and performance status. Insulin therapy for diabetes and other early carbohydrate metabolism disorders may improve lung function and nutritional status of patients with cystic fibrosis. Copyright © 2014 SEEN. Published by Elsevier Espana. All rights reserved.

Genomics of mucoepidermoid and adenoid cystic carcinomas.

PubMed

Yan, Kenneth; Yesensky, Jessica; Hasina, Rifat; Agrawal, Nishant

2018-02-01

To report on the current state of the literature on the genetics of mucoepidermoid and adenoid cystic carcinomas of the salivary glands with a focus on genomic screens and recently discovered genetic translocations. A PubMed based literature review was performed to query for genetics related basic science and preclinical studies about mucoepidermoid and adenoid cystic carcinomas of the salivary glands. Genetic translocations between CRTC1 and MAML2 in mucoepidermoid carcinoma and between MYB and NFIB in adenoid cystic carcinoma have been recently discovered and have therapeutic implications. Key signaling pathways such as the EGFR pathway in mucoepidermoid carcinoma and the Notch pathway, chromatin regulation, and c-kit mediated epithelial-mesenchymal transitions in adenoid cystic carcinoma have recently been elucidated, pointing to possible therapeutic targets in both cancers.

Management of the Upper Airway in Cystic Fibrosis

PubMed Central

Illing, Elisa A.; Woodworth, Bradford A.

2015-01-01

Purpose of Review Upper airway disease engenders significant morbidity for patients with cystic fibrosis and is increasingly recognized as having a much greater role in pulmonary outcomes and quality of life than originally believed. Widespread disparate therapeutic strategies for cystic fibrosis chronic rhinosinusitis underscore the absence of a standardized treatment paradigm. This review outlines the most recent evidence-based trends in the management of upper airway disease in cystic fibrosis. Recent Findings The unified airway theory proposes that the sinuses are a focus of initial bacterial colonization which seeds the lower airway and may play a large role in maintaining lung infections. Mounting evidence suggests more aggressive treatment of the sinuses may confer significant improvement in pulmonary disease and quality of life outcomes in cystic fibrosis patients. However, there is a lack of high-level evidence regarding medical and surgical management of cystic fibrosis chronic rhinosinusitis that makes generalizations difficult. Summary Well designed clinical trials with long-term follow-up concerning medical and surgical interventions for cystic fibrosis sinus disease are required to establish standardized treatment protocols, but increased interest in the sinuses as a bacterial reservoir for pulmonary infections has generated considerable attention. PMID:25250804

Challenges in pulmonary fibrosis · 3: Cystic lung disease

PubMed Central

Cosgrove, Gregory P; Frankel, Stephen K; Brown, Kevin K

2007-01-01

Cystic lung disease is a frequently encountered problem caused by a diverse group of diseases. Distinguishing true cystic lung disease from other entities, such as cavitary lung disease and emphysema, is important given the differing prognostic implications. In this paper the features of the cystic lung diseases are reviewed and contrasted with their mimics, and the clinical and radiographic features of both diffuse (pulmonary Langerhans' cell histiocytosis and lymphangioleiomyomatosis) and focal or multifocal cystic lung disease are discussed. PMID:17726170

[Genetic counseling in cystic fibrosis].

PubMed

Julia, S; Bieth, E

2000-08-01

Genetic counseling is an important part of health care in patients with cystic fibrosis or respiratory diseases associated with the CFTR (cystic fibrosis transmembrane conductance regulator) gene, including certain types of allergic bronchopulmonary aspergilloses or bronchial diseases (diffuse bronchiectasia). The basic goal is to provide patients with information on the transmission of cystic fibrosis and to asses the risk of recurrence. This risk is determined from molecular biology analyses examining the CFTR gene. Genotyping is the only means of screening for the heterozygous state, frequent in the French population (about 1/30). Because of the large number of mutated alleles not covered entirely by the genetic tests, there remains a question of probability expressed as a residual risk of a heterozygous state. A prenatal genotype diagnosis should be proposed to heterozygous couples who have a 25% risk of having a diseased child. Technically, this is almost always possible and the results are highly reliable. Nevertheless, there remains the risks related to sample taking and the ethical issue about which the patients must be informed. Management of these at risk couples who desire a child must be based on a multidisciplinary approach, particularly important when one of the parents has overt cystic fibrosis.

Cystic meningiomas.

PubMed

Borovich, B; Guilburd, J N; Doron, Y; Soustiel, J F; Zaaroor, M; Braun, J; Gruszkiewicz, J; Feinsod, M

1988-01-01

Four cases of cystic meningiomas were found among 194 meningiomas diagnosed by computed tomography (CT) and operated on during a 7 year period, an incidence of 2%. The cysts were in all cases peritumoral. The cyst's wall was the brain itself, and the ependymal ventricular wall was part of their medial boundary. They contained xanthochromic fluid with a high protein content. Three meningiomas were parasagittal and one was adjacent to the pteryon and the external part of the sphenoid ridge. The mural nodules were in 2 cases apparent single nodes although in one it was part of multiple distant and regional growths, in another the tumour was built by the aggregation of 2 nodes, the remaining case was an "en plaque" meningioma. All were definitely attached to the dura. Histological pattern was different in every case. Those parasagittal were: one pure meningotheliomatous, one mixed meningotheliomatous with pseudo psammomatous and lipoblastic sections and one highly vascular angioblastic; the pteryonal case was psammomatous and microcystic. CT diagnosis is difficult because glial, metastatic and other tumours may look cystic and resemble cystic meningiomas. Nevertheless in 3 cases the correct diagnosis was suspected preoperatively because the solid portion of the tumour showed intense and homogeneous contrast enhancement with a sharp edge and was located adjacent to the dura. On the other hand in the remaining case, the parasagittal solid tumour was not readily apparent on CT (the "en plaque", case), and the tentative preoperative diagnosis was of an epidermoid tumour.(ABSTRACT TRUNCATED AT 250 WORDS)

Echinococcus granulosus sensu lato genotypes infecting humans--review of current knowledge.

PubMed

Alvarez Rojas, Cristian A; Romig, Thomas; Lightowlers, Marshall W

2014-01-01

Genetic variability in the species group Echinococcus granulosus sensu lato is well recognised as affecting intermediate host susceptibility and other biological features of the parasites. Molecular methods have allowed discrimination of different genotypes (G1-10 and the 'lion strain'), some of which are now considered separate species. An accumulation of genotypic analyses undertaken on parasite isolates from human cases of cystic echinococcosis provides the basis upon which an assessment is made here of the relative contribution of the different genotypes to human disease. The allocation of samples to G-numbers becomes increasingly difficult, because much more variability than previously recognised exists in the genotypic clusters G1-3 (=E. granulosus sensu stricto) and G6-10 (Echinococcus canadensis). To accommodate the heterogeneous criteria used for genotyping in the literature, we restrict ourselves to differentiate between E. granulosus sensu stricto (G1-3), Echinococcus equinus (G4), Echinococcus ortleppi (G5) and E. canadensis (G6-7, G8, G10). The genotype G1 is responsible for the great majority of human cystic echinococcosis worldwide (88.44%), has the most cosmopolitan distribution and is often associated with transmission via sheep as intermediate hosts. The closely related genotypes G6 and G7 cause a significant number of human infections (11.07%). The genotype G6 was found to be responsible for 7.34% of infections worldwide. This strain is known from Africa and Asia, where it is transmitted mainly by camels (and goats), and South America, where it appears to be mainly transmitted by goats. The G7 genotype has been responsible for 3.73% of human cases of cystic echinococcosis in eastern European countries, where the parasite is transmitted by pigs. Some of the samples (11) could not be identified with a single specific genotype belonging to E. canadensis (G6/10). Rare cases of human cystic echinococcosis have been identified as having been caused by

Genomics of mucoepidermoid and adenoid cystic carcinomas

PubMed Central

Yan, Kenneth; Yesensky, Jessica; Hasina, Rifat

2018-01-01

Objective To report on the current state of the literature on the genetics of mucoepidermoid and adenoid cystic carcinomas of the salivary glands with a focus on genomic screens and recently discovered genetic translocations. Methods A PubMed based literature review was performed to query for genetics related basic science and preclinical studies about mucoepidermoid and adenoid cystic carcinomas of the salivary glands. Results and conclusions Genetic translocations between CRTC1 and MAML2 in mucoepidermoid carcinoma and between MYB and NFIB in adenoid cystic carcinoma have been recently discovered and have therapeutic implications. Key signaling pathways such as the EGFR pathway in mucoepidermoid carcinoma and the Notch pathway, chromatin regulation, and c‐kit mediated epithelial‐mesenchymal transitions in adenoid cystic carcinoma have recently been elucidated, pointing to possible therapeutic targets in both cancers. PMID:29492469

Cystic echinococcosis in slaughtered sheep in Sardinia (Italy).

PubMed

Scala, A; Garippa, G; Varcasia, A; Tranquillo, V M; Genchi, C

2006-01-15

Of 771 regularly slaughtered Sardinian breed sheep, 580 (75%) were found infected with Echinococcus granulosus hydatid cysts. Seventy-nine sheep (10.3%) had at least 1 fertile cyst. The prevalence of sheep infected with purulent/caseous cysts, calcified cysts and sterile cysts was 13, 59 and 28%, respectively. The age of sheep was positively associated with the probability of infection that increased 1.15 fold for each further year of age. Fertile cysts were found in the lungs of 46 sheep (6%) and in the liver of 13 sheep (1.7%), and in the lung and the liver of 20 sheep (2.6%). Most fertile cysts were found in the lungs (314) and most sheep were infected with less than 10 cysts. When analyzed by a mixed-effect logistic model, the probability to find fertile cysts in the lungs was three times higher compared to the liver and it increased with the age of the sheep (rho = 0.70, p < 0.001). Of 4072 collected cysts, 532 were fertile, 178 purulent/caseous, 2339 calcified and 1023 sterile.

Cystic lesion around the hip joint

PubMed Central

Yukata, Kiminori; Nakai, Sho; Goto, Tomohiro; Ikeda, Yuichi; Shimaoka, Yasunori; Yamanaka, Issei; Sairyo, Koichi; Hamawaki, Jun-ichi

2015-01-01

This article presents a narrative review of cystic lesions around the hip and primarily consists of 5 sections: Radiological examination, prevalence, pathogenesis, symptoms, and treatment. Cystic lesions around the hip are usually asymptomatic but may be observed incidentally on imaging examinations, such as computed tomography and magnetic resonance imaging. Some cysts may enlarge because of various pathological factors, such as trauma, osteoarthritis, rheumatoid arthritis, or total hip arthroplasty (THA), and may become symptomatic because of compression of surrounding structures, including the femoral, obturator, or sciatic nerves, external iliac or common femoral artery, femoral or external iliac vein, sigmoid colon, cecum, small bowel, ureters, and bladder. Treatment for symptomatic cystic lesions around the hip joint includes rest, nonsteroidal anti-inflammatory drug administration, needle aspiration, and surgical excision. Furthermore, when these cysts are associated with osteoarthritis, rheumatoid arthritis, and THA, primary or revision THA surgery will be necessary concurrent with cyst excision. Knowledge of the characteristic clinical appearance of cystic masses around the hip will be useful for determining specific diagnoses and treatments. PMID:26495246

Detection of cystic structures using pulsed ultrasonically induced resonant cavitation

NASA Technical Reports Server (NTRS)

Bar-Cohen, Yoseph (Inventor); Kovach, John S. (Inventor)

2002-01-01

Apparatus and method for early detection of cystic structures indicative of ovarian and breast cancers uses ultrasonic wave energy at a unique resonance frequency for inducing cavitation in cystic fluid characteristic of cystic structures in the ovaries associated with ovarian cancer, and in cystic structures in the breast associated with breast cancer. Induced cavitation bubbles in the cystic fluid implode, creating implosion waves which are detected by ultrasonic receiving transducers attached to the abdomen of the patient. Triangulation of the ultrasonic receiving transducers enables the received signals to be processed and analyzed to identify the location and structure of the cyst.

[News in cystic fibrosis].

PubMed

Delaisi, B

2013-08-01

The improvement over the last two decades in the treatment of cystic fibrosis led to an increase in life expectancy approaching 40 years at birth. Logically, the population of adult patients has been increasing and is currently 50% of patients followed in France. These therapeutic advances have justified the establishment in 2003 of a generalized neonatal screening for cystic fibrosis. The latest data of this screening show an incidence of CF of 1/5359 live births, far below the incidence of 1/2500 which was widely accepted twenty years ago. The performance of this screening is currently based on the dosage of trypsin immuno reactive, followed in case of exceeding the threshold of a search of the 30 most common mutations, can detect around 96% of 150 to 200 CF cases every year. Therefore, the possibility of a false negative of the screening cannot be excluded and evocative symptoms of cystic fibrosis, even for children born after 2003, will lead to prescribe a sweat test. While treatments available so far goal consequences of cystic fibrosis, a new therapeutic class to correct the functional defect of the mutated protein, called CFTR modulators, is emerging. Ivacaftor, leader of this new class, belonging to the category of "CFTR potentiator" got its access on the market in September 2012 for patients carrying the G551D mutation. New other molecules, named "CFTR correctors" which can have synergistic effect with ivacaftor and concern patients carrying the most common mutation--DF 508--are under development. Copyright © 2013. Published by Elsevier Masson SAS.

Evaluation of oxfendazole, praziquantel and albendazole against cystic echinococcosis: a randomized clinical trial in naturally infected sheep.

PubMed

Gavidia, Cesar M; Gonzalez, Armando E; Barron, Eduardo A; Ninaquispe, Berenice; Llamosas, Monica; Verastegui, Manuela R; Robinson, Colin; Gilman, Robert H

2010-02-23

Cystic Echinococosis (CE) is a zoonotic disease caused by larval stage Echinococcus granulosus. We determined the effects of high dose of Oxfendazole (OXF), combination Oxfendazole/Praziquantel (PZQ), and combination Albendazole (ABZ)/Praziquantel against CE in sheep. A randomized placebo-controlled trial was carried out on 118 randomly selected ewes. They were randomly assigned to one of the following groups: 1) placebo; 2) OXF 60 mg/Kg of body weight (BW) weekly for four weeks; 3) ABZ 30 mg/Kg BW + PZQ 40 mg/Kg BW weekly for 6 weeks, and 4) OXF 30 mg/Kg BW+ PZQ 40 mg/Kg BW biweekly for 3 administrations (6 weeks). Percent protoscolex (PSC) viability was evaluated using a 0.1% aqueous eosin vital stain for each cyst. "Noninfective" sheep were those that had no viable PSCs; "low-medium infective" were those that had 1% to 60% PSC viability; and "high infective" were those with more than 60% PSC viability. We evaluated 92 of the 118 sheep. ABZ/PZQ led the lowest PSC viability for lung cysts (12.7%), while OXF/PZQ did so for liver cysts (13.5%). The percentage of either "noninfective" or "low-medium infective" sheep was 90%, 93.8% and 88.9% for OXF, ABZ/PZQ and OXF/PZQ group as compared to 50% "noninfective" or "low-medium infective" for placebo. After performing all necropsies, CE prevalence in the flock of sheep was 95.7% (88/92) with a total number of 1094 cysts (12.4 cysts/animal). On average, the two-drug-combination groups resulted pulmonary cysts that were 6 mm smaller and hepatic cysts that were 4.2 mm smaller than placebo (p<0.05). We demonstrate that Oxfendazole at 60 mg, combination Oxfendazole/Praziquantel and combination Albendazole/Praziquantel are successful schemas that can be added to control measures in animals and merits further study for the treatment of animal CE. Further investigations on different schedules of monotherapy or combined chemotherapy are needed, as well as studies to evaluate the safety and efficacy of Oxfendazole in humans.

Peripheral calcifying cystic odontogenic tumour of the maxillary gingiva.

PubMed

de Lima, Ana Paula; Kitakawa, Dárcio; Almeida, Janete Dias; Brandão, Adriana Aigotti Haberbeck; Anbinder, Ana Lia

2012-08-23

Odontogenic tumors are lesions that are derived from remnants of the components of the developing tooth germ. The calcifying cystic odontogenic tumor or calcifying odontogenic cyst is a benign cystic neoplasm of odontogenic origin that is characterized by an ameloblastoma-like epithelium and ghost cells. Calcifying cystic odontogenic tumor may be centrally or peripherally located, and its ghost cells may exhibit calcification, as first described by Gorlin in 1962. Most peripheral calcifying cystic odontogenic tumors are located in the anterior gingiva of the mandible or maxilla. Authors report a rare case of a peripheral calcifying cystic odontogenic tumor of the maxillary gingiva. A 39-year-old male patient presented with a fibrous mass on the attached buccal gingiva of the upper left cuspid teeth. It was 0.7-cm-diameter, painless and it was clinically diagnosed as a peripheral ossifying fibroma. After an excisional biopsy, the diagnosis was peripheric calcifying cystic odontogenic tumor. The patient was monitored for five years following the excision, and no recurrence was detected. All biopsy material must be sent for histological examination. If the histological examination of gingival lesions with innocuous appearance is not performed, the frequency of peripheral calcifying cystic odontogenic tumor and other peripheral odontogenic tumors may be underestimated.

Prevalence of Mycobacterium lentiflavum in cystic fibrosis patients, France.

PubMed

Phelippeau, Michael; Dubus, Jean-Christophe; Reynaud-Gaubert, Martine; Gomez, Carine; Stremler le Bel, Nathalie; Bedotto, Marielle; Prudent, Elsa; Drancourt, Michel

2015-10-26

Mycobacterium lentiflavum is rarely isolated in respiratory tract samples from cystic fibrosis patients. We herein describe an unusually high prevalence of M. lentiflavum in such patients. M. lentiflavum, isolated from the respiratory tract of cystic fibrosis patients, was identified using both rpoB partial sequencing and detected directly in the sputum by using real-time PCR targeting the smpB gene. M. lentiflavum emerged as the third most prevalent nontuberculous mycobacterial species isolated in cystic fibrosis patients in Marseille, France. Six such patients were all male, and two of them may have fulfilled the American Thoracic Society clinical and microbiological criteria for M. lentiflavum potential lung infection. M. lentiflavum was the third most common mycobacteria isolated in cystic fibrosis patients, particularly in six male patients. M. lentiflavum outbreaks are emerging particularly in cystic fibrosis patients.

Cystic fibrosis chronic rhinosinusitis: A comprehensive review

PubMed Central

Chaaban, Mohamad R.; Kejner, Alexandra; Rowe, Steven M.

2013-01-01

Background: Advances in the care of patients with cystic fibrosis (CF) have improved pulmonary outcomes and survival. In addition, rapid developments regarding the underlying genetic and molecular basis of the disease have led to numerous novel targets for treatment. However, clinical and basic scientific research focusing on therapeutic strategies for CF-associated chronic rhinosinusitis (CRS) lags behind the evidence-based approaches currently used for pulmonary disease. Methods: This review evaluates the available literature and provides an update concerning the pathophysiology, current treatment approaches, and future pharmaceutical tactics in the management of CRS in patients with CF. Results: Optimal medical and surgical strategies for CF CRS are lacking because of a dearth of well-performed clinical trials. Medical and surgical interventions are supported primarily by level 2 or 3 evidence and are aimed at improving clearance of mucus, infection, and inflammation. A number of novel therapeutics that target the basic defect in the cystic fibrosis transmembrane conductance regulator channel are currently under investigation. Ivacaftor, a corrector of the G551D mutation, was recently approved by the Food and Drug Administration. However, sinonasal outcomes using this and other novel drugs are pending. Conclusion: CRS is a lifelong disease in CF patients that can lead to substantial morbidity and decreased quality of life. A multidisciplinary approach will be necessary to develop consistent and evidence-based treatment paradigms. PMID:24119602

Topical cystic fibrosis transmembrane conductance regulator gene replacement for cystic fibrosis-related lung disease.

PubMed

Lee, T; Southern, K W

2007-04-18

Cystic fibrosis is caused by a defective gene encoding a protein called the cystic fibrosis transmembrane conductance regulator (CFTR), and is characterised by chronic lung infection resulting in inflammation and progressive lung damage that results in a reduced life expectancy. To determine whether topical CFTR gene replacement therapy to the lungs in people with cystic fibrosis is associated with improvements in clinical outcomes, and to assess any adverse effects. We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group Trials Register comprising references identified from comprehensive electronic database searches, handsearching relevant journals and abstract books of conference proceedings. Date of most recent search: February 2007 Randomised controlled trials comparing topical CFTR gene delivery to the lung, using either viral or non-viral delivery systems, with placebo or an alternative delivery system in people with confirmed cystic fibrosis. The authors independently extracted data and assessed study quality. Authors of included studies were contacted and asked for any available additional data. Meta-analysis was limited due to differing study designs. Three randomised controlled trials met the inclusion criteria for this review, involving a total of 155 participants. Thirteen studies were excluded. The included studies differed in terms of CFTR gene replacement agent and study design, which limited the meta-analysis. Although the first Moss study reported a significant improvement in respiratory function (FEV(1)) 30 days after participants had received their first dose of gene therapy agent, this finding was not confirmed in their larger second study or in our meta-analysis.In participants who received the CFTR gene transfer agents in the Alton study, "influenza-like" symptoms were found (relative risk 7.00 (95% confidence interval (CI) 1.10 to 44.61)). There were no other significant increases in adverse events in any of the studies. Alton

Diagnosis and treatment of endocrine comorbidities in patients with cystic fibrosis.

PubMed

Siwamogsatham, Oranan; Alvarez, Jessica A; Tangpricha, Vin

2014-10-01

The aim of this review is to provide an update on various relevant endocrine aspects of care in adolescents and adults with cystic fibrosis. As life expectancy in cystic fibrosis has continuously improved, endocrine complications have become more apparent. The common endocrine complications include cystic fibrosis related diabetes, cystic fibrosis related bone disease, vitamin D deficiency and poor growth and pubertal development. Thyroid and adrenal disorders have also been reported, although the prevalence appears to be less common. Endocrine diseases are an increasingly recognized complication that has a significant impact on the overall health of individuals with cystic fibrosis. This review summarizes the updated screening and management of endocrine diseases in the cystic fibrosis population.

About Cystic Fibrosis

MedlinePlus

... Testing for Cystic Fibrosis CFTR-Related Metabolic Syndrome (CRMS) How Babies Are Screened in IRT-Only vs. ... Guidelines Infant Care Clinical Care Guidelines Management of CRMS in First 2 Years and Beyond Clinical Care ...

Coproantigens in taeniasis and echinococcosis.

PubMed

Allan, James C; Craig, Philip S

2006-01-01

The application of modern immunodiagnostic or molecular diagnostic techniques has improved the diagnosis of the taeniid cestode infections, echinococcosis and taeniasis. One particularly promising approach is the detection of parasite-specific antigens in faeces (coproantigens). This approach has been applied to both Echinoccocus and Taenia species and it has gained increasingly widespread use. Taeniid coproantigen tests are based on either monoclonal or polyclonal antibodies raised against adult tapeworm antigens. These tests have the following common characteristics; they are largely genus-specific, specificity is high (>95%), parasite antigen can be detected in faeces weeks prior to patency, levels of coproantigen are independent of egg output, coproantigen is stable for days at a range of temperatures (-80 degrees C to 35 degrees C), for several months in formalin-fixed faecal samples, and coproantigen levels drop rapidly (1-5 days) following successful treatment. In the genus Taenia, most work has been done on Taenia solium and coproantigen tests have reliably detected many more tapeworm carriers than microscopy. For Echinococcus species, there is a broad positive correlation between test sensitivity and worm burden with a reliable threshold level for the test of >50 worms. Characterisation of taeniid coproantigens in order to further improve the tests is ongoing. Studies indicate taeniid coproantigens to include high molecular weight (>150 kDa), heavily glycosylated molecules with carbohydrate moieties contributing substantially to the levels of antigen detected in faeces. Application of the existing coproantigen tests in epidemiological and control programmes for Echinococcus and Taenia species infection has begun to contribute to an improved understanding of transmission and of surveillance of these important zoonotic cestodes.

Air trapping and airflow obstruction in newborn cystic fibrosis piglets.

PubMed

Adam, Ryan J; Michalski, Andrew S; Bauer, Christian; Abou Alaiwa, Mahmoud H; Gross, Thomas J; Awadalla, Maged S; Bouzek, Drake C; Gansemer, Nicholas D; Taft, Peter J; Hoegger, Mark J; Diwakar, Amit; Ochs, Matthias; Reinhardt, Joseph M; Hoffman, Eric A; Beichel, Reinhard R; Meyerholz, David K; Stoltz, David A

2013-12-15

Air trapping and airflow obstruction are being increasingly identified in infants with cystic fibrosis. These findings are commonly attributed to airway infection, inflammation, and mucus buildup. To learn if air trapping and airflow obstruction are present before the onset of airway infection and inflammation in cystic fibrosis. On the day they are born, piglets with cystic fibrosis lack airway infection and inflammation. Therefore, we used newborn wild-type piglets and piglets with cystic fibrosis to assess air trapping, airway size, and lung volume with inspiratory and expiratory X-ray computed tomography scans. Micro-computed tomography scanning was used to assess more distal airway sizes. Airway resistance was determined with a mechanical ventilator. Mean linear intercept and alveolar surface area were determined using stereologic methods. On the day they were born, piglets with cystic fibrosis exhibited air trapping more frequently than wild-type piglets (75% vs. 12.5%, respectively). Moreover, newborn piglets with cystic fibrosis had increased airway resistance that was accompanied by luminal size reduction in the trachea, mainstem bronchi, and proximal airways. In contrast, mean linear intercept length, alveolar surface area, and lung volume were similar between both genotypes. The presence of air trapping, airflow obstruction, and airway size reduction in newborn piglets with cystic fibrosis before the onset of airway infection, inflammation, and mucus accumulation indicates that cystic fibrosis impacts airway development. Our findings suggest that early airflow obstruction and air trapping in infants with cystic fibrosis might, in part, be caused by congenital airway abnormalities.

L206W mutation of the cystic fibrosis gene, relatively frequent in French Canadians, is associated with atypical presentations of cystic fibrosis

DOE Office of Scientific and Technical Information (OSTI.GOV)

Rozen, R.; Ferreira-Rajabi, L.; Robb, L.

Cystic fibrosis is caused by mutations in the cystic fibrosis transmembrane conductance regulator (CFTR) gene. Over 400 mutations have been reported at this locus. Although severe forms of cystic fibrosis are usually associated with pancreatic insufficiency, pulmonary dysfunction, and elevated sweat chloride, there is a wide range of phenotypes, including congenital absence of the vas deferens, observed with some of the milder mutations. The L206W mutation, which was first identified in patients from South France, is relatively frequent in French Canadians from Quebec. In this report, we document the atypical form of cystic fibrosis associated with this mutation in amore » cohort of 7 French Canadian probands. 20 refs.« less

A universal array-based multiplexed test for cystic fibrosis carrier screening.

PubMed

Amos, Jean A; Bridge-Cook, Philippa; Ponek, Victor; Jarvis, Michael R

2006-01-01

Cystic fibrosis is a multisystem autosomal recessive disorder with high carrier frequencies in caucasians and significant, but lower, carrier frequencies in other ethnicities. Based on technology that allows high detection of mutations in caucasians and significant detection in other ethnic groups, the American College of Medical Genetics (ACMG) and American College of Obstetricians and Gynecologists (ACOG) have recommended pan-ethnic cystic fibrosis carrier screening for all reproductive couples. This paper discusses carrier screening using the Tag-It multiplex mutation platform and the Cystic Fibrosis Mutation Detection Kit. The Tag-It cystic fibrosis assay is a multiplexed genotyping assay that detects a panel of 40 cystic fibrosis transmembrane conductance regulator mutations including the 23 mutations recommended by the ACMG and ACOG for population screening. A total of 16 additional mutations detected by the Tag-It cystic fibrosis assay may also be common. The assay method is described in detail, and its performance in a genetics reference laboratory performing high-volume cystic fibrosis carrier screening is assessed.

Living with Cystic Fibrosis: A Guide for the Young Adult.

ERIC Educational Resources Information Center

Cystic Fibrosis Foundation, Atlanta, GA.

Intended for the young adult with cystic fibrosis, the booklet provides information on dealing with problems and on advances in treatment and detection related to the disease. Addressed are the following topics: description of cystic fibrosis; inheritance of cystic fibrosis; early diagnosis; friends, careers, and other matters; treatment;…

Effects of Puberty on Cystic Fibrosis Related Pulmonary Exacerbations in Women Versus Men

PubMed Central

Sutton, Shelby; Rosenbluth, Daniel; Raghavan, Deepa; Zheng, Jie; Jain, Raksha

2014-01-01

Summary Background Epidemiologic data from studies of airway diseases, such as asthma, chronic obstructive pulmonary disease, and cystic fibrosis indicate a gender disparity where women have worse outcomes. The explanation for this is largely unknown. We hypothesize that female sex hormones play a role in this gender disparity, predisposing women to more exacerbations and decreased lung function post-puberty. Objective In Cystic Fibrosis, to determine if puberty marks a point of increasing exacerbations and decreasing lung function in women relative to men. Methods Using the United States Cystic Fibrosis Foundation Patient Registry, we used linear regression to compare lung function and rate of pulmonary exacerbations in men versus women before and after puberty. Results Of 5,137 subjects who met inclusion criteria, 2,689 were male and 2,448 were female. Average age of puberty was found to be 13.2 ± 2.2 years in men and 11.2 ± 2.0 years of age in women. Percent predicted FEV1 pre-and post-puberty were no different between males versus females (P = 0.44 pre-puberty and P = 0.16 post-puberty). In contrast, women had a significantly higher rate of pulmonary exacerbations post-puberty than men (1.17 ± 1.35 exacerbations per year in women versus 0.95 ± 1.27 in men; P < 0.001) despite controlling for morphometrics, co-morbidities, and microbiologic variables. Conclusion After puberty, the rate of pulmonary exacerbations increased in adolescent women relative to men with cystic fibrosis, supporting a role for sex hormones in the disease process. Further understanding of the mechanisms that modulate sex hormone receptors in airway disease may serve as future targets for therapy. PMID:23460461

Rehabilitation with Cystic Fibrosis: From Utopia to Reality.

ERIC Educational Resources Information Center

Goldberg, Richard T.; And Others

1980-01-01

The paper dispels some of the myths regarding cystic fibrosis (a genetic metabolism disorder), provides information on the latest developments in rehabilitation, summarizes research in the field, and projects future needs of the patient with cystic fibrosis. (SBH)

Topical cystic fibrosis transmembrane conductance regulator gene replacement for cystic fibrosis-related lung disease.

PubMed

Lee, Tim W R; Southern, Kevin W

2013-11-26

Cystic fibrosis is caused by a defective gene encoding a protein called the cystic fibrosis transmembrane conductance regulator (CFTR), and is characterised by chronic lung infection resulting in inflammation and progressive lung damage that results in a reduced life expectancy. To determine whether topical CFTR gene replacement therapy to the lungs in people with cystic fibrosis is associated with improvements in clinical outcomes, and to assess any adverse effects. We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group Trials Register comprising references identified from comprehensive electronic database searches, handsearching relevant journals and abstract books of conference proceedings.Date of most recent search: 22 August 2013.An additional search of the National Institutes for Health (NIH) Genetic Modification Clinical Research Information System (GeMCRIS) was also performed for the years 1992 to 2013.Date of most recent search: 04 September 2013. Randomised controlled trials comparing topical CFTR gene delivery to the lung, using either viral or non-viral delivery systems, with placebo or an alternative delivery system in people with confirmed cystic fibrosis. The authors independently extracted data and assessed study quality. Authors of included studies were contacted and asked for any available additional data. Meta-analysis was limited due to differing study designs. Three randomised controlled trials met the inclusion criteria for this review, involving a total of 155 participants. Fourteen studies were excluded. The included studies differed in terms of CFTR gene replacement agent and study design, which limited the meta-analysis.Although the first Moss study reported a significant improvement in respiratory function (forced expiratory volume at one second) 30 days after participants had received their first dose of gene therapy agent, this finding was not confirmed in their larger second study or in our meta-analysis.In participants

Topical cystic fibrosis transmembrane conductance regulator gene replacement for cystic fibrosis-related lung disease.

PubMed

Lee, Tim W R; Southern, Kevin W

2012-10-17

Cystic fibrosis is caused by a defective gene encoding a protein called the cystic fibrosis transmembrane conductance regulator (CFTR), and is characterised by chronic lung infection resulting in inflammation and progressive lung damage that results in a reduced life expectancy. To determine whether topical CFTR gene replacement therapy to the lungs in people with cystic fibrosis is associated with improvements in clinical outcomes, and to assess any adverse effects. We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group Trials Register comprising references identified from comprehensive electronic database searches, handsearching relevant journals and abstract books of conference proceedings.Date of most recent search: 19 July 2012.An additional search of the National Institutes for Health (NIH) Genetic Modification Clinical Research Information System (GeMCRIS) was also performed for the years 1992 to 2012.Date of most recent search: 25 July 2012. Randomised controlled trials comparing topical CFTR gene delivery to the lung, using either viral or non-viral delivery systems, with placebo or an alternative delivery system in people with confirmed cystic fibrosis. The authors independently extracted data and assessed study quality. Authors of included studies were contacted and asked for any available additional data. Meta-analysis was limited due to differing study designs. Three randomised controlled trials met the inclusion criteria for this review, involving a total of 155 participants. Fourteen studies were excluded. The included studies differed in terms of CFTR gene replacement agent and study design, which limited the meta-analysis.Although the first Moss study reported a significant improvement in respiratory function (forced expiratory volume at one second) 30 days after participants had received their first dose of gene therapy agent, this finding was not confirmed in their larger second study or in our meta-analysis.In participants who

A cystic fibrosis handbook for teachers.

PubMed

Ryan, L L; Williams, J K

1996-01-01

The purposes of this project were to (1) develop a handbook on cystic fibrosis for elementary school teachers and to (2) pilot this handbook with a group of teachers and school nurses. The project used a descriptive survey design in which parents, teachers, and school nurses of 14 elementary-age children with cystic fibrosis were recruited from one cystic fibrosis clinic. Interest in using the handbook with their child's teachers was elicited from parents; also, interest in using the handbook was obtained by open-ended questions in a mailed survey sent to teachers and school nurses. Levels of teacher and school nurse knowledge were measured with a true/false pretest and posttest instrument. All parents expressed a desire to use the handbook with their child's teachers. Sixty-seven percent of the teachers and 89% of the school nurses returned the survey, and all endorsed the use of the handbook in their classrooms or schools. Comparison of the pretest and posttest scores from the teachers revealed an increase in teachers' knowledge. Scores on pretest and posttest measures from school nurses were high at each testing time. Results support the use of a printed handbook to promote knowledge of cystic fibrosis in teachers and to support communication among nurses, parents, and teachers.

Liver Disease in Cystic Fibrosis: an Update

PubMed Central

Parisi, Giuseppe Fabio; Di Dio, Giovanna; Franzonello, Chiara; Gennaro, Alessia; Rotolo, Novella; Lionetti, Elena; Leonardi, Salvatore

2013-01-01

Context Cystic fibrosis (CF) is the most widespread autosomal recessive genetic disorder that limits life expectation amongst the Caucasian population. As the median survival has increased related to early multidisciplinary intervention, other manifestations of CF have emergedespecially for the broad spectrum of hepatobiliary involvement. The present study reviews the existing literature on liver disease in cystic fibrosis and describes the key issues for an adequate clinical evaluation and management of patients, with a focus on the pathogenetic, clinical and diagnostic-therapeutic aspects of liver disease in CF. Evidence Acquisition A literature search of electronic databases was undertaken for relevant studies published from 1990 about liver disease in cystic fibrosis. The databases searched were: EMBASE, PubMed and Cochrane Library. Results CF is due to mutations in the gene on chromosome 7 that encodes an amino acidic polypeptide named CFTR (cystic fibrosis transmembrane regulator). The hepatic manifestations include particular changes referring to the basic CFTR defect, iatrogenic lesions or consequences of the multisystem disease. Even though hepatobiliary disease is the most common non-pulmonary cause ofmortalityin CF (the third after pulmonary disease and transplant complications), only about the 33%ofCF patients presents clinically significant hepatobiliary disease. Conclusions Liver disease will have a growing impact on survival and quality of life of cystic fibrosis patients because a longer life expectancy and for this it is important its early recognition and a correct clinical management aimed atdelaying the onset of complications. This review could represent an opportunity to encourage researchers to better investigate genotype-phenotype correlation associated with the development of cystic fibrosis liver disease, especially for non-CFTR genetic polymorphisms, and detect predisposed individuals. Therapeutic trials are needed to find strategies of

Dental treatment for people with cystic fibrosis.

PubMed

Harrington, N; Barry, P J; Barry, S M

2016-06-01

To describe the nature and consequences of the multi-system genetic condition cystic fibrosis with a view to ensuring optimal dental treatment planning for these patients. A literature search was conducted to identify the key medical and dental manifestations of cystic fibrosis. These findings are discussed and utilised to create recommendations for treatment planning in patients with cystic fibrosis for the practising dental practitioner. Cystic fibrosis is a complex, lethal, multisystem autosomal recessive disorder resulting from mutations on chromosome 7 which result in dysfunction of an ion channel that sits on epithelial surfaces. Respiratory disease remains the leading cause of mortality. Survival has greatly increased in recent decades secondary to improved treatment and specialist care. Specific dental manifestations of the disease may result from the condition itself or complications of treatment. Modification of patient management may be necessary to provide optimum patient care. The pathophysiology and clinical manifestations are relevant to practicing dental practitioners and inform recommendations to be utilised to ensure optimal treatment planning for these patients.

Congenital cystic neck masses: embryology and imaging appearances, with clinicopathological correlation.

PubMed

Gaddikeri, Santhosh; Vattoth, Surjith; Gaddikeri, Ramya S; Stuart, Royal; Harrison, Keith; Young, Daniel; Bhargava, Puneet

2014-01-01

Congenital cystic masses of the neck are uncommon and can present in any age group. Diagnosis of these lesions can be sometimes challenging. Many of these have characteristic locations and imaging findings. The most common of all congenital cystic neck masses is the thyroglossal duct cyst. The other congenital cystic neck masses are branchial cleft cyst, cystic hygroma (lymphangioma), cervical thymic and bronchogenic cysts, and the floor of the mouth lesions including dermoid and epidermoid cysts. In this review, we illustrate the common congenital cystic neck masses including embryology, clinical findings, imaging features, and histopathological findings. Copyright © 2014 Elsevier, Inc. All rights reserved.

How the airway smooth muscle in cystic fibrosis reacts in proinflammatory conditions: implications for airway hyper-responsiveness and asthma in cystic fibrosis.

PubMed

McCuaig, Sarah; Martin, James G

2013-04-01

Among patients with cystic fibrosis there is a high prevalence (40-70%) of asthma signs and symptoms such as cough and wheezing and airway hyper-responsiveness to inhaled histamine or methacholine. Whether these abnormal airway responses are due to a primary deficiency in the cystic fibrosis transmembrane conductance regulator (CFTR) or are secondary to the inflammatory environment in the cystic fibrosis lungs is not clear. A role for the CFTR in smooth muscle function is emerging, and alterations in contractile signalling have been reported in CFTR-deficient airway smooth muscle. Persistent bacterial infection, especially with Pseudomonas aeruginosa, stimulates interleukin-8 release from the airway epithelium, resulting in neutrophilic inflammation. Increased neutrophilia and skewing of CFTR-deficient T-helper cells to type 2 helper T cells creates an inflammatory environment characterised by high concentrations of tumour necrosis factor α, interleukin-8, and interleukin-13, which might all contribute to increased contractility of airway smooth muscle in cystic fibrosis. An emerging role of interleukin-17, which is raised in patients with cystic fibrosis, in airway smooth muscle proliferation and hyper-responsiveness is apparent. Increased understanding of the molecular mechanisms responsible for the altered smooth muscle physiology in patients with cystic fibrosis might provide insight into airway dysfunction in this disease. Copyright © 2013 Elsevier Ltd. All rights reserved.

[Recurrent benign cystic peritoneal mesothelioma].

PubMed

Stroescu, C; Negulescu, Raluca; Herlea, V; David, L; Ivanov, B; Nitipir, Cornelia; Popescu, I

2008-01-01

The benign cystic peritoneal mesothelioma (BCPM) is a rare neoplasm affecting mainly females at reproductive age. The natural history and physiopathology of the BCPM are not entirely known. It is mainly characterized by the lack of malignant elements, no tendency to metastasis and by a pervasive tendency to generate local recurrences after surgical removal. The clinical manifestations are insidious, uncharacteristic; the benign cystic peritoneal mesothelioma is often discovered during a surgical procedure addressing another condition. Imaging tests can raise the suspicion of BCPM but the diagnostic can only be confirmed by histopathological examination corroborated with an immunohistochemical analysis. There are no long term studies dictating a single therapeutic attitude but a high risk of local recurrences and the possibility of transformation into malignant mesothelioma have lead to the current tendency towards an aggressive treatment of the tumor. We present the case of a recurrent benign cystic peritoneal mesothelioma in a 40 years old female patient, emphasizing the therapeutic approach and the role of radical surgery in the treatment of BPCM.

Physical exercise training for cystic fibrosis.

PubMed

Radtke, Thomas; Nevitt, Sarah J; Hebestreit, Helge; Kriemler, Susi

2017-11-01

Physical exercise training may form an important part of regular care for people with cystic fibrosis. This is an update of a previously published review. To assess the effects of physical exercise training on exercise capacity by peak oxygen consumption, pulmonary function by forced expiratory volume in one second, health-related quality of life and further important patient-relevant outcomes in people with cystic fibrosis. We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group Trials Register which comprises references identified from comprehensive electronic database searches and handsearches of relevant journals and abstract books of conference proceedings.Date of the most recent search: 04 May 2017.We searched ongoing trials registers (clinicaltrials.gov and the WHO ICTRP). Date of most recent search: 10 August 2017. All randomised and quasi-randomised controlled clinical trials comparing exercise training of any type and a minimum duration of two weeks with conventional care (no training) in people with cystic fibrosis. Two authors independently selected studies for inclusion, assessed methodological quality and extracted data. The quality of the evidence was assessed using the GRADE system. Of the 83 studies identified, 15 studies which included 487 participants, met the inclusion criteria. The numbers in each study ranged from nine up to 72 participants; two studies were in adults, seven were in children and adolescents and six studies included all age ranges. Four studies of hospitalised participants lasted less than one month and 11 studies were outpatient-based, lasting between two months and three years. The studies included participants with a wide range of disease severity and employed differing levels of supervision with a mixture of types of training. There was also wide variation in the quality of the included studies.This systematic review shows very low- to low-quality evidence from both short- and long-term studies that in people

A cost description of an adult cystic fibrosis unit and cost analyses of different categories of patients.

PubMed Central

Robson, M; Abbott, J; Webb, K; Dodd, M; Walsworth-Bell, J

1992-01-01

BACKGROUND: There is little information on the costs of running an adult cystic fibrosis centre. The aim of this study was to provide detailed costs to assist funding and planning for these patients. METHODS: The cost of a regional adult cystic fibrosis centre serving 119 cystic fibrosis patients, categorised according to four treatment regimens, was determined. District health authority, family health service authority, and voluntary resources used from April 1989 to March 1990 were determined, with appropriate bases for allocation of costs and patient based costs from local information. RESULTS: The total annual cost of treating the 119 patients was 980,646 pounds, with an average cost 8241 pounds per patient. An outpatient reviewed at three monthly intervals cost 2792 pounds a year; an outpatient receiving intravenous antibiotics cost 8606 pounds; an inpatient receiving intravenous antibiotics cost 13,501 pounds; and a patient needing a high level of care cost 19,955 pounds. Medication accounted for 57% (561,395 pounds) of the total cost. CONCLUSIONS: This analysis has helped us to secure funding for patients with cystic fibrosis and it facilitates the prediction of future requirements. The study also indicates the limitations of using average patient costs and difficulties as a result of the poorly structured British National Health Service accounting and information systems. PMID:1440461

Colonoscopic screening shows increased early incidence and progression of adenomas in cystic fibrosis

PubMed Central

Niccum, David E.; Billings, Joanne L.; Dunitz, Jordan M.; Khoruts, Alexander

2018-01-01

Background Colorectal cancer is an emerging problem in cystic fibrosis (CF). The goal of this study was to evaluate adenoma detection by systematic colonoscopic screening and surveillance. Methods We analyzed prospectively collected results of colonoscopies initiated at age 40 years from 88 CF patients at a single Cystic Fibrosis Center. We also reviewed results of diagnostic colonoscopies from 27 patients aged 30–39 years performed during the same time period at the Center. Results The incidence of polyp detection increased markedly after age 40 in CF patients. Greater than 50% were found to have adenomatous polyps; approximately 25% had advanced adenomas as defined by size and/or histopathology; 3% were found to have colon cancer. Multivariate analysis demonstrated specific risk factors for adenoma formation and progression. Conclusions Early screening and more frequent surveillance should be considered in patients with CF due to early incidence and progression of adenomas in this patient population. PMID:26851188

Microbiology of airway disease in a cohort of patients with Cystic Fibrosis

PubMed Central

Lambiase, Antonietta; Raia, Valeria; Pezzo, Mariassunta Del; Sepe, Angela; Carnovale, Vincenzo; Rossano, Fabio

2006-01-01

Background Recent reports document an increasing incidence of new Gram-negative pathogens such as Stenotrophomonas maltophilia and Alcaligenes xylosoxidans isolated from patients with Cystic Fibrosis, along with an increase in common Gram-negative pathogens such as Pseudomonas aeruginosa and Burkholderia cepacia complex. Furthermore, the increase in multidrug-resistance of such organisms makes the therapeutic management of these patients more problematic. Therefore, careful isolation and identification, and accurate studies of susceptibility to antibiotics are critical for predicting the spread of strains, improving therapeutic measures and facilitating our understanding of the epidemiology of emerging pathogens. The first aim of this study was to determine the incidence and the prevalence of colonization by Gram-negative organisms isolated from respiratory samples of Cystic Fibrosis patients in the Regional Referral Cystic Fibrosis Centre of Naples; the second was to evaluate the spectrum of multidrug-resistance of these organisms. Methods Patients (n = 300) attending the Regional Cystic Fibrosis Unit were enrolled in this study over 3 years. Sputum was processed for microscopic tests and culture. An automated system, Phoenix (Becton Dickinson, Sparks, Maryland, USA), was used for phenotypic identification of all strains; the API 20 NE identification system (bioMérieux, Marcy l'Etoile, France) was used when the identification with the Phoenix system was inaccurate. A PCR-RFLP method was used to characterize the organisms in the Burkholderia cepacia complex. A chemosusceptibility test on microbroth dilutions (Phoenix) was used. Primary outcomes such as FEV1 were correlate with different pathogens. Results During the period of study, 40% of patients was infected by Pseudomonas aeruginosa, 7% by Burkholderia cepacia complex, 11% by Stenotrophomonas maltophilia and 7% by Alcaligenes xylosoxidans. Of the strains isolated, 460 were multidrug-resistant. Multiresistant

Inadequate erythroid response to hypoxia in cystic fibrosis.

PubMed

Vichinsky, E P; Pennathur-Das, R; Nickerson, B; Minor, M; Kleman, K; Higashino, S; Lubin, B

1984-07-01

An increase in hemoglobin concentration characterizes the normal compensatory response to chronic tissue hypoxia. We observed no such increase in 42 chronically hypoxic patients with cystic fibrosis, in whom the mean concentration was 12.6 gm/dl; one third of the patients were anemic. Compared with patients with cyanotic heart disease, patients with cystic fibrosis did not have a compensatory increase in P50 or 2,3-diphosphoglycerate. Despite anemia, erythropoietin levels in patients with cystic fibrosis were not significantly different from normal control values. The growth of colony-forming units-erythroid in patients with cystic fibrosis was similar to that in control subjects, and there was no inhibition of growth with the addition of autologous serum. Erythropoietin sensitivity, determined by measuring the CFUe dose response curve, was normal in both patients and controls. Results of iron studies were consistent with iron deficiency in the majority of patients. Impaired absorption of iron was observed in six of 13 iron-deficient patients with cystic fibrosis. An inverse correlation between erythrocyte sedimentation rate and peak serum iron was obtained during the iron absorption study. Eight patients who underwent a therapeutic trial of iron demonstrated a 1.8 gm/dl rise in hemoglobin concentration. Two patients with previously documented iron malabsorption responded to parenteral iron therapy after failure to respond to oral supplementation. These studies demonstrate that patients with cystic fibrosis not only have an impaired erythroid response to hypoxia, but are frequently anemic. Their inadequate erythroid response to hypoxia results in part from disturbances in erythropoietin regulation and iron availability.

Adult cystic hygroma: successful use of OK-432 (Picibanil).

PubMed

Woolley, S L; Smith, D R K; Quine, S

2008-11-01

We report an adult case of cystic lymphangioma treated with OK-432 (Picibanil). A case report and review of the literature concerning the use of OK-432 to treat cystic lymphangioma is presented. A 31-year-old woman developed a cystic lymphangioma four weeks post-partum. This was treated initially by aspiration, for diagnostic purposes. Investigation suggested that surgery would be challenging. A review of the literature demonstrated success with OK-432 in the treatment of this condition, although primarily in the paediatric population. This patient was successfully treated thus, and at the time of writing remained symptom free. A suggested management plan is outlined. Treatment with OK-432 is useful in the management of cystic lymphangiomas in adults and should be considered as first line treatment.

Normal sweat chloride test does not rule out cystic fibrosis.

PubMed

Başaran, Abdurrahman Erdem; Karataş-Torun, Nimet; Maslak, İbrahim Cemal; Bingöl, Ayşen; Alper, Özgül M

2017-01-01

Başaran AE, Karataş-Torun N, Maslak İC, Bingöl A, Alper ÖM. Normal sweat chloride test does not rule out cystic fibrosis. Turk J Pediatr 2017; 59: 68-70. A 5-month-old patient presented with complaints of fever and cough. He was hospitalized with the diagnosis of bronchopneumonia and pseudo-Bartter's syndrome. Patient was further investigated for diagnosis of cystic fibrosis. The chloride (Cl) level in sweat was determined within the normal range (25.1 mmol/L, 20.3 mmol/L). CFTR (Cystic Fibrosis Transmembrane Regulator gene; NM_000492.2) genotyping results were positive for p.E92K; p.F1052V mutations. The patient was diagnosed with cystic fibrosis. In our patient, with features of CF and normal sweat test, mutation analysis was helpful for the diagnosis of cystic fibrosis.

Serum lipoprotein concentrations in cystic fibrosis

DOE Office of Scientific and Technical Information (OSTI.GOV)

Vaughan, W.J.; Lindgren, F.T.; Whalen, J.B.

1978-02-17

Two major classes of lipoproteins, low density and high density, are decreased in the serum of patients with cystic fibrosis; major apoproteins are also decreased. Since essential fatty acids and certain fat-soluble vitamins depend on lipoproteins for transport in the serum, knowledge of lipoprotein levels in cystic fibrosis patients could prove valuable in understanding (i) the basis for the abnormally low serum levels of these fatty acids and vitamins and (ii) the effects of therapies involving these molecules.

Laparoscopic management of cystic disease of the liver.

PubMed

Albrink, M H; McAllister, E W; Rosemurgy, A S; Karl, R C; Carey, L C

1994-04-01

Laparoscopic surgical procedures are increasing in scope and in variety. The benefits of decreased wound morbidity and pain have been well documented for multiple procedures that have traditionally required laparotomy. Although there are few controlled studies to document them, these benefits may be evident from simple clinical observation. Cystic disease of the liver is a condition that is treated largely for symptomatic reasons. The so-called noninvasive or radiographic guided methods of treatment for cystic disease of the liver are fraught with high recurrence rates. We present four cases of cystic disease of the liver treated laparoscopically, followed with pertinent discussion.

Lung ultrasound has limited diagnostic value in rare cystic lung diseases: a cross-sectional study.

PubMed

Davidsen, Jesper Rømhild; Bendstrup, Elisabeth; Henriksen, Daniel P; Graumann, Ole; Laursen, Christian B

2017-01-01

Background : Lung ultrasound (LUS) used to identify interstitial syndrome (IS) and pleural thickening related to diffuse parenchymal lung disease (DPLD) has shown significant correlations with ground glass opacity (GGO) on high-resolution computed tomography (HRCT). However, the applicability of LUS in patients with DPLD subtypes as rare cystic lung diseases has not previously been investigated. This study aimed to observe if distinctive LUS findings could be found in patients with lymphangioleiomyomatosis (LAM), pulmonary Langerhans cell histiocytosis (PLCH), and Birt-Hogg-Dubé syndrome (BHDS). Methods : This single centre case-based cross-sectional study of patients diagnosed with LAM, PCLH and BHDS was conducted at a Danish DPLD specialist centre. Patients underwent clinical examination including LUS. LUS findings were compared to findings scored according to a modified Belmaati score on HRCT and reviewed in consensus between two pulmonologists and one radiologist. Results : Twelve patients with HRCT proven cystic lung disease were included, six with LAM, three with PLCH, two with BHDS, and one with uncharacteristic cystic lung disease. The mean age was 48.7 years (SD ± 15.8). In general all had normal LUS findings. IS could not be found in any patients despite GGO presentation on HRCT among 75% of the patients with a Belmaati in the highest category of 0.76-1.00. Pleural thickening on LUS was present in three patients, but with inconsistent findings. Conclusion : This study indicates that LUS has limited value as a diagnostic tool in patients with LAM, PLCH, and BHDS as normal LUS findings did not rule out severe cystic lung disease.

[MALIGNANT TUMORS IN OVARIAN MATURE CYSTIC TERATOMAS INTRAOPERATIVE DIAGNOSTIC BASIS].

PubMed

Khachatryan, A

2016-11-01

Extremely rare ovarian primary tumors formed in a mature cystic teratomaare described in the literature. This research work studies the frequency of malignant mature cystic teratoma, as well as their clinical and morphological features and necessity of intraoperative histological examination of all teratomas. Cases histories of 56 patients, suffering from ovarian mature cystic teratomahave been studied in MC Shengavit in the period of 2003 - 2015. Among them 4 patients with the somatic malignancies were identified. Morphological methods, which are considered to be "gold standard" of tumor investigation, were used in staining the slides with hematoxylin - eosin. According to the literature the secondary malignant transformation rarely occurs and is typical in postmenopausal women, with a frequency of 0.17-3%. According to the results of our study, malignant tumors in mature cystic teratomas were observed in 4 (7,14%) from the total number of mature cystic teratomas (n=56). There was not revealed a correlation between the duration of the complaints, age of the patients, sizes of ovarian mature teratoma and malignization degree. Thus, the greatest difficulties of clinical diagnosis of malignant tumors in the ovarian mature cystic teratomas were in the early stage of the disease, because of a variety of clinical manifestations, not pathognomonic for malignization. All mentioned symptoms may be observed in the patients with usual mature cystic teratomas. Тhis cases confirm the necessity to take tissue samples from the other ovary for intraoperative histopathological evaluation in each case of mature cystic teratomas. It is necessary to examine a large number of tumor sites, to prevent errors in the assessment of the maturity degree of teratoma.

The role of anaerobic bacteria in the cystic fibrosis airway.

PubMed

Sherrard, Laura J; Bell, Scott C; Tunney, Michael M

2016-11-01

Anaerobic bacteria are not only normal commensals, but are also considered opportunistic pathogens and have been identified as persistent members of the lower airway community in people with cystic fibrosis of all ages and stages of disease. Currently, the role of anaerobic bacteria in cystic fibrosis lower airway disease is not well understood. Therefore, this review describes the recent studies relating to the potential pathophysiological role(s) of anaerobes within the cystic fibrosis lungs. The most frequently identified anaerobic bacteria in the lower airways are common to both cystic fibrosis and healthy lungs. Studies have shown that in cystic fibrosis, the relative abundance of anaerobes fluctuates in the lower airways with reduced lung function and increased inflammation associated with a decreased anaerobic load. However, anaerobes found within the lower airways also produce virulence factors, may cause a host inflammatory response and interact synergistically with recognized pathogens. Anaerobic bacteria are potentially members of the airway microbiota in health but could also contribute to the pathogenesis of lower airway disease in cystic fibrosis via both direct and indirect mechanisms. A personalized treatment strategy that maintains a normal microbial community may be possible in the future.

Bilateral Cystic Lymphangioma of Ovary Associated with Chylous Ascites.

PubMed

Nerune, Savitri Mallikarjun; Arakeri, Surekha Ulhas; Patil, Vijaya L; Mulay, Himanshu Dilip

2015-08-01

Intraabdominal cystic lymphangiomas are rare and are located in retroperitoneum, mesentery, omentum and other visceral organs. Lymphangiomas of the ovary are rare and are usually unilateral. Cases with bilateral cystic lymphangiomas of the ovary are reported very rarely in literature. We report a rare case of bilateral cystic lymphangioma of ovary associated with chylous ascites in a 35-year-old lady who presented with complaints of severe dysmenorrhoea and oligomenorrhoea since 6 months with history of chyluria for the past 3 years.

Adenoid Cystic Carcinoma of Buccal Mucosa: A Rare Case Report.

PubMed

Garg, Vipul; Roy, Swati; Khanna, Kaveri Surya; Bakshi, Preeti Sethi; Chauhan, Isha

2016-09-01

Adenoid cystic carcinoma is a malignant neoplasm most commonly originating in salivary glands of head and neck region. Among intra oral adenoid cystic carcinoma, buccal mucosa is among the rarest sites. We report a case of adenoid cystic of buccal mucosa in a 40-year old female. We have discussed the clinical features, histopathology, diagnosis and treatment along with a brief review of the relevant literature. Although the buccal mucosa is an uncommon site for adenoid cystic carcinoma, the relatively indolent growth pattern of this case and its location which is rather atypical for this type of salivary gland malignancy primarily warrants the necessity behind reporting of this case. Secondly, adenoid cystic carcinoma should be considered in the differential diagnosis of mass of buccal mucosa. It is important to identify such cases rather early and surgical removal with adequate margins is the treatment of choice .

Frequent NFIB-associated Gene Rearrangement in Adenoid Cystic Carcinoma of the Vulva.

PubMed

Xing, Deyin; Bakhsh, Salwa; Melnyk, Nataliya; Isacson, Christina; Ho, Julie; Huntsman, David G; Gilks, C Blake; Ronnett, Brigitte M; Horlings, Hugo M

2017-05-01

Adenoid cystic carcinoma is a rare malignant tumor that usually arises in the major and minor salivary glands and other locations containing secretory glands, including the lower female genital tract. Lower female genital tract carcinomas with adenoid cystic differentiation can be subclassified into 2 distinct groups based on the presence or absence of high-risk HPV. Cervical mixed carcinomas with some adenoid cystic differentiation are high-risk HPV-related but pure adenoid cystic carcinomas of vulvar and cervical origin appear to be unrelated to high-risk HPV. Mechanisms by which normal cells give rise to an HPV-unrelated adenoid cystic carcinoma remain largely unknown. Studies demonstrate that chromosomal translocation involving the genes encoding the transcription factors MYB and NFIB functions as a driving force of adenoid cystic carcinomas development regardless of anatomic site. The current study used fluorescence in situ hybridization with 3 different probes including MYB break-apart probe, NFIB break-apart probe, and MYB-NFIB fusion probe to assess for the presence of gene rearrangements in adenoid cystic carcinomas of the vulva. Six (66.7%) of 9 vulvar adenoid cystic carcinomas demonstrated NFIB rearrangement. Of these 6 cases with a disturbed NFIB, only 2 cases (33.3%) were positive for a MYB rearrangement that was also confirmed by a positive MYB-NFIB fusion pattern. NFIB-associated gene rearrangement is a frequent genetic event in vulvar adenoid cystic carcinomas. Chromosome translocations involving NFIB but with an intact MYB indicate the presence of novel oncogenic mechanisms for the development of adenoid cystic carcinomas of the vulva.

Festival food coma in cystic fibrosis.

PubMed

Pandit, Chetan; Graham, Christie; Selvadurai, Hiran; Gaskin, Kevin; Cooper, Peter; van Asperen, Peter

2013-07-01

Children with cystic fibrosis liver disease and portal hypertension are at risk of developing acute hepatic encephalopathy. Even in the presence of normal synthetic liver function these children may have porto-systemic shunting. We report a case of an adolosecent who had cystic fibrosis liver disease and presented with life threatening hepatinc encephalopathy. This case illustrates that it is necessary to consider an appropriate dietary regimen in adolosecents with liver disease to prevent hepatic decompensation. Copyright © 2012 Wiley Periodicals, Inc.

Cytologic characteristics of adenoid cystic carcinoma of the cervix uteri--case report.

PubMed

Barisić, Ana; Mahovlić, Vesna; Ovanin-Rakić, Ana; Skopljanac-Macina, Lada; Rajhvajn, Sanda; Juric, Danijela; Babić, Damir

2010-03-01

Adenoid cystic carcinoma is a rare malignancy, usually found in the salivary glands, although this unusual tumor can be found at other sites of the body. In particular, regarding adenoid cystic carcinoma of the cervix (ACCC) most frequently reports are given for postmenopausal women. In this respect, our work is one among very few in the literature that considers a cytologic picture of this uncommon cervix carcinoma. The case of 74 year old patient with postmenopausal bleeding is described. Both Pap smear and air dried smear of the uterine cervix were analyzed. The cytologic findings revealed very few small clusters of abnormal glandular cells, as well as some amorphous eosinophilic globule-like material, with granulomatous and necrotic background. The latter includes a lot of histiocytes, multinucleated giant cells, large aggregates of epitheloid cells and lymphocytes. Histology revealed the diagnosis of ACCC. We emphasize the importance of careful screening of Pap smear that might be crucial in the case of suspicious clinical findings in postmenopausal women, when the possibility of ACCC has to be considered.

YEAR-TO-YEAR CHANGES IN LUNG FUNCTION IN INDIVIDUALS WITH CYSTIC FIBROSIS

PubMed Central

Liou, Theodore G.; Elkin, Eric P.; Pasta, David J.; Jacobs, Joan R.; Konstan, Michael W.; Morgan, Wayne J.; Wagener, Jeffrey S.

2014-01-01

Background We examined the year-to-year change in FEV1 for individuals and the overall cystic fibrosis population to better understand how individual trends may differ from population trends. Methods We calculated individual yearly changes using the largest annual FEV1 percent predicted (FEV1%) measurement in 20,644 patients (6–45 years old) included in the Epidemiologic Study of Cystic Fibrosis. We calculated yearly population changes using age-specific medians. Results FEV1% predicted decreased 1–3 points per year for individuals, with maximal decreases in 14–15 year olds. Population changes agreed with individual changes up to age 15; however after age 30, yearly population change approximated zero while individual FEV1% predicted decreases were 1–2 points per year. Conclusions Adolescents have the greatest FEV1% predicted decreases; however. loss of FEV1 is a persistent risk in 6–45 year old CF patients. Recognizing individual year-to-year changes may improve patient-specific care and may suggest new methods for measuring program quality. PMID:20471331

Vorinostat in Treating Patients With Locally Advanced, Recurrent, or Metastatic Adenoid Cystic Carcinoma

ClinicalTrials.gov

2018-05-23

Recurrent Oral Cavity Adenoid Cystic Carcinoma; Recurrent Salivary Gland Carcinoma; Salivary Gland Adenoid Cystic Carcinoma; Stage III Major Salivary Gland Cancer AJCC v7; Stage III Oral Cavity Adenoid Cystic Carcinoma AJCC v6 and v7; Stage IVA Major Salivary Gland Cancer AJCC v7; Stage IVA Oral Cavity Adenoid Cystic Carcinoma AJCC v6 and v7; Stage IVB Major Salivary Gland Cancer AJCC v7; Stage IVB Oral Cavity Adenoid Cystic Carcinoma AJCC v6 and v7; Stage IVC Major Salivary Gland Cancer AJCC v7; Stage IVC Oral Cavity Adenoid Cystic Carcinoma AJCC v6 and v7; Tongue Carcinoma

Bilateral Cystic Lymphangioma of Ovary Associated with Chylous Ascites

PubMed Central

Arakeri, Surekha Ulhas; Patil, Vijaya L.; Mulay, Himanshu Dilip

2015-01-01

Intraabdominal cystic lymphangiomas are rare and are located in retroperitoneum, mesentery, omentum and other visceral organs. Lymphangiomas of the ovary are rare and are usually unilateral. Cases with bilateral cystic lymphangiomas of the ovary are reported very rarely in literature. We report a rare case of bilateral cystic lymphangioma of ovary associated with chylous ascites in a 35-year-old lady who presented with complaints of severe dysmenorrhoea and oligomenorrhoea since 6 months with history of chyluria for the past 3 years. PMID:26435959

Cystic brain metastasis is associated with poor prognosis in patients with advanced breast cancer.

PubMed

Sun, Bing; Huang, Zhou; Wu, Shikai; Ding, Lijuan; Shen, Ge; Cha, Lei; Wang, Junliang; Song, Santai

2016-11-08

Brain metastasis (BM) with a cystic component from breast cancer is rare and largely uncharacterized. The purpose of this study was to identify the characteristics of cystic BM in a large cohort of breast cancer patients. A total of 35 eligible patients with cystic BM and 255 patients with solid BM were analyzed. Three factors were significantly associated with an increased probability of developing cystic lesions: age at diagnosis ≤ 40 years, age at BM ≤ 45 years, and poor histological grade (p < 0.05). Patients with cystic metastasis were also characterized by a larger metastasis volume, a shorter progression-free survival (PFS) following their first treatment for BM, and poor overall survival after BM (p < 0.05). Multivariate analysis further demonstrated that local control of cystic BM was only potentially achieved for HER2-negative primary tumors (p = 0.084). Breast cancer patients with parenchymal BM were reviewed from consecutive cases treated at our institution. Cystic BM was defined when the volume of a cystic lesion was greater than 50% of the aggregated volume of all lesions present. Clinicopathologic and radiographic variables were correlated with development of cystic lesions and with prognosis of cystic BM. This study shows that cystic BM from breast cancer, a special morphological type of BM, had worse prognosis than the more commonly observed solid BM. Younger age and low tumor grade were associated with the development of cystic lesions. Further comprehensive research and management of cystic BM are warranted to improve its poor prognosis.

The optimum cut-off value to differentiate Echinococcus granulosus sensu stricto from other species of E. granulosus sensu lato using larval rostellar hook morphometry.

PubMed

Soriano, S V; Pierangeli, N B; Pianciola, L A; Mazzeo, M; Lazzarini, L E; Debiaggi, M F; Bergagna, H F J; Basualdo, J A

2015-01-01

Cystic echinococcosis caused by Echinococcus granulosus sensu lato is one of the most important helminth zoonoses in the world; it affects both humans and livestock. The disease is endemic in Argentina and highly endemic in the province of Neuquén. Considerable genetic and phenotypic variation has been demonstrated in E. granulosus, and ten different genotypes (G1-G10) have been identified using molecular tools. Echinococcus granulosus sensu lato may be considered a species complex, comprised of E. granulosus sensu stricto (G1-G3), E. equinus (G4), E. ortleppi (G5) and E. canadensis (G6-G10). In endemic areas, the characterization of cystic echinococcosis molecular epidemiology is important in order to apply adequate control strategies. A cut-off value for larval large hook total length to distinguish E. granulosus sensu stricto isolates from those produced by other species of the complex was defined for the first time. Overall, 1780 larval hooks of 36 isolates obtained from sheep (n= 11, G1), goats (n= 10, G6), cattle (n= 5, G6) and pigs (n= 10, G7) were analysed. Validation against molecular genotyping as gold standard was carried out using the receiver operating characteristic (ROC) curve analysis. The optimum cut-off value was defined as 26.5 μm. The proposed method showed high sensitivity (97.8%) and specificity (91.1%). Since in most endemic regions the molecular epidemiology of echinococcosis includes the coexistence of the widely distributed E. granulosus sensu stricto G1 strain and other species of the complex, this technique could be useful as a quick and economical tool for epidemiological and surveillance field studies, when fertile cysts are present.

Therapies for Cystic Fibrosis

MedlinePlus

... Testing for Cystic Fibrosis CFTR-Related Metabolic Syndrome (CRMS) How Babies Are Screened in IRT-Only vs. ... Guidelines Infant Care Clinical Care Guidelines Management of CRMS in First 2 Years and Beyond Clinical Care ...

Study of Hydatidosis-Attributed Mortality in Endemic Area

PubMed Central

Belhassen-García, Moncef; Romero-Alegria, Angela; Velasco-Tirado, Virginia; Alonso-Sardón, Montserrat; Lopez-Bernus, Amparo; Alvela-Suarez, Lucia; del Villar, Luis Perez; Carpio-Perez, Adela; Galindo-Perez, Inmaculada; Cordero-Sanchez, Miguel; Pardo-Lledias, Javier

2014-01-01

Background Cystic hydatid disease is still an important health problem in European Mediterranean areas. In spite of being traditionally considered as a “benign” pathology, cystic echinococcosis is an important cause of morbidity in these areas. Nevertheless, there are few analyses of mortality attributed to human hydatidosis. Objective To describe the epidemiology, the mortality rate and the causes of mortality due to E. granulosus infection in an endemic area. Methodology A retrospective study followed up over a period of 14 years (1998–2011). Principal Findings Of the 567 patients diagnosed with hydatid disease over the period 1998–2011, eleven deaths directly related to hydatid disease complications were recorded. Ten patients (90.9%) died due to infectious complications and the remaining one (9.1%) died due to mechanical complications after a massive hemoptysis. We registered a case fatality rate of 1.94% and a mortality rate of 3.1 per 100.000 inhabitants. Conclusions Hydatidosis is still a frequent parasitic disease that causes a considerable mortality. The main causes of mortality in patients with hydatidosis are complications related to the rupture of CE cysts with supurative collangitis. Therefore, an expectant management can be dangerous and it must be only employed in well-selected patients. PMID:24632824

Genetic counselling issues in cystic fibrosis.

PubMed

Culling, Bronwyn; Ogle, Robert

2010-06-01

Cystic fibrosis is a chronic condition for which genetic testing offers much for the individuals affected in terms of an early diagnosis and offers timely additional information for families with regard to family planning and prenatal testing. Genetic counselling encompasses a range of clinical issues for families and forms a complementary resource for clinicians caring for people with cystic fibrosis. This review will discuss the range of genetic information readily available to patients and families through genetic counselling. Copyright 2010 Elsevier Ltd. All rights reserved.

Cystic fibrosis carrier screening in a North American population.

PubMed

Zvereff, Val V; Faruki, Hawazin; Edwards, Marcia; Friedman, Kenneth J

2014-07-01

The aim of this study was to compare the mutation frequency distribution for a 32-mutation panel and a 69-mutation panel used for cystic fibrosis carrier screening. Further aims of the study were to examine the race-specific detection rates provided by both panels and to assess the performance of extended panels in large-scale, population-based cystic fibrosis carrier screening. Although genetic screening for the most common CFTR mutations allows detection of nearly 90% of cystic fibrosis carriers, the large number of other mutations, and their distribution within different ethnic groups, limits the utility of general population screening. Patients referred for cystic fibrosis screening from January 2005 through December 2010 were tested using either a 32-mutation panel (n = 1,601,308 individuals) or a 69-mutation panel (n = 109,830). The carrier frequencies observed for the 69-mutation panel study population (1/36) and Caucasian (1/27) and African-American individuals (1/79) agree well with published cystic fibrosis carrier frequencies; however, a higher carrier frequency was observed for Hispanic-American individuals (1/48) using the 69-mutation panel as compared with the 32-mutation panel (1/69). The 69-mutation panel detected ~20% more mutations than the 32-mutation panel for both African-American and Hispanic-American individuals. Expanded panels using race-specific variants can improve cystic fibrosis carrier detection rates within specific populations. However, it is important that the pathogenicity and the relative frequency of these variants are confirmed.

Vitamin D supplementation for cystic fibrosis.

PubMed

Ferguson, Janet H; Chang, Anne B

2014-05-14

Cystic fibrosis (CF) is a genetic disorder with multiorgan effects. In a subgroup with pancreatic insufficiency malabsorption of the fat soluble vitamins (A, D, E, K) may occur. Vitamin D is involved in calcium homeostasis and bone mineralisation and may have extraskeletal effects. This review examines the evidence for vitamin D supplementation in cystic fibrosis. To assess the effects of vitamin D supplementation on the frequency of vitamin D deficiency, respiratory outcomes and vitamin D toxicity in the cystic fibrosis population. We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group Trials Register comprising references identified from comprehensive electronic database searches and handsearches of relevant journals and abstract books of conference proceedings.Date of the most recent search: 08 July 2013. Randomised and quasi-randomised controlled studies of vitamin D supplementation compared to placebo in the cystic fibrosis population regardless of exocrine pancreatic function. Both authors independently assessed the risk of bias of each included study and extracted outcome data (from published study information) for assessment of bone mineralization, growth and nutritional status, frequency of vitamin D deficiency, respiratory status, quality of life and adverse events. Six studies (239 participants) are included, although only three studies provided data from 69 adults and children with cystic fibrosis for analysis. One study compared a single high dose of vitamin D (250,000 IU) to placebo at the time of hospital admission with a respiratory exacerbation in 30 pancreatic insufficient adults with cystic fibrosis. The second study compared supplemental 800 international units (IU) vitamin D and placebo for 12 months in 30 osteopenic pancreatic insufficient adults; both groups continued 900 IU vitamin D daily. The third study compared supplemental 1 g calcium alone, 1600 IU vitamin D alone, 1600 IU vitamin D and 1 g calcium and placebo in a double

Laboratory Diagnosis of Echinococcus spp. in Human Patients and Infected Animals.

PubMed

Siles-Lucas, M; Casulli, A; Conraths, F J; Müller, N

2017-01-01

Among the species composing the genus Echinococcus, four species are of human clinical interest. The most prevalent species are Echinococcus granulosus and Echinococcus multilocularis, followed by Echinococcus vogeli and Echinococcus oligarthrus. The first two species cause cystic echinococcosis (CE) and alveolar echinococcosis (AE) respectively. Both diseases have a complex clinical management, in which laboratory diagnosis could be an adjunctive to the imaging techniques. To date, several approaches have been described for the laboratory diagnosis and followup of CE and AE, including antibody, antigen and cytokine detection. All of these approaches are far from being optimal as adjunctive diagnosis particularly for CE, since they do not reach enough sensitivity and/or specificity. A combination of several methods (e.g., antibody and antigen detection) or of several (recombinant) antigens could improve the performance of the adjunctive laboratory methods, although the complexity of echinococcosis and heterogeneity of clinical cases make necessary a deep understanding of the host-parasite relationships and the parasite phenotype at different developmental stages to reach the best diagnostic tool and to make it accepted in clinical practice. Standardization approaches and a deep understanding of the performance of each of the available antigens in the diagnosis of echinococcosis for the different clinical pictures are also needed. The detection of the parasite in definitive hosts is also reviewed in this chapter. Finally, the different methods for the detection of parasite DNA in different analytes and matrices are also reviewed. Copyright © 2017 Elsevier Ltd. All rights reserved.

Expression of cystic fibrosis transmembrane conductance regulator corrects defective chloride channel regulation in cystic fibrosis airway epithelial cells

NASA Astrophysics Data System (ADS)

Rich, Devra P.; Anderson, Matthew P.; Gregory, Richard J.; Cheng, Seng H.; Paul, Sucharita; Jefferson, Douglas M.; McCann, John D.; Klinger, Katherine W.; Smith, Alan E.; Welsh, Michael J.

1990-09-01

The cystic fibrosis transmembrane conductance regulator (CFTR) was expressed in cultured cystic fibrosis airway epithelial cells and Cl- channel activation assessed in single cells using a fluorescence microscopic assay and the patch-clamp technique. Expression of CFTR, but not of a mutant form of CFTR (ΔF508), corrected the Cl- channel defect. Correction of the phenotypic defect demonstrates a causal relationship between mutations in the CFTR gene and defective Cl- transport which is the hallmark of the disease.

Cystic renal cell carcinoma carries an excellent prognosis regardless of tumor size.

PubMed

Winters, Brian R; Gore, John L; Holt, Sarah K; Harper, Jonathan D; Lin, Daniel W; Wright, Jonathan L

2015-12-01

Cystic renal cell carcinoma (cystic RCC) is thought to carry an improved prognosis relative to clear cell RCC (CCRCC); however, this is based on small case series. We used a population-based tumor registry to compare clinicopathologic features and cancer-specific mortality (CSM) of cystic RCC with those of CCRCC. The Surveillance, Epidemiology, and End Results database was queried for all patients diagnosed and treated for cystic RCC and CCRCC between 2001 and 2010. Clinical and pathologic factors were compared using t tests and chi-square tests as appropriate. Kaplan-Meier survival analysis compared CSM differences between cystic RCC and CCRCC. A total of 678 patients with cystic RCC and 46,677 with CCRCC were identified. The mean follow-up duration was 52 and 40 months, respectively. When compared with CCRCC patients, those with cystic RCC were younger (mean age 58 vs. 61 y, P < 0.001), more commonly black (22% vs. 9%, P < 0.001), and female (45% vs. 41%, P = 0.02). Cystic RCCs were more commonly T1a tumors (66% vs. 55%, P < 0.001), well differentiated (33% vs. 16%, P < 0.001), and smaller (mean size = 3.8 vs. 4.5 cm, P < 0.001). Cystic RCC was associated with a reduction in CSM when compared with CCRCC (P = 0.002). In a subset analysis, this reduction in CSM was seen only for those with T1b/T2 tumors (P = 0.01) but not for those with T1a RCCs lesions (P = 0.31). We report the largest series of cystic RCC and corroborate the findings of improved CSM when compared with CCRCC for larger tumors; however, no difference was noted in smaller tumors, suggesting that tumor biology becomes more relevant to prognosis with increasing size. These data may suggest a role for active surveillance in appropriately selected patients with small, cystic renal masses. Copyright © 2015 Elsevier Inc. All rights reserved.

Cystic renal cell carcinoma carries an excellent prognosis regardless of tumor size

PubMed Central

Winters, Brian R.; Gore, John L.; Holt, Sarah K.; Harper, Jonathan D.; Lin, Daniel W.; Wright, Jonathan L.

2016-01-01

Introduction Cystic renal cell carcinoma (cystic RCC) is thought to carry an improved prognosis relative to clear cell RCC (CCRCC); however, this is based on small case series. We used a population-based tumor registry to compare clinicopathologic features and cancer-specific mortality (CSM) of cystic RCC with those of CCRCC. Materials and methods The Surveillance, Epidemiology, and End Results database was queried for all patients diagnosed and treated for cystic RCC and CCRCC between 2001 and 2010. Clinical and pathologic factors were compared using t tests and chi-square tests as appropriate. Kaplan-Meier survival analysis compared CSM differences between cystic RCC and CCRCC. Results A total of 678 patients with cystic RCC and 46,677 with CCRCC were identified. The mean follow-up duration was 52 and 40 months, respectively. When compared with CCRCC patients, those with cystic RCC were younger (mean age 58 vs. 61 y, P < 0.001), more commonly black (22% vs. 9%, P < 0.001), and female (45% vs. 41%, P = 0.02). Cystic RCCs were more commonly T1a tumors (66% vs. 55%, P < 0.001), well differentiated (33% vs. 16%, P < 0.001), and smaller (mean size 3.8 vs. 4.5 cm, P < 0.001). Cystic RCC was associated with a reduction in CSM when compared with CCRCC (P = 0.002). In a subset analysis, this reduction in CSM was seen only for those with T1b/T2 tumors (P = 0.01) but not for those with T1a RCCs lesions (P = 0.31). Conclusions We report the largest series of cystic RCC and corroborate the findings of improved CSM when compared with CCRCC for larger tumors; however, no difference was noted in smaller tumors, suggesting that tumor biology becomes more relevant to prognosis with increasing size. These data may suggest a role for active surveillance in appropriately selected patients with small, cystic renal masses. PMID:26319351

Nutrient Status of Adults with Cystic Fibrosis

PubMed Central

GORDON, CATHERINE M.; ANDERSON, ELLEN J.; HERLYN, KAREN; HUBBARD, JANE L.; PIZZO, ANGELA; GELBARD, RONDI; LAPEY, ALLEN; MERKEL, PETER A.

2011-01-01

Nutrition is thought to influence disease status in patients with cystic fibrosis (CF). This cross-sectional study sought to evaluate nutrient intake and anthropometric data from 64 adult outpatients with cystic fibrosis. Nutrient intake from food and supplements was compared with the Dietary Reference Intakes for 16 nutrients and outcomes influenced by nutritional status. Attention was given to vitamin D and calcium given potential skeletal implications due to cystic fibrosis. Measurements included weight, height, body composition, pulmonary function, and serum metabolic parameters. Participants were interviewed about dietary intake, supplement use, pulmonary function, sunlight exposure, and pain. The participants’ mean body mass index (±standard deviation) was 21.8±4.9 and pulmonary function tests were normal. Seventy-eight percent used pancreatic enzyme replacement for malabsorption. Vitamin D deficiency [25-hydroxyvitamin D (25OHD)<37.5 nmol/L] was common: 25 (39%) were deficient despite adequate vitamin D intake. Lipid profiles were normal in the majority, even though total and saturated fat consumption represented 33.0% and 16.8% of energy intake, respectively. Reported protein intake represented 16.9% of total energy intake (range 10%–25%). For several nutrients, including vitamin D and calcium, intake from food and supplements in many participants exceeded recommended Tolerable Upper Intake Levels. Among adults with cystic fibrosis, vitamin D deficiency was common despite reported adequate intake, and lipid profiles were normal despite a relatively high fat intake. Mean protein consumption was adequate, but the range of intake was concerning, as both inadequate or excessive intake may have deleterious skeletal effects. These findings call into question the applicability of established nutrient thresholds for patients with cystic fibrosis. PMID:18060897

Mucocele of the cystic duct remnant after orthotopic liver transplant: a problem revisited.

PubMed

Chatterjee, Suvadip; Das, Debasish; Hudson, Mark; Bassendine, Margaret Fiona; Scott, John; Oppong, Kofi Ernest; Sen, Gourab; French, Jeremy J

2011-06-01

Mucocele of the cystic duct remnant is an uncommon hepatobiliary complication of a liver transplant. Current practice usually involves either excising the cystic duct, or incorporating the distal end of the transected cystic duct into the suture line of the biliary anastomosis to ensure drainage. We report a patient who developed cystic duct remnant mucocele after the latter approach was adopted. We believe that this is likely related to delayed anastomotic stricturing, which prevented draining from the remnant cystic duct. We also discuss the incidence, pathology, investigations, and treatment of this condition.

Adenoid cystic carcinoma: An unusual presentation.

PubMed

Pushpanjali, M; Sujata, D Naga; Subramanyam, S Bala; Jyothsna, M

2014-05-01

The adenoid cystic carcinoma is a relatively rare epithelial tumor of the major and minor salivary glands, accounting for about 1% of all malignant tumor of the oral and maxillofacial regions. Peak incidence occurs between the 5(th) and 6(th) decades of life. The clinical and pathological findings typical of this tumor include slow growth, peri-neural invasion, multiple local recurrences and distant metastasis. Herein, we report a case of adenoid cystic carcinoma of oropharynx with unusual clinical presentation. The diagnosis of this case and importance of cytology in diagnosing such cases is discussed.

Episodic seasonal Pseudo-Bartter syndrome in cystic fibrosis.

PubMed

Kintu, Brett; Brightwell, Alex

2014-06-01

Pseudo-Bartter syndrome (PBS) describes an uncommon but well recognised complication of cystic fibrosis leading to hypochloraemic, hypokalaemic metabolic alkalosis. Pseudo-Bartter syndrome is usually seen at initial presentation or within the first two years of life in children with cystic fibrosis. Risk factors for development of PBS include warm weather conditions, severe respiratory or pancreatic disease and gastrointestinal losses (e.g. vomiting and diarrhoea). PBS is rare in older children and adolescents although epidemics have been associated with heat wave conditions in warmer climates. In this era of climate change, it is crucial that clinicians consider Pseudo-Bartter syndrome when patients with cystic fibrosis present unwell during summer. Copyright © 2014 Elsevier Ltd. All rights reserved.

Cystic echinococcosis in a fox-hound hunt worker, UK

PubMed Central

Craig, Philip S; Woods, Marion L; Boufana, Belgees; O’Loughlin, Barry; Gimpel, John; San Lett, Wai; McManus, Donald P

2012-01-01

A male resident in Vanuatu with prior history of employment as a hunt kennel-man in England (1980–2001) was surgically treated for the removal of a hydatid cyst subsequently confirmed as Echinococcus granulosus (G1 genotype). This is the first human molecularly identified CE case reported from the UK and a first in a fox-hound handler and indicates a general neglected occupational risk. PMID:23182144

Report of the European Respiratory Society/European Cystic Fibrosis Society task force on the care of adults with cystic fibrosis.

PubMed

Elborn, J Stuart; Bell, Scott C; Madge, Susan L; Burgel, Pierre-Regis; Castellani, Carlo; Conway, Steven; De Rijcke, Karleen; Dembski, Birgit; Drevinek, Pavel; Heijerman, Harry G M; Innes, J Alistair; Lindblad, Anders; Marshall, Bruce; Olesen, Hanne V; Reimann, Andreas L; Solé, Ampara; Viviani, Laura; Wagner, Thomas O F; Welte, Tobias; Blasi, Francesco

2016-02-01

The improved survival in people with cystic fibrosis has led to an increasing number of patients reaching adulthood. This trend is likely to be maintained over the next decades, suggesting a need to increase the number of centres with expertise in the management of adult patients with cystic fibrosis. These centres should be capable of delivering multidisciplinary care addressing the complexity of the disease, in addition to addressing the psychological burden on patients and their families. Further issues that require attention are organ transplantation and end of life management.Lung disease in adults with cystic fibrosis drives most of the clinical care requirements, and major life-threatening complications, such as respiratory infection, respiratory failure, pneumothorax and haemoptysis, and the management of lung transplantation require expertise from trained respiratory physicians. The taskforce therefore strongly reccommends that medical leadership in multidisciplinary adult teams should be attributed to a respiratory physician adequately trained in cystic fibrosis management.The task force suggests the implementation of a core curriculum for trainees in adult respiratory medicine and the selection and accreditation of training centres that deliver postgraduate training to the standards of the HERMES programme. Copyright ©ERS 2016.

Lung ultrasound has limited diagnostic value in rare cystic lung diseases: a cross-sectional study

PubMed Central

Davidsen, Jesper Rømhild; Bendstrup, Elisabeth; Henriksen, Daniel P.; Graumann, Ole; Laursen, Christian B.

2017-01-01

ABSTRACT Background: Lung ultrasound (LUS) used to identify interstitial syndrome (IS) and pleural thickening related to diffuse parenchymal lung disease (DPLD) has shown significant correlations with ground glass opacity (GGO) on high-resolution computed tomography (HRCT). However, the applicability of LUS in patients with DPLD subtypes as rare cystic lung diseases has not previously been investigated. This study aimed to observe if distinctive LUS findings could be found in patients with lymphangioleiomyomatosis (LAM), pulmonary Langerhans cell histiocytosis (PLCH), and Birt-Hogg-Dubé syndrome (BHDS). Methods: This single centre case-based cross-sectional study of patients diagnosed with LAM, PCLH and BHDS was conducted at a Danish DPLD specialist centre. Patients underwent clinical examination including LUS. LUS findings were compared to findings scored according to a modified Belmaati score on HRCT and reviewed in consensus between two pulmonologists and one radiologist. Results: Twelve patients with HRCT proven cystic lung disease were included, six with LAM, three with PLCH, two with BHDS, and one with uncharacteristic cystic lung disease. The mean age was 48.7 years (SD ± 15.8). In general all had normal LUS findings. IS could not be found in any patients despite GGO presentation on HRCT among 75% of the patients with a Belmaati in the highest category of 0.76–1.00. Pleural thickening on LUS was present in three patients, but with inconsistent findings. Conclusion: This study indicates that LUS has limited value as a diagnostic tool in patients with LAM, PLCH, and BHDS as normal LUS findings did not rule out severe cystic lung disease. PMID:28649310

Classification of malnutrition in cystic fibrosis: implications for evaluating and benchmarking clinical practice performance2

PubMed Central

HuiChuan, J Lai; Suzanne, M Shoff

2008-01-01

Background In 2005, the Cystic Fibrosis Foundation (CFF) revised the nutrition classification guidelines to eliminate the use of percentage of ideal body weight (%IBW) to define “nutritional failure”; the CFF also recommended that children with cystic fibrosis maintain a body mass index percentile (BMIp) ≥ 50th. Objective We assessed the effect of the 2005 CFF nutrition classification guidelines on evaluating the performance of nutritional care practices. Design Data from 14 702 children reported to the 2002 CFF Patient Registry were analyzed to compare malnutrition rates in 113 cystic fibrosis centers in the United States. Nutritional failure was defined according to the 2002 CFF criteria—ie, height < 5th percentile, %IBW < 90%, or BMIp < 10th. “Below BMI goal” was defined according to the 2005 CFF criterion, ie BMIp < 50th. Results Eliminating %IBW resulted in a 6% reduction (from 33% to 27%) in the nutritional failure rate in the United States. The use of BMIp < 50th led to the classification of 57% of children as below the BMI goal. Misclassification of nutritional failure according to %IBW ranged from 1% to 16% among 113 centers and was greater in the centers with a larger proportion of tall patients. After the elimination of %IBW, one-third of centers changed to a different tertile ranking for nutritional failure rates (kappa = 0.50, moderate-to-poor agreement). More than half the centers changed to a different tertile ranking, from nutritional failure to below BMI goal (kappa = 0.22, poor agreement). Conclusion Eliminating misclassification by %IBW and implementing the new BMI goal led to profound and unequal changes in malnutrition rates across cystic fibrosis centers. PMID:18614737

Renal diseases in adults with cystic fibrosis: a 40 year single centre experience.

PubMed

Wilcock, M J; Ruddick, A; Gyi, K M; Hodson, M E

2015-10-01

There is a sizable literature describing renal disease in patients with cystic fibrosis. Previous studies have focused on single disease processes alone, most commonly renal stone disease or acute kidney injury. In this study we report for the first time on the prevalence of all forms of renal disease in a cystic fibrosis population. A retrospective review of adult patients with cystic fibrosis attending the Adult Cystic Fibrosis Department at the Royal Brompton Hospital was carried out by searching the department's database to identify patients with renal problems and subsequently retrieving clinical information from medical notes. The prevalence of all renal diseases in our population was 5.1 %. The most commonly identified problem was renal stones. At 2.0 % the prevalence of renal stones in adult patients with cystic fibrosis was comparable to the general population. A range of other renal diseases were identified, the next most common being drug-induced acute kidney injury. A range of cystic fibrosis independent and attributable diseases has been identified but no cystic fibrosis specific disease. In contrast to other cystic fibrosis centres no increased prevalence of renal stones was found.

[The problem of residual cavity and local recurrences in surgical treatment of hepatic echinococcosis].

PubMed

Chetverikov, S G; Akhmad, Z M

2014-06-01

Results of examination and treatment of 326 patients, suffering hepatic echinococcosis, in whom 449 surgical interventions were performed, were analyzed. In 123 patients the disease recurrence was diagnosed. In 80 (17.8%) patients laparoscopic access was applied, in 261 (58.1%)--laparotomic, in 108 (24.1%)--the puncture treatment of parasitic cysts under ultrasonographic investigation control. There was established, that the residual cavity walls after echinococcectomy, if the condition of their chemical and thermal processing do not constitute a danger for the disease recurrence occurrence, but the residual cavity suppuration or formation of durably persisting residual cavities in liver is possible. So the optimal operative intervention must be echinococcectomy with partial resection of fibrose capsule with the thinned hepatic parenchyma.

[Historical compilation of cystic fibrosis].

PubMed

Navarro, Salvador

2016-01-01

Cystic fibrosis is the most common life-shortening recessively inherited disorder in the Caucasian population. The genetic mutation that most frequently provokes cystic fibrosis (ΔF508) appeared at least 53,000years ago. For many centuries, the disease was thought to be related to witchcraft and the "evil eye" and it was only in 1938 that Dorothy H. Andersen characterized this disorder and suspected its genetic origin. The present article reviews the pathological discoveries and diagnostic and therapeutic advances made in the last 75 years. The review ends with some considerations for the future. Copyright © 2015 Elsevier España, S.L.U. and AEEH y AEG. All rights reserved.

Caring for Children with Cystic Fibrosis: A Collaborative Clinical and School Approach

ERIC Educational Resources Information Center

Strawhacker, MaryAnn Tapper; Wellendorf, Joyce

2004-01-01

Earlier diagnosis and more effective treatments have improved both morbidity and mortality associated with cystic fibrosis, making regular school attendance a reality. School nurses have a unique opportunity to assist students with cystic fibrosis successfully manage their disease. Medical treatment for cystic fibrosis can be complex, leaving…

Cystic fibrosis respiratory tract salt concentration: An Exploratory Cohort Study.

PubMed

Grandjean Lapierre, Simon; Phelippeau, Michael; Hakimi, Cyrine; Didier, Quentin; Reynaud-Gaubert, Martine; Dubus, Jean-Christophe; Drancourt, Michel

2017-11-01

In cystic fibrosis patients, electrolytic and osmolality imbalance secondary to cystic fibrosis transmembrane conductance regulator mutations may impact on mucoid secretion accumulation and secondary colonization by opportunistic pathogens such as nontuberculous mycobacteria.We performed a noninvasive exploratory prospective controlled clinical study comparing sputum salinity and acid-base characteristics of cystic fibrosis and noncystic fibrosis control patients. A total of 57 patients and 62 controls were included.Sputum salt concentrations were 10.5 g/L (95% CI: 7.7-13.3) in patients and 7.4 g/L (95% CI: 5.9-8.9) in aged-matched controls, a difference that was found to be statistically significant (P < .05). No difference in pH was observed between patients and controls.These differences in respiratory secretions salt concentrations could influence host-pathogen interactions in the context of cystic fibrosis respiratory infections. We propose to include respiratory secretion salt measurement as a routine analysis on cystic fibrosis patients' sputum submitted for bacterial culture. Copyright © 2017 The Authors. Published by Wolters Kluwer Health, Inc. All rights reserved.

Intrinsic pro-angiogenic status of cystic fibrosis airway epithelial cells

DOE Office of Scientific and Technical Information (OSTI.GOV)

Verhaeghe, Catherine; Tabruyn, Sebastien P.; Oury, Cecile

Cystic fibrosis is a common genetic disorder characterized by a severe lung inflammation and fibrosis leading to the patient's death. Enhanced angiogenesis in cystic fibrosis (CF) tissue has been suggested, probably caused by the process of inflammation, as similarly described in asthma and chronic bronchitis. The present study demonstrates an intrinsic pro-angiogenic status of cystic fibrosis airway epithelial cells. Microarray experiments showed that CF airway epithelial cells expressed several angiogenic factors such as VEGF-A, VEGF-C, bFGF, and PLGF at higher levels than control cells. These data were confirmed by real-time quantitative PCR and, at the protein level, by ELISA. Conditionedmore » media of these cystic fibrosis cells were able to induce proliferation, migration and sprouting of cultured primary endothelial cells. This report describes for the first time that cystic fibrosis epithelial cells have an intrinsic angiogenic activity. Since excess of angiogenesis is correlated with more severe pulmonary disease, our results could lead to the development of new therapeutic applications.« less

Cystic adventitial degeneration: ectopic ganglia from adjacent joint capsules.

PubMed

Ortmann, J; Widmer, M K; Gretener, S; Do, D D; Willenberg, T; Daliri, A; Baumgartner, I

2009-11-01

Cystic adventitial degeneration is a rare non-atherosclerotic cause of peripheral arterial occlusive disease, mainly seen in young men without other evidence of vascular disease. Diagnosis will be established by clinical findings and by ultrasound or angiography and can be treated by excision or enucleation of the affected arterial segment or by percutaneous ultrasound-guided aspiration. However, the etiology of adventitial cysts remains unknown. We report a case of cystic adventitial degeneration showing a connection between the joint capsule and the adventitial cyst, supporting the theory that cystic adventitial degeneration may represent ectopic ganglia from adjacent joint capsules.

Predicting the intrauterine fetal death of fetuses with cystic hygroma in early pregnancy.

PubMed

Shimura, Mai; Ishikawa, Hiroshi; Nagase, Hiromi; Mochizuki, Akihiko; Sekiguchi, Futoshi; Koshimizu, Naho; Itai, Toshiyuki; Odagami, Mizuha

2018-01-11

We investigated whether it was possible to predict the prognosis of fetuses with cystic hygroma in early pregnancy based on the degree of neck thickening. We retrospectively analyzed 57 singleton pregnancies with fetuses with cystic hygroma who were examined before the 22nd week of pregnancy. The fetuses were categorized according to the outcome, structural abnormalities at birth, and chromosomal abnormalities. Here, we proposed a new sonographic predictor with which we assessed neck thickening by dividing the width of the neck thickening by the biparietal diameter, which is expressed as the cystic hygroma width/biparietal diameter ratio. The median cystic hygroma width/biparietal diameter ratio in the intrauterine fetal death group (0.51) was significantly higher than that in the live birth group (0.27). No significant difference in the median cystic hygroma width/biparietal diameter ratio was found between the structural abnormalities group at birth and the no structural abnormalities group, and no significant difference in the median cystic hygroma width/biparietal diameter ratio was found between the chromosomal abnormality group and the no chromosomal abnormality group. We used receiver operating characteristic analysis to evaluate the cystic hygroma width/biparietal diameter ratio to predict intrauterine fetal death. When the cystic hygroma width/biparietal diameter ratio cut-off value was 0.5, intrauterine fetal death could be predicted with a sensitivity of 52.9% and a specificity of 100%. It is possible to predict intrauterine fetal death in fetuses with cystic hygroma in early pregnancy if cystic hygroma width/biparietal diameter ratio is measured. However, even if cystic hygroma width/biparietal diameter ratio is measured, predicting the presence or absence of a structural abnormality at birth or a chromosomal abnormality is difficult. © 2018 Japanese Teratology Society.

Diagnostic problems in cystic fibrosis - specific characteristics of a group of infants and young children diagnosed positive through neonatal screening, in whom cystic fibrosis had not been diagnosed.

PubMed

Woś, Halina; Sankiewicz-Szkółka, Magda; Więcek, Sabina; Kordys-Darmolińska, Bożena; Grzybowska-Chlebowczyk, Urszula; Kniażewska, Maria

2015-01-01

Neonatal cystic fibrosis screening contributes to an early diagnosis of cystic fibrosis and to implementing appropriate therapeutic management. Long-standing screening tests have made it possible to identify a group of newborns in whom the diagnosis was ambiguous and required further specialised tests. The aim is to present cases of patients with a positive result of newborn screening for cystic fibrosis who were found to be carriers of the mutation in both alleles, however the lack of clinical symptoms and correct sweat testing values did not lead doctors to diagnosing cystic fibrosis and by the same token implementing the treatment. The analysis encompassed a group of 22 infants and children 3 months to 3 years of age, in whom, in spite of a positive result of newborn screening for cystic fibrosis and the presence of 2 mutations in the CFTR gene, the diagnosis of cystic fibrosis was not made, and appropriate treatment was not administered because of diagnostic doubts (due to correct concentration of chlorides in sweat, correct IRT level and lack of clinical signs of cystic fibrosis). The control group consisted of 55 children treated in our centre, in whom neonatal screening for cystic fibrosis was positive and the diagnosis was confirmed by genetic testing, sweat chloride testing and IRT concentration. There were no differences in birth body weight between the groups. The differences in chlorideion levels in sweat secretion tests and mean IRT values were statistically significant and were: 97.5 for the control group and 26.4 for the test group. At the present time there are no clinical symptoms to give a diagnosis of cystic fibrosis and start treatment in the test group. Newborn screening contributes not only to an early diagnosis of cystic fibrosis but also to CFTR-related metabolic syndromes (CRMS), which is a phenomenon requiring further observation. This fact constitutes a definite psychological problem for the parents of these patients. .

Hormonal abnormalities of the pancreas and gut in cystic fibrosis.

PubMed

Adrian, T E; McKiernan, J; Johnstone, D I; Hiller, E J; Vyas, H; Sarson, D L; Bloom, S R

1980-09-01

We have investigated the effect of cystic fibrosis on alimentary hormones in 10 children by measuring the pancreatic and gut hormone rsponse to a milk drink. Plasma insulin and gastric inhibitory peptide were both significantly reduced (P < 0.05 and P < 0.005, respectively, at 15 min) in the patients with cystic fibrosis, compared with controls, even though the early glucose rise was greater in the former group (P < 0.05 at 15 min). Fasting levels of pancreatic polypeptide were significantly lower in the fibrocystic children (P < 0.01), and the normal response to milk was completely abolished in these patients (P < 0.001). Fasting plasma enteroglucagon concentrations were grossly abolished in the cystic fibrosis patients (P < 0.001) and these remained elevated throughout the test. No significant differences were seen in basal or postmilk responses of plasma glucagon, gastrin, secretin, vasoactive intestinal peptide, or motilin in cystic fibrosis. It would thus appear that the pancreatic polypeptide cell is more susceptible to the effects of the disease process than the beta or alpha cell in cystic fibrosis. Some aspects of the abnormalities in the gastrointestinal endocrine system were similar to those seen in celiac disease and tropical sprue and may, therefore, effect a similar hormonal response in these patients with cystic fibrosis to those with mucosal damage.

Radiological Diagnoses in the Context of Emigration: Infectious diseases.

PubMed

Stojkovic, Marija; Müller, Jan; Junghanss, Thomas; Weber, Tim Frederik

2018-02-01

 Globalization and emigration impact on the spectrum of diseases challenging health care systems. Medical practitioners have to particularly prepare for infectious diseases.  The database of a health care center specialized on tropical medicine was screened for patients with history of migration and one of the following diagnoses: Cystic echinococcosis, tuberculosis, schistosomiasis, visceral leishmaniosis, and neurocysticercosis. Representative casuistics were prepared from select case histories. Radiological pertinent knowledge was compiled based on literature search.  A small selection of frequently imported infectious diseases covers a considerable fraction of health care problems associated with migration. For cystic echinococcosis, schistosomiasis, and neurocysticercosis imaging is the most relevant diagnostic procedure defining also disease stages. Tuberculosis and visceral leishmaniosis are important differentials for malignant diseases.  Imaging plays a meaningful role in diagnosis, treatment stratification, and follow-up of imported infectious diseases. Radiological skills concerning these diseases are important for providing health care for patients in context of migration.   · Imaging plays a meaningful role in multidisciplinary care for imported infectious diseases.. · A small selection covers a considerable fraction of infectious diseases expected in context of migration.. · Stojkovic M, Müller J, Junghanss T et al. Radiological Diagnoses in the Context of Emigration: Infectious diseases. Fortschr Röntgenstr 2018; 190: 121 - 133. © Georg Thieme Verlag KG Stuttgart · New York.

In Vitro and In Vivo Effects of Tamoxifen against Larval Stage Echinococcus granulosus

PubMed Central

Nicolao, María Celeste; Elissondo, María Celina; Denegri, Guillermo M.; Goya, Alejandra B.

2014-01-01

Cystic echinococcosis is a zoonotic infection caused by the larval stage of the cestode Echinococcus granulosus. Chemotherapy currently employs benzimidazoles; however, 40% of cases do not respond favorably. With regard to these difficulties, novel therapeutic tools are needed to optimize treatment in humans. The aim of this work was to explore the in vitro and in vivo effects of tamoxifen (TAM) against E. granulosus. In addition, possible mechanisms for the susceptibility of TAM are discussed in relation to calcium homeostasis, P-glycoprotein inhibition, and antagonist effects on a putative steroid receptor. After 24 h of treatment, TAM, at a low micromolar concentration range (10 to 50 μM), inhibited the survival of E. granulosus protoscoleces and metacestodes. Moreover, we demonstrated the chemotherapeutic and chemopreventive pharmacological effects of the drug. At a dose rate of 20 mg/kg of body weight, TAM induced protection against the infection in mice. In the clinical efficacy studies, a reduction in cyst weight was observed after the administration of 20 mg/kg in mice with cysts developed during 3 or 6 months, compared to that of those collected from control mice. Since the collateral effects of high TAM doses have been largely documented in clinical trials, the use of low doses of this drug as a short-term therapy may be a novel alternative approach for human cystic echinococcosis treatment. PMID:24936598

"Cystic fibrotics could survive cholera, choleraics could survive cystic fibrosis"; hypothesis that explores new horizons in treatment of cystic fibrosis.

PubMed

Azimi, Arsalan

2015-12-01

Cystic fibrosis, the most common inherited disease of white population, is a disease of CFTR channels, in which mucosal function of many organs especially respiratory tract is impaired. Decreased mucociliary clearance and accumulation of mucus in airways facilitates colonization of infectious microorganisms, followed by infection. Following chronic infection, persistent inflammation ensues, which results in airway remodeling and deterioration of mucociliary clearance and result in a vicious cycle. Here, it is hypothesized that cholera toxin (CT) could ameliorate symptoms of cystic fibrosis as CT could dilute the thickened mucus, improve mucociliary clearance and alleviate airway obstruction. CT strengthens immunity of airway mucosa and it could attenuates bacterial growth and reduce persistency of infection. CT also modulates cellular immune response and it could decrease airway inflammation, hinder airway remodeling and prevent respiratory deterioration. Thereby it is hypothesized that CT could target and ameliorate many of pathophysiologic steps of the disease and it explores new horizons in treatment of CF. Copyright © 2015 Elsevier Ltd. All rights reserved.

68Ga-PSMA PET-CT Imaging of Metastatic Adenoid Cystic Carcinoma.

PubMed

de Keizer, Bart; Krijger, Gerard C; Ververs, F Tessa; van Es, Robert J J; de Bree, Remco; Willems, Stefan

2017-12-01

A patient with a history of adenoid cystic carcinoma of the nasal cavity presented himself with bone pain and an elevated PSA level. On suspicion of metastatic prostate cancer a 68 Ga-PSMA PET-CT was performed. The PET-CT showed numerous lung and non-sclerotic bone metastasis. Biopsy of a bone metastasis was performed and pathology showed adenoid cystic carcinoma instead of prostate cancer. Immunohistochemical PSMA staining of the primary tumour showed intense PSMA expression in adenoid cystic carcinoma tumour cells. Because of the high PSMA expression of adenoid cystic carcinoma, 68 Ga-PSMA PET-CT might be a promising imaging modality for this malignancy.

Gamma Knife Radiosurgery for the Treatment of Cystic Cerebral Metastases

DOE Office of Scientific and Technical Information (OSTI.GOV)

Ebinu, Julius O.; Lwu, Shelly; Monsalves, Eric

Purpose: To assess the role of Gamma Knife radiosurgery (GKRS) in the treatment of nonsurgical cystic brain metastasis, and to determine predictors of response to GKRS. Methods: We reviewed a prospectively maintained database of brain metastases patients treated at our institution between 2006 and 2010. All lesions with a cystic component were identified, and volumetric analysis was done to measure percentage of cystic volume on day of treatment and consecutive follow-up MRI scans. Clinical, radiologic, and dosimetry parameters were reviewed to establish the overall response of cystic metastases to GKRS as well as identify potential predictive factors of response. Results:more » A total of 111 lesions in 73 patients were analyzed; 57% of lesions received prior whole-brain radiation therapy (WBRT). Lung carcinoma was the primary cancer in 51% of patients, 10% breast, 10% colorectal, 4% melanoma, and 26% other. Fifty-seven percent of the patients were recursive partitioning analysis class 1, the remainder class 2. Mean target volume was 3.3 mL (range, 0.1-23 mL). Median prescription dose was 21 Gy (range, 15-24 Gy). Local control rates were 91%, 63%, and 37% at 6, 12, and 18 months, respectively. Local control was improved in lung primary and worse in patients with prior WBRT (univariate). Only lung primary predicted local control in multivariate analysis, whereas age and tumor volume did not. Lesions with a large cystic component did not show a poorer response compared with those with a small cystic component. Conclusions: This study supports the use of GKRS in the management of nonsurgical cystic metastases, despite a traditionally perceived poorer response. Our local control rates are comparable to a matched cohort of noncystic brain metastases, and therefore the presence of a large cystic component should not deter the use of GKRS. Predictors of response included tumor subtype. Prior WBRT decreased effectiveness of SRS for local control rates.« less

Breakthrough Therapies: Cystic Fibrosis (CF) Potentiators and Correctors

PubMed Central

Solomon, George M.; Marshall, Susan G.; Ramsey, Bonnie W.; Rowe, Steven M.

2015-01-01

Cystic Fibrosis is caused by mutations in the Cystic Fibrosis Transmembrane conductance Regulator (CFTR) gene resulting in abnormal protein function. Recent advances of targeted molecular therapies and high throughput screening have resulted in multiple drug therapies that target many important mutations in the CFTR protein. In this review, we provide the latest results and current progress of CFTR modulators for the treatment of cystic fibrosis, focusing on potentiators of CFTR channel gating and Phe508del processing correctors for the Phe508del CFTR mutation. Special emphasis is placed on the molecular basis underlying these new therapies and emerging results from the latest clinical trials. The future directions for augmenting the rescue of Phe508del with CFTR modulators is also emphasized. PMID:26097168

Benign cystic lesions of the vagina: a literature review.

PubMed

Eilber, Karyn Schlunt; Raz, Shlomo

2003-09-01

Knowledge of the etiology, evaluation and treatment of cystic lesions of the vagina is essential as these lesions are often encountered in urological and gynecological practices. We searched MEDLINE and MeSH for literature from the last 50 years referring to cysts of the vagina. Review of the literature regarding etiology, clinical and pathological diagnosis, prognosis and treatment identified 18 journal articles and 6 books. Benign cystic lesions of the vagina present a spectrum, from small asymptomatic lesions to cysts large enough to cause urinary obstruction. History, physical examination and radiological imaging, including voiding cystourethrogram and magnetic resonance imaging, are useful in diagnosis. Treatment is determined by the severity of symptoms. Cystic lesions of the vagina are relatively common and usually represent benign conditions. A vaginal cyst may be an embryological derivative, ectopic tissue or urological abnormality. Awareness of the various diagnoses of benign cystic lesions of the vagina and associated abnormalities will aid in evaluation and treatment.

Cystic Benign Metastasizing Leiomyoma of the Lung Mimicking Hydatid Cyst.

PubMed

Alimi, Faouzi; El Hadj Sidi, Chighali; Ghannouchi, Chams

2016-12-01

A 60-year-old woman, with history of a resected uterine benign leiomyoma, is operated for a cystic lesion in the left lung, mimicking an hydatid cyst, and confirmed histologically as Cystic Benign Metastasizing Leiomyoma.

Recent progress in translational cystic fibrosis research using precision medicine strategies.

PubMed

Cholon, Deborah M; Gentzsch, Martina

2018-03-01

Significant progress has been achieved in developing precision therapies for cystic fibrosis; however, highly effective treatments that target the ion channel, CFTR, are not yet available for many patients. As numerous CFTR therapeutics are currently in the clinical pipeline, reliable screening tools capable of predicting drug efficacy to support individualized treatment plans and translational research are essential. The utilization of bronchial, nasal, and rectal tissues from individual cystic fibrosis patients for drug testing using in vitro assays such as electrophysiological measurements of CFTR activity and evaluation of fluid movement in spheroid cultures, has advanced the prediction of patient-specific responses. However, for precise prediction of drug effects, in vitro models of CFTR rescue should incorporate the inflamed cystic fibrosis airway environment and mimic the complex tissue structures of airway epithelia. Furthermore, novel assays that monitor other aspects of successful CFTR rescue such as restoration of mucus characteristics, which is important for predicting mucociliary clearance, will allow for better prognoses of successful therapies in vivo. Additional cystic fibrosis treatment strategies are being intensively explored, such as development of drugs that target other ion channels, and novel technologies including pluripotent stem cells, gene therapy, and gene editing. The multiple therapeutic approaches available to treat the basic defect in cystic fibrosis combined with relevant precision medicine models provide a framework for identifying optimal and sustained treatments that will benefit all cystic fibrosis patients. Copyright © 2017 European Cystic Fibrosis Society. Published by Elsevier B.V. All rights reserved.

Adenoid cystic carcinoma: An unusual presentation

PubMed Central

Pushpanjali, M; Sujata, D Naga; Subramanyam, S Bala; Jyothsna, M

2014-01-01

The adenoid cystic carcinoma is a relatively rare epithelial tumor of the major and minor salivary glands, accounting for about 1% of all malignant tumor of the oral and maxillofacial regions. Peak incidence occurs between the 5th and 6th decades of life. The clinical and pathological findings typical of this tumor include slow growth, peri-neural invasion, multiple local recurrences and distant metastasis. Herein, we report a case of adenoid cystic carcinoma of oropharynx with unusual clinical presentation. The diagnosis of this case and importance of cytology in diagnosing such cases is discussed. PMID:25328314

Cystic tumors of the liver: A practical approach

PubMed Central

Poggio, Paolo Del; Buonocore, Marco

2008-01-01

Biliary cyst tumors (cystadenoma and cystadeno-carcinoma) are an indication for liver resection. They account for only 5% of all solitary cystic lesions of the liver, but differential diagnosis with multiloculated or complicated biliary cysts, atypical hemangiomas, hamartomas and lymphangiomas may be difficult. The most frequent challenge is to differentiate biliary cyst tumors from hemorrhagic cysts. Computerized tomography (CT) and magnetic resonance imaging (MRI) are often not diagnostic and in these cases fine needle aspiration (FNA) is used to confirm the presence of atypical biliary cells. FNA, however, lacks adequate sensitivity and specificity and should always be used in conjunction with imaging. Pre-operative differentiation of cystadenoma from cystadenocarcinoma is impossible and surgery must be performed if a biliary cyst tumor is suspected. When multiple cystic lesions are observed throughout the liver parenchyma, it is important to exclude liver metastasis, of which colonic cancer is the most common primary site. Multiple biliary hamartomas (von Meyenburg complex) can appear as a mixture of solid and cystic lesions and can be confused with cystic metastasis. Strong and uniform T2 hyperintensity on MRI is usually diagnostic, but occasionally a percutaneous biopsy may be required. PMID:18595127

Predictive 5-Year Survivorship Model of Cystic Fibrosis

PubMed Central

Liou, Theodore G.; Adler, Frederick R.; FitzSimmons, Stacey C.; Cahill, Barbara C.; Hibbs, Jonathan R.; Marshall, Bruce C.

2007-01-01

The objective of this study was to create a 5-year survivorship model to identify key clinical features of cystic fibrosis. Such a model could help researchers and clinicians to evaluate therapies, improve the design of prospective studies, monitor practice patterns, counsel individual patients, and determine the best candidates for lung transplantation. The authors used information from the Cystic Fibrosis Foundation Patient Registry (CFFPR), which has collected longitudinal data on approximately 90% of cystic fibrosis patients diagnosed in the United States since 1986. They developed multivariate logistic regression models by using data on 5,820 patients randomly selected from 11,630 in the CFFPR in 1993. Models were tested for goodness of fit and were validated for the remaining 5,810 patients for 1993. The validated 5-year survivorship model included age, forced expiratory volume in 1 second as a percentage of predicted normal, gender, weight-for-age z score, pancreatic sufficiency, diabetes mellitus, Staphylococcus aureus infection, Burkerholderia cepacia infection, and annual number of acute pulmonary exacerbations. The model provides insights into the complex nature of cystic fibrosis and supplies a rigorous tool for clinical practice and research. PMID:11207152

New and emerging targeted therapies for cystic fibrosis

PubMed Central

Rowe, Steven M

2016-01-01

Cystic fibrosis (CF) is a monogenic autosomal recessive disorder that affects about 70 000 people worldwide. The clinical manifestations of the disease are caused by defects in the cystic fibrosis transmembrane conductance regulator (CFTR) protein. The discovery of the CFTR gene in 1989 has led to a sophisticated understanding of how thousands of mutations in the CFTR gene affect the structure and function of the CFTR protein. Much progress has been made over the past decade with the development of orally bioavailable small molecule drugs that target defective CFTR proteins caused by specific mutations. Furthermore, there is considerable optimism about the prospect of gene replacement or editing therapies to correct all mutations in cystic fibrosis. The recent approvals of ivacaftor and lumacaftor represent the genesis of a new era of precision medicine in the treatment of this condition. These drugs are having a positive impact on the lives of people with cystic fibrosis and are potentially disease modifying. This review provides an update on advances in our understanding of the structure and function of the CFTR, with a focus on state of the art targeted drugs that are in development. PMID:27030675

What is Cystic Fibrosis?

MedlinePlus

... pass the faulty CFTR gene to their children. Example of an Inheritance Pattern for Cystic Fibrosis The image shows how CFTR genes are inherited. A person inherits two copies of the CFTR gene—one from each parent. If each parent has a ...

Topical Bibliography of Published Works Regarding the Health of Veterans of the Persian Gulf War

DTIC Science & Technology

1995-08-01

strain). Modern Pathology. 1991;6:750-754. Lubani MM, Lulu AR, Araj GF, Khateeb MI, Qurtom MAF, Dudin KI. Pulmonary brucellosis. Q JMed. 1989;71:319-324...Hyg. 1985;88:319-322. Molan AL, Saida LA. Echinococcosis in Iraq: prevalence of Echinococcus granulosus in stray dogs in Arbil Province. Jpn J Med Sci...in the western region of Saudi Arabia. Trans R Soc Trop Med Hyg. 1987;81:219- 221. Shweiki HM, Hira PR, Behbehani K. Cystic hydatid disease: aspects of

Early bronchiectasis in cystic fibrosis detected by surveillance CT.

PubMed

Pillarisetti, Naveen; Linnane, Barry; Ranganathan, Sarath

2010-08-01

There is emerging evidence that cystic fibrosis lung disease begins early in infancy. Newborn screening allows early detection and surveillance of pulmonary disease and the possibility of early intervention in this life-shortening condition. We report two children with cystic fibrosis who underwent a comprehensive assessment from diagnosis that included measurement of lung function, limited-slice high-resolution CT and BAL performed annually. Early aggressive surveillance enabled significant lung disease and bronchiectasis to be detected during the first few years of life and led to a change in management, highlighting a clinical role for CT scanning during the preschool years in children with cystic fibrosis.

Pharmacogenetics of cystic fibrosis treatment.

PubMed

Carter, Suzanne C; McKone, Edward F

2016-08-01

Cystic fibrosis (CF) is genetic autosomal recessive disease caused by reduced or absent function of CFTR protein. Treatments for patients with CF have primarily focused on the downstream end-organ consequences of defective CFTR. Since the discovery of the CFTR gene that causes CF in 1989 there have been tremendous advances in our understanding of the genetics and pathophysiology of CF. This has recently led to the development of new CFTR mutation-specific targeted therapies for select patients with CF. This review will discuss the characteristics of the CFTR gene, the CFTR mutations that cause CF and the new mutation specific pharmacological treatments including gene therapy that are contributing to the dawning of a new era in cystic fibrosis care.

Respiratory infections in patients with cystic fibrosis undergoing lung transplantation.

PubMed

Lobo, Leonard J; Noone, Peadar G

2014-01-01

Cystic fibrosis is an inherited disease characterised by chronic respiratory infections associated with bronchiectasis. Lung transplantation has helped to extend the lives of patients with cystic fibrosis who have advanced lung disease. However, persistent, recurrent, and newly acquired infections can be problematic. Classic cystic fibrosis-associated organisms, such as Staphylococcus aureus and Pseudomonas aeruginosa, are generally manageable post-transplantation, and are associated with favourable outcomes. Burkholderia cenocepacia poses particular challenges, although other Burkholderia species are less problematic. Despite concerns about non-tuberculous mycobacteria, especially Mycobacterium abscessus, post-transplantation survival has not been definitively shown to be less than average in patients with these infections. Fungal species can be prevalent before and after transplantation and are associated with high morbidity, so should be treated aggressively. Appropriate viral screening and antiviral prophylaxis are necessary to prevent infection with and reactivation of Epstein-Barr virus and cytomegalovirus and their associated complications. Awareness of drug pharmacokinetics and interactions in cystic fibrosis is crucial to prevent toxic effects and subtherapeutic or supratherapeutic drug dosing. With the large range of potential infectious organisms in patients with cystic fibrosis, infection control in hospital and outpatient settings is important. Despite its complexity, lung transplantation in the cystic fibrosis population is safe, with good outcomes if the clinician is aware of all the potential pathogens and remains vigilant by means of surveillance and proactive treatment. Copyright © 2014 Elsevier Ltd. All rights reserved.

Circulating cell-free DNA in patients with alveolar echinococcosis.

PubMed

Baraquin, Alice; Hervouet, Eric; Richou, Carine; Flori, Pierre; Peixoto, Paul; Azizi, Amel; Delabrousse, Eric; Blagosklonov, Oleg; Umhang, Gerald; Bresson-Hadni, Solange; Valot, Benoit; Grenouillet, Frédéric

2018-06-01

Alveolar echinococcosis (AE) is a parasitic disease, due to Echinococcus multilocularis. Often compared to liver cancer, it develops by infiltration from its primary site to the surrounding tissue, and can then metastasize to other organs. Detection of circulating cell-free DNA (ccfDNA) is a useful analytical tool in oncology, for diagnosis, prognosis, and therapy monitoring. This study sought to investigate the presence of ccfDNA in patients with AE, and its potential usefulness for the evaluation of treatment efficiency. To achieve these aims, a quantitative PCR and a droplet digital PCR were developed to detect E. multilocularis ccfDNA. An AE animal model identified, for the first time, the presence of large quantities of ccfDNA. Samples from patients with AE (n = 31) were then analyzed twice, at diagnosis, and after three months of chemotherapy: about 25% were positive, almost always with very low concentrations of ccfDNA. These results confirmed that E. multilocularis produces ccfDNA, as solid tumors do, but detection may not yet be sufficient for AE diagnosis nor for the evaluation of treatment efficiency, due to the low levels of ccfDNA detected in patient serum. Copyright © 2018 Elsevier B.V. All rights reserved.

Lung function imaging methods in Cystic Fibrosis pulmonary disease.

PubMed

Kołodziej, Magdalena; de Veer, Michael J; Cholewa, Marian; Egan, Gary F; Thompson, Bruce R

2017-05-17

Monitoring of pulmonary physiology is fundamental to the clinical management of patients with Cystic Fibrosis. The current standard clinical practise uses spirometry to assess lung function which delivers a clinically relevant functional readout of total lung function, however does not supply any visible or localised information. High Resolution Computed Tomography (HRCT) is a well-established current 'gold standard' method for monitoring lung anatomical changes in Cystic Fibrosis patients. HRCT provides excellent morphological information, however, the X-ray radiation dose can become significant if multiple scans are required to monitor chronic diseases such as cystic fibrosis. X-ray phase-contrast imaging is another emerging X-ray based methodology for Cystic Fibrosis lung assessment which provides dynamic morphological and functional information, albeit with even higher X-ray doses than HRCT. Magnetic Resonance Imaging (MRI) is a non-ionising radiation imaging method that is garnering growing interest among researchers and clinicians working with Cystic Fibrosis patients. Recent advances in MRI have opened up the possibilities to observe lung function in real time to potentially allow sensitive and accurate assessment of disease progression. The use of hyperpolarized gas or non-contrast enhanced MRI can be tailored to clinical needs. While MRI offers significant promise it still suffers from poor spatial resolution and the development of an objective scoring system especially for ventilation assessment.

[Treatment of autonomous and cystic thyroid nodules with intranodular ethanol injection].

PubMed

Braga-Basaria, Milena; Trippia, Marcus Adriano; Stolf, Anderson Ravy; Mesa, Cléo; Graf, Hans

2002-01-01

Intranodular ethanol injection has been used for the past 10 years as an efficient modality for treating patients with thyroid nodules. Several studies have reported the success of this therapy in autonomous and cystic nodules and, more recently, in cold benign nodules. To evaluate the efficacy of this therapeutic modality on the treatment of autonomous and cystic thyroid nodules. 42 patients (26 with cystic and 16 with autonomous nodules) were treated with ultrasound guided intranodular 99% ethanol injection and followed for 6 months. No major complications were observed during or after treatment, however, most of the patients reported slight to moderate pain and/or discomfort after the injection. Most of the nodules showed reduction after the treatment. Autonomous nodules had a mean reduction of 50.3% and cystic nodules of 69.3%. No significant differences in pretreatment serum total T3, total T4 or TSH were observed among the patients in the cystic group. Patients in the autonomous group with hyperfunctioning nodules showed a decrease in serum total T3, total T4 and an increase in serum TSH levels, hence, proving the effectiveness of this therapy. Intranodular ethanol injection is a safe and efficient treatment for autonomous and cystic nodules of the thyroid.

Desmoplastic fibroma of the rib with cystic change: a case report and literature review.

PubMed

Okubo, Taketo; Saito, Tsuyoshi; Takagi, Tatsuya; Suehara, Yoshiyuki; Kaneko, Kazuo

2014-05-01

Desmoplastic fibroma (DF) is a rare, locally aggressive, solitary tumor microscopically composed of well-differentiated myofibroblasts with abundant dense collagen deposition. The most common sites are the long tubular bones and mandible. To our knowledge, only five cases of DF in the ribs have been reported. Here, we report a case of DF in this rare location with unusual radiological findings. A 40-year-old man presented with a 4-year history of swelling of the right chest wall. Radiographs revealed a mass at the right 9th rib, and computed tomography demonstrated a mass of 14 × 12 × 8 cm at the right 9th rib with expanded cystic change and marked calcification that appeared to have arisen from the bone. Open biopsy suggested DF. Total excision was performed, and the chest wall was reconstructed. The surgical specimen was a yellowish tumor with multilocular cystic change containing a viscous liquid. The tumor was composed of a proliferation of less-atypical spindle-shaped cells in a collagenous background. The cystic change was observed in the extra-osseous lesion. No β-catenin cytoplasmic/nuclear accumulation was detected, and no β-catenin or GNAS genetic mutations were detected. A final diagnosis of DF was made on the basis of the pathological and radiological findings. The patient was successfully treated with total excision of the tumor with no evidence of recurrence 6 months after surgery.

Functional cystic thyroid adenoma in a cat.

PubMed

Hofmeister, E; Kippenes, H; Mealey, K L; Cantor, G H; Löhr, C V

2001-07-15

A 9-year-old cat with hyperthyroidism was referred for radioactive iodine treatment. The cat also had a ventral cervical mass that the owners reported had been present for several years and had increased in size during the past few weeks. On physical examination, the mass was found to have caused lateral displacement of the trachea, esophagus, jugular vein, and common carotid artery. The mass was aspirated and was determined to be cystic in nature. Concentrations of thyroid hormones in the cystic fluid were similar to serum concentrations, and nuclear scintigraphy revealed thyroactive tissue lining the cyst wall. Magnetic resonance imaging suggested that the cyst originated from the right lobe of the thyroid gland. The cat was treated with sodium iodide I 131 but died 4 days later, presumably as a result of aspiration of gastric or esophageal contents. A necropsy was not performed, but histologic examination of a biopsy specimen of the mass indicated that it was a cystic thyroid adenoma.

Open randomised prospective comparative multi-centre intervention study of patients with cystic fibrosis and early diagnosed diabetes mellitus

PubMed Central

2014-01-01

Background Diabetes mellitus may be present in patients with cystic fibrosis starting in the second decade of life. The prevalence increases rapidly with increasing age. As life-expectancy increases in cystic fibrosis, cystic fibrosis related diabetes will be diagnosed more frequently in the future. Up to date, no data are available to answer the question if cystic fibrosis related diabetes should always initially be treated by insulin therapy. Missing data regarding oral antidiabetic treatment of newly diagnosed cystic fibrosis related diabetes are an important reason to recommend insulin treatment. Several centres report the successful management of cystic fibrosis related diabetes using oral anti-diabetic drugs at least for some years. Oral therapies would be less invasive for a patient group which is highly traumatized by a very demanding therapy. Based on an initiative of the German Mukoviszidosis-Foundation, the present study tries to answer the question, whether oral therapy with repaglinide is as effective as insulin therapy in cystic fibrosis patients with early diagnosed diabetes mellitus. Methods/Design In all cystic fibrosis patients with an age of 10 years or older, an oral glucose tolerance test is recommended. The result of this test is classified according to the WHO cut off values. It is required to have two diabetes positive oral glucose tolerance tests for the diagnosis of diabetes mellitus. This study is a multi-national, multicentre, open labelled, randomized and prospective controlled parallel group’s trial, with 24 months treatment. The primary objective of this trial is to compare the glycaemic control of oral therapy with Repaglinide with insulin injections in patients with cystic fibrosis related diabetes after 2 years of treatment. The trial should include 74 subjects showing cystic fibrosis related diabetes newly diagnosed by oral glucose tolerance test during annual screening for cystic fibrosis related diabetes. Patients are

Bone Mineral Density of Indian Children and Adolescents with Cystic Fibrosis.

PubMed

Gupta, Sumita; Mukherjee, Aparna; Khadgawat, Rajesh; Kabra, Madhulika; Lodha, Rakesh; Kabra, Sushil K

2017-07-15

To document bone mineral density of children and adolescents with cystic fibrosis. Cross-sectional study. Tertiary-care center of Northern India, July 2012 to August 2015. 52 children aged 6-18 years with cystic fibrosis and 62 healthy controls of similar age and sex. Both patients and controls were stratified into two groups, as pre-pubertal and peri-/post-pubertal, and compared for whole body bone mineral density, measured using dual energy X-ray absorptiometry. Serum levels of calcium, phosphate, alkaline phosphatase, 25-hydroxyvitamin D and parathyroid hormone were measured in children with cystic fibrosis. Compared with controls, the mean (SD) bone mineral density of children with cystic fibrosis was significantly lower in both the pre-pubertal (0.7 (0.1) g/cm2 vs 0.9 (0.1) g/cm2; P<0.001)) and peri-/post-pubertal groups (0.9 (0.1) g/cm2 vs 1.1 (0.1) g/cm2; P<0.001). Also, the mean (SD) bone mineral apparent density of pre-pubertal and peri-/post-pubertal cystic fibrosis patients was lower than the controls (P <0.001 and P= 0.01, respectively). Thirty-seven (71.2%) cystic fibrosis patients had serum 25-hydroxyvitamin D level below 15 ng/mL. Bone mineral density of children with cystic fibrosis was significantly lower than controls; majority of them were vitamin-D deficient. Intervening at an early stage of the disease and providing optimal therapy involving simultaneous management of the several factors affecting bone mineral accretion may be beneficial in improving bone health of these patients.

Archetypal analysis of diverse Pseudomonas aeruginosa transcriptomes reveals adaptation in cystic fibrosis airways

PubMed Central

2013-01-01

Background Analysis of global gene expression by DNA microarrays is widely used in experimental molecular biology. However, the complexity of such high-dimensional data sets makes it difficult to fully understand the underlying biological features present in the data. The aim of this study is to introduce a method for DNA microarray analysis that provides an intuitive interpretation of data through dimension reduction and pattern recognition. We present the first “Archetypal Analysis” of global gene expression. The analysis is based on microarray data from five integrated studies of Pseudomonas aeruginosa isolated from the airways of cystic fibrosis patients. Results Our analysis clustered samples into distinct groups with comprehensible characteristics since the archetypes representing the individual groups are closely related to samples present in the data set. Significant changes in gene expression between different groups identified adaptive changes of the bacteria residing in the cystic fibrosis lung. The analysis suggests a similar gene expression pattern between isolates with a high mutation rate (hypermutators) despite accumulation of different mutations for these isolates. This suggests positive selection in the cystic fibrosis lung environment, and changes in gene expression for these isolates are therefore most likely related to adaptation of the bacteria. Conclusions Archetypal analysis succeeded in identifying adaptive changes of P. aeruginosa. The combination of clustering and matrix factorization made it possible to reveal minor similarities among different groups of data, which other analytical methods failed to identify. We suggest that this analysis could be used to supplement current methods used to analyze DNA microarray data. PMID:24059747

Cystic fibrosis: a mucosal immunodeficiency syndrome

PubMed Central

Cohen, Taylor Sitarik; Prince, Alice

2013-01-01

Cystic fibrosis transmembrane conductance regulator (CFTR) functions as a channel that regulates the transport of ions and the movement of water across the epithelial barrier. Mutations in CFTR, which form the basis for the clinical manifestations of cystic fibrosis, affect the epithelial innate immune function in the lung, resulting in exaggerated and ineffective airway inflammation that fails to eradicate pulmonary pathogens. Compounding the effects of excessive neutrophil recruitment, the mutant CFTR channel does not transport antioxidants to counteract neutrophil-associated oxidative stress. Whereas mutant CFTR expression in leukocytes outside of the lung does not markedly impair their function, the expected regulation of inflammation in the airways is clearly deficient in cystic fibrosis. The resulting bacterial infections, which are caused by organisms that have substantial genetic and metabolic flexibility, can resist multiple classes of antibiotics and evade phagocytic clearance. The development of animal models that approximate the human pulmonary phenotypes—airway inflammation and spontaneous infection—may provide the much-needed tools to establish how CFTR regulates mucosal immunity and to test directly the effect of pharmacologic potentiation and correction of mutant CFTR function on bacterial clearance. PMID:22481418

Mutation spectrum and differential gene expression in cystic and solid vestibular schwannoma.

PubMed

Zhang, Zhihua; Wang, Zhaoyan; Sun, Lianhua; Li, Xiaohua; Huang, Qi; Yang, Tao; Wu, Hao

2014-03-01

We sought to characterize the mutation spectrum of NF2 and the differential gene expression in cystic and solid vestibular schwannomas. We collected tumor tissue and blood samples of 31 cystic vestibular schwannomas and 114 solid vestibular schwannomas. Mutation screening of NF2 was performed in both tumor and blood DNA samples of all patients. cDNA microarray was used to analyze the differential gene expression between 11 cystic vestibular schwannomas and 6 solid vestibular schwannomas. Expression levels of top candidate genes were verified by quantitative reverse transcription PCR. NF2 mutations were identified in 34.5% of sporadic vestibular schwannomas, with all mutations being exclusively somatic. No significant difference was found between the mutation detection rates of cystic vestibular schwannoma (35.5%) and solid vestibular schwannoma (34.2%). cDNA microarray analysis detected a total of 46 differentially expressed genes between the cystic vestibular schwannoma and solid vestibular schwannoma samples. The significantly decreased expression of four top candidate genes, C1orf130, CNTF, COL4A3, and COL4A4, was verified by quantitative reverse transcription PCR. NF2 mutations are not directly involved in the cystic formation of vestibular schwannoma. In addition, the differential gene expression of cystic vestibular schwannoma reported in our study may provide useful insights into the molecular mechanism underlying this process.

Congenital cystic lesions of the head and neck.

PubMed

Ibrahim, Mohannad; Hammoud, Khaled; Maheshwari, Mohit; Pandya, Amit

2011-08-01

This article presents clinical characteristics and radiologic features of congenital cervical cystic masses, among them thyroglossal duct cysts, cystic hygromas, branchial cleft cysts, and the some of the rare congenital cysts, such as thymic and cervical bronchogenic cysts. The imaging options and the value of each for particular masses, as well as present clinical and radiologic images for each, are discussed. Copyright © 2011 Elsevier Inc. All rights reserved.

Solitary Hepatocellular Carcinoma Fed by the Cystic Artery: Limitation of Transcatheter Arterial Embolization

DOE Office of Scientific and Technical Information (OSTI.GOV)

Hirota, Shozo; Matsumoto, Shinichi; Fukuda, Tetsuya

1999-05-15

Purpose: To clarify the limitations of transcatheter treatment for hepatocellular carcinoma (HCC) with parasitic feeders from the cystic artery. Methods: Three male patients had a solitary HCC (average diameter 3 cm) fed by the cystic artery among 221 patients with HCC from 1994 to 1997. One tumor was nourished entirely from the cystic artery arising from the medial branch of the left hepatic artery, and two tumors were fed partially by the cystic arteries arising from the anterior inferior branch of the right hepatic artery. We analyzed the indications for transcatheter treatment for these three patients. Results: We chose notmore » to embolize the cystic artery for fear of necrosis of the gallbladder. Although embolization of the anterior branch of the right hepatic artery was performed in one patient with a tumor fed partially by the cystic artery, only half the tumor was embolized. Two patients underwent hepatic resection, and one received percutaneous ethanol injection therapy. At follow-up of 28-40 months (average 33 months) all patients are alive. Conclusion: Feeding by the cystic artery represents a limitation of TAE for HCC.« less

Human Hydatidosis/Echinococosis in North Eastern Iran from 2003–2012

PubMed Central

ANDALIB ALIABADI, Zohreh; BERENJI, Fariba; FATA, Abdolmajid; JARAHI, Lida

2015-01-01

Background: Human cystic echinococcosis (hydatidosis) continues to be an essential cause of morbidity and mortality in many parts of the world. Methods: We studied hydatid cyst pattern in hospitalized adult patients from 2003 to 2012 in Mashhad and Neyshabour, northeast of Iran. Results: Overall, 1342 patients, 711 females (53%) and 631 males (47%) diagnosed as infected with hydatid cyst were evaluated. Their age was between 1 and 91 yr (mean age 37.75). The most affected age group was 20–30 yr old. Totally, 953 cases (71%) were urban and 375 cases (27.8%) were rural residents. The most common localization of cysts was the liver and lung. The housewives were the most frequently infected occupations. Conclusion: The rate of infection with hydatid cyst is high in Mashhad, northeast of Iran, and the incidence of human hydatidosis tends to increase in recent years so control and prevention programs are recommended. PMID:26811735

Clinicopathological review and survival characteristics of adenoid cystic carcinoma.

PubMed

Binesh, Fariba; Akhavan, Ali; Masumi, Omid; Mirvakili, Abbas; Behniafard, Nasim

2015-03-01

To study the clinical characters, the outcomes of treatments and the factors affecting treatment results of adenoid cystic carcinomas at Shahid Sadoughi Hospital and Shahid Ramazanzadeh radiotherapy center, Yazd, Iran. The clinical data of 31 patients with adenoid cystic carcinoma of any anatomic site diagnosed over an 8 year period (2004-2012), were investigated retrospectively. Data regarding patients' characteristics, pathological features and follow-up were obtained from patients records. Survival rate, local recurrence and distant metastasis were analyzed using Kaplan-Meier method. Prognosis factors were analyzed by Log-rank test and Cox regression. The study included31 patients with adenoid cystic carcinoma. The mean age at presentation was 50.2 ± 24.8 years. There were 11 (35.5 %) males and 20 (64.5 %) females with a female predilection (M:F = 0.55:1). Parotid gland was the most common site (8/31, 25.7 %) followed by submandibular gland (7/31, 22.6 %). Perineural invasion was detected in 67.7 % of the cases. Positive surgical status was reported in 48.4 % of the specimens. Metastasis was detected in 25.8 % of the patients and the most common site of distant metastasis was lung. Overall survival rates at 2, 5, and 7 years were 95, 75, and 57 % respectively. Margin status showed significant effect on survival (P value = 0.01). Positive surgical margin is an important factor affecting the prognosis of the patients with adenoid cystic carcinoma. Surgery with negative surgical margin is the first choice of treatment for the patients with adenoid cystic carcinoma. Our findings show that the prognosis of patients with adenoid cystic carcinoma in our center is fair.

Characteristics of Resting Metabolic Rate in Critically Ill, Mechanically Ventilated Adults With Cystic Fibrosis.

PubMed

Frankenfield, David C; Ashcraft, Christine M; Drasher, Tammy L; Reid, Elizabeth K; Vender, Robert L

2017-05-01

Critically ill patients with cystic fibrosis may be especially sensitive to the negative consequences of overfeeding and underfeeding, yet there is almost no information available about the energy needs of these patients. The purpose of this study was to characterize the metabolic rate of critically ill adult patients with cystic fibrosis requiring mechanical ventilation. This was an observational study in which the resting metabolic rate, oxygen consumption, and carbon dioxide production of adult patients with cystic fibrosis requiring critical care, sedation, and mechanical ventilation were measured with indirect calorimetry. This group was compared with a cohort of adult critical care patients without cystic fibrosis. Twelve patients with cystic fibrosis were identified and measured. These were compared with a control group of 25 critically ill patients. Both groups were underweight (body mass index, 17.4 ± 4.0 kg/m 2 in cystic fibrosis and 18.4 ± 2.3 kg/m 2 in control). Adjusting for differences in age, sex, height, and weight, there was no difference in resting metabolic rate between the cystic fibrosis and control groups (1702 ± 193 vs 1642 ± 194 kcal/d, P = .388). Measured resting metabolic rate matched predicted values 58% of the time in cystic fibrosis and 60% of the time in control. The resting metabolic rate of sedated adult patients with cystic fibrosis being assisted with mechanical ventilation is not different from that of adult critical care patients without cystic fibrosis. In both these underweight groups, accurate prediction of resting metabolic rate is difficult to obtain.

[Four Cases Report on Primary Lung Adenoid Cystic Carcinoma].

PubMed

He, Xilan; Chen, Jianhua

2017-11-20

Lung adenoid cystic carcinoma is a kind of rare lung cancer. Diagnosis and treatment is not enough understandable for them. We collected and analyzed 4 cases of lung adenoid cystic carcinoma for broadening the sight of this disease. Retrospectively analysed the 4 cases we collected from Hunan Cancer Hospital Between January 2012 and December 2016. We depicted the pathology, immunohistochemical, epidermal growth factor receptor (EGFR) mutation and anaplastic lymphoma kinase (ALK) arrangement in these cases. And the methods of the diagnosis and treatment were analyzed. Lung adenoid cystic carcinoma is usually located in the airway, EGFR mutation and ALK arrangement is rare in this disease. Generally the metastasis of the lung cancer occurred in the advanced stage. The prognosis is good if the mass could be resected completely. Diagnosis of the lung adenoid cystic carcinoma depends on pathological experiments, surgery is the main treatment in the early stage, radiotherapy and chemotherapy is an advisable therapy in the advanced stage. And the prognosis of this kind of lung cancer is better than small cell lung cancer and non-small cell lung cancer.

Identical twins with mature cystic teratomas treated with laparoscopic surgery: Two case reports.

PubMed

Mabuchi, Yasushi; Ota, Nami; Kobayashi, Aya; Tanizaki, Yuko; Minami, Sawako; Ino, Kazuhiko

2017-02-01

Mature cystic teratomas are the most common among all ovarian neoplasms, representing 30-40% of the cases. However, to the best of our knowledge, there have been only two reports of mature cystic teratomas occurring in identical twins to date. We herein report a case of identical twins with mature cystic teratomas who were treated with laparoscopic surgery. A 32-year-old woman was referred to our hospital due to a tumor in the right ovary. The patient underwent laparoscopic resection of the ovarian tumor and the pathological diagnosis was benign mature cystic teratoma. Two years later, the identical twin of the abovementioned patient was referred to our hospital also due to a right ovarian tumor. The patient underwent laparoscopic resection of the ovarian tumor and the pathological diagnosis was benign mature cystic teratoma. Therefore, for early diagnosis, it may be important to consider the possibility of mature cystic teratoma in the identical twin of a patient, even in the absence of symptoms.

Vitamin A absorption in cystic fibrosis: risk of hypervitaminosis A.

PubMed Central

James, D R; Owen, G; Campbell, I A; Goodchild, M C

1992-01-01

Vitamin A status was examined in nine adult cystic fibrosis patients and six adult control subjects, together with an assessment of their ability to absorb 10,000 IU of retinyl palmitate from a test meal, taken with appropriate pancreatic enzyme supplements. Median baseline values for plasma retinol and carotene, as well as median serum retinol binding protein concentrations, were significantly lower in cystic fibrosis patients than in control subjects. One cystic fibrosis patient had a raised fasting plasma retinyl ester concentration suggestive of chronic hypervitaminosis A, but no symptoms of toxicity. Measures of vitamin A absorption were also significantly lower in cystic fibrosis patients, although there was considerable overlap with control values. No correlation was observed between measures of baseline status and vitamin A absorption. Measurement of plasma retinyl esters may be an appropriate investigation in those patients considered to be at risk of chronic hypervitaminosis A. PMID:1612491

Respiratory muscle training for cystic fibrosis.

PubMed

Hilton, Nathan; Solis-Moya, Arturo

2018-05-24

Cystic fibrosis is the most common autosomal recessive disease in white populations, and causes respiratory dysfunction in the majority of individuals. Numerous types of respiratory muscle training to improve respiratory function and health-related quality of life in people with cystic fibrosis have been reported in the literature. Hence a systematic review of the literature is needed to establish the effectiveness of respiratory muscle training (either inspiratory or expiratory muscle training) on clinical outcomes in cystic fibrosis. This is an update of a previously published review. To determine the effectiveness of respiratory muscle training on clinical outcomes in people with cystic fibrosis. We searched the Cochrane Cystic Fibrosis and Genetic Disorders Group Trials register comprising of references identified from comprehensive electronic database searches and handsearches of relevant journals and abstract books of conference proceedings.Date of most recent search: 17 April 2018.A hand search of the Journal of Cystic Fibrosis and Pediatric Pulmonology was performed, along with an electronic search of online trial databases up until 07 May 2018. Randomised controlled studies comparing respiratory muscle training with a control group in people with cystic fibrosis. Review authors independently selected articles for inclusion, evaluated the methodological quality of the studies, and extracted data. Additional information was sought from trial authors where necessary. The quality of the evidence was assessed using the GRADE system MAIN RESULTS: Authors identified 19 studies, of which nine studies with 202 participants met the review's inclusion criteria. There was wide variation in the methodological and written quality of the included studies. Four of the nine included studies were published as abstracts only and lacking concise details, thus limiting the information available. Seven studies were parallel studies and two of a cross-over design. Respiratory

21 CFR 866.5910 - Quality control material for cystic fibrosis nucleic acid assays.

Code of Federal Regulations, 2013 CFR

2013-04-01

... 21 Food and Drugs 8 2013-04-01 2013-04-01 false Quality control material for cystic fibrosis... Test Systems § 866.5910 Quality control material for cystic fibrosis nucleic acid assays. (a) Identification. Quality control material for cystic fibrosis nucleic acid assays. A quality control material for...

21 CFR 866.5910 - Quality control material for cystic fibrosis nucleic acid assays.

Code of Federal Regulations, 2011 CFR

2011-04-01

... 21 Food and Drugs 8 2011-04-01 2011-04-01 false Quality control material for cystic fibrosis... Test Systems § 866.5910 Quality control material for cystic fibrosis nucleic acid assays. (a) Identification. Quality control material for cystic fibrosis nucleic acid assays. A quality control material for...

21 CFR 866.5910 - Quality control material for cystic fibrosis nucleic acid assays.

Code of Federal Regulations, 2012 CFR

2012-04-01

... 21 Food and Drugs 8 2012-04-01 2012-04-01 false Quality control material for cystic fibrosis... Test Systems § 866.5910 Quality control material for cystic fibrosis nucleic acid assays. (a) Identification. Quality control material for cystic fibrosis nucleic acid assays. A quality control material for...

21 CFR 866.5910 - Quality control material for cystic fibrosis nucleic acid assays.

Code of Federal Regulations, 2014 CFR

2014-04-01

... 21 Food and Drugs 8 2014-04-01 2014-04-01 false Quality control material for cystic fibrosis... Test Systems § 866.5910 Quality control material for cystic fibrosis nucleic acid assays. (a) Identification. Quality control material for cystic fibrosis nucleic acid assays. A quality control material for...

Distribution and function of the peptide transporter PEPT2 in normal and cystic fibrosis human lung

PubMed Central

Groneberg, D; Eynott, P; Doring, F; Thai, D; Oates, T; Barnes, P; Chung, K; Daniel, H; Fischer, A

2002-01-01

Background: Aerosol administration of peptide based drugs has an important role in the treatment of various pulmonary and systemic diseases. The characterisation of pulmonary peptide transport pathways can lead to new strategies in aerosol drug treatment. Methods: Immunohistochemistry and ex vivo uptake studies were established to assess the distribution and activity of the ß-lactam transporting high affinity proton coupled peptide transporter PEPT2 in normal and cystic fibrosis human airway tissue. Results: PEPT2 immunoreactivity in normal human airways was localised to cells of the tracheal and bronchial epithelium and the endothelium of small vessels. In peripheral lung immunoreactivity was restricted to type II pneumocytes. In sections of cystic fibrosis lung a similar pattern of distribution was obtained with signals localised to endothelial cells, airway epithelium, and type II pneumocytes. Functional ex vivo uptake studies with fresh lung specimens led to an uptake of the fluorophore conjugated dipeptide derivative D-Ala-L-Lys-AMCA into bronchial epithelial cells and type II pneumocytes. This uptake was competitively inhibited by dipeptides and cephalosporins but not ACE inhibitors, indicating a substrate specificity as described for PEPT2. Conclusions: These findings provide evidence for the expression and function of the peptide transporter PEPT2 in the normal and cystic fibrosis human respiratory tract and suggest that PEPT2 is likely to play a role in the transport of pulmonary peptides and peptidomimetics. PMID:11809991

A giant cystic pheochromocytoma mimicking liver abscess an unusual presentation - a case report.

PubMed

Sarveswaran, Venugopal; Kumar, Surees; Kumar, Amit; Vamseedharan, Muthukumar

2015-01-01

Giant cystic pheochromocytoma is a rare neuroendocrine tumor. The possibility of cystic pheochromocytoma should be considered for any peri-adrenal mass even in absence of characteristic symptoms and negative biochemical analysis. The key in the management of a case of cystic pheochromocytoma is the preoperative suspicion and the intraoperative crisis management.

Cost-Effectiveness of Screening Individuals With Cystic Fibrosis for Colorectal Cancer.

PubMed

Gini, Andrea; Zauber, Ann G; Cenin, Dayna R; Omidvari, Amir-Houshang; Hempstead, Sarah E; Fink, Aliza K; Lowenfels, Albert B; Lansdorp-Vogelaar, Iris

2017-12-27

Individuals with cystic fibrosis are at increased risk of colorectal cancer (CRC) compared to the general population, and risk is higher among those who received an organ transplant. We performed a cost-effectiveness analysis to determine optimal CRC screening strategies for patients with cystic fibrosis. We adjusted the existing Microsimulation Screening Analysis-Colon microsimulation model to reflect increased CRC risk and lower life expectancy in patients with cystic fibrosis. Modeling was performed separately for individuals who never received an organ transplant and patients who had received an organ transplant. We modeled 76 colonoscopy screening strategies that varied the age range and screening interval. The optimal screening strategy was determined based on a willingness to pay threshold of $100,000 per life-year gained. Sensitivity and supplementary analyses were performed, including fecal immunochemical test (FIT) as an alternative test, earlier ages of transplantation, and increased rates of colonoscopy complications, to assess whether optimal screening strategies would change. Colonoscopy every 5 years, starting at age 40 years, was the optimal colonoscopy strategy for patients with cystic fibrosis who never received an organ transplant; this strategy prevented 79% of deaths from CRC. Among patients with cystic fibrosis who had received an organ transplant, optimal colonoscopy screening should start at an age of 30 or 35 years, depending on the patient's age at time of transplantation. Annual FIT screening was predicted to be cost-effective for patients with cystic fibrosis. However, the level of accuracy of the FIT in population is not clear. Using a Microsimulation Screening Analysis-Colon microsimulation model, we found screening of patients with cystic fibrosis for CRC to be cost-effective. Due to the higher risk in these patients for CRC, screening should start at an earlier age with a shorter screening interval. The findings of this study

Coexistence of aspergilloma and pulmonary hydatid cyst in an immunocompetent individual.

PubMed

Aala, F; Badali, H; Hashemi Fesharaki, S; Boroumand, M; Sotoudeh Anvari, M; Davari, H; Agha Kuchak Afshari, S; Khodavaisy, S

2017-09-01

Echinococcosis is a zoonotic disease caused by Echinococcus granulosus sensu lato. The liver and lungs are the most commonly sites of infections, but involvements of other organs were also observed. Recently, the coinfection of pulmonary hydatid cyst with aspergilloma has been reported in the literature. Herein, we report a successful treatment of coinfection of cystic echinoccosis with aspergilloma due to Aspergillus flavus in a 34-year-old female. In vitro antifungal susceptibility tests revealed that the MIC values for antifungals employed in this case were posaconazole (0.031μg/ml), itraconazole (0.125μg/ml), voriconazole (0.25μg/ml), and amphotericin B (1μg/ml). The minimum effective concentration for caspofungin was 0.008μg/ml. This coexistence of active pulmonary echinococcosis and aspergillosis is being reported because of its rarity and clinical importance for its management. Copyright © 2017 Elsevier Masson SAS. All rights reserved.

A Serological Survey About Zoonoses in the Verkhoyansk Area, Northeastern Siberia (Sakha Republic, Russian Federation).

PubMed

Magnaval, Jean-François; Leparc-Goffart, Isabelle; Gibert, Morgane; Gurieva, Alla; Outreville, Jonathan; Dyachkovskaya, Praskovia; Fabre, Richard; Fedorova, Sardana; Nikolaeva, Dariya; Dubois, Damien; Melnitchuk, Olga; Daviaud-Fabre, Pascale; Marty, Marie; Alekseev, Anatoly; Crubezy, Eric

2016-02-01

In 2012, a seroprevalence survey concerning 10 zoonoses, which were bacterial (Lyme borreliosis and Q fever), parasitic (alveolar echinococcosis [AE] and cystic echinococcosis [CE], cysticercosis, toxoplasmosis, toxocariasis, and trichinellosis), or arboviral (tick-borne encephalitis and West Nile virus infection), was conducted among 77 adult volunteers inhabiting Suordakh and Tomtor Arctic villages in the Verkhoyansk area (Yakutia). Following serological testing by enzyme-linked immunosorbent assay and/or western blot, no positive result was found for cysticercosis, CE, toxocariasis, trichinellosis, and both arboviral zoonoses. Four subjects (5.2%) had anti-Toxoplasma IgG, without the presence of specific IgM. More importantly, eight subjects (10.4%) tested positive for Lyme borreliosis, two (2.6%) for recently acquired Q fever, and one (1.3%) for AE. Lyme infection and Q fever, whose presence had not been reported so far in Arctic Yakutia, appeared therefore to be a major health threat for people dwelling, sporting, or working in the Arctic area of the Sakha Republic.

Appetite stimulants for people with cystic fibrosis.

PubMed

Chinuck, Ruth; Dewar, Jane; Baldwin, David R; Hendron, Elizabeth

2014-07-27

Chronic loss of appetite in cystic fibrosis concerns both individuals and families. Appetite stimulants have been used to help cystic fibrosis patients with chronic anorexia attain optimal body mass index and nutritional status. However, these may have adverse effects on clinical status. The aim of this review is to systematically search for and evaluate evidence on the beneficial effects of appetite stimulants in the management of CF-related anorexia and synthesize reports of any side-effects. Trials were identified by searching the Cochrane Cystic Fibrosis and Genetic Disorders Group's Cystic Fibrosis Trials Register, MEDLINE, Embase, CINAHL, handsearching reference lists and contacting local and international experts.Last search of online databases: 01 April 2014.Last search of the Cystic Fibrosis Trials Register: 08 April 2014. Randomised and quasi-randomised controlled trials of appetite stimulants, compared to placebo or no treatment for at least one month in adults and children with cystic fibrosis. Authors independently extracted data and assessed the risk of bias within eligible trials. Meta-analyses were performed. Three trials (total of 47 recruited patients) comparing appetite stimulants (cyproheptadine hydrochloride and megesterol acetate) to placebo were included; the numbers of adults or children within each trial were not always reported. The risk of bias of the included trials was graded as moderate.A meta-analysis of all three trials showed appetite stimulants produced a larger increase in weight z score at three months compared to placebo, mean difference 0.61 (95% confidence interval 0.29 to 0.93) (P < 0.001) (n = 40) with no evidence of a difference in effect between two different appetite stimulants. One of these trials also reported a significant weight increase with megesterol acetate compared to placebo at six months (n = 17). The three trials reported no significant differences in forced expiratory volume at one second (per cent predicted

Imaging of benign and malignant cystic pancreatic lesions and a strategy for follow up

PubMed Central

Bhosale, Priya; Balachandran, Aparna; Tamm, Eric

2010-01-01

Cystic lesions in a variety of organs are being increasingly recognized as an incidental finding on cross-sectional imaging. These lesions can be benign, premalignant or malignant. When these cystic lesions are small it can be difficult to characterize them radiologically. However, with appropriate clinical history and knowledge of typical imaging features of cystic pancreatic lesions this can enable accurate diagnosis and thus guide appropriate treatment. In this review, we provide an overview of the most common types of cystic lesions and their appearance on computer tomography, magnetic resonance imaging and ultrasound. We will also discuss the follow up and management strategies of these cystic lesions. PMID:21160696

Variation in Cilia Protein Genes and Progression of Lung Disease in Cystic Fibrosis.

PubMed

Blue, Elizabeth; Louie, Tin L; Chong, Jessica X; Hebbring, Scott J; Barnes, Kathleen C; Rafaels, Nicholas M; Knowles, Michael R; Gibson, Ronald L; Bamshad, Michael J; Emond, Mary J

2018-04-01

Cystic fibrosis, like primary ciliary dyskinesia, is an autosomal recessive disorder characterized by abnormal mucociliary clearance and obstructive lung disease. We hypothesized that genes underlying the development or function of cilia may modify lung disease severity in persons with cystic fibrosis. To test this hypothesis, we compared variants in 93 candidate genes in both upper and lower tertiles of lung function in a large cohort of children and adults with cystic fibrosis with those of a population control dataset. Variants within candidate genes were tested for association using the SKAT-O test, comparing cystic fibrosis cases defined by poor (n = 127) or preserved (n = 127) lung function with population controls (n = 3,269 or 3,148, respectively). Associated variants were then tested for association with related phenotypes in independent datasets. Variants in DNAH14 and DNAAF3 were associated with poor lung function in cystic fibrosis, whereas variants in DNAH14 and DNAH6 were associated with preserved lung function in cystic fibrosis. Associations between DNAH14 and lung function were replicated in disease-related phenotypes characterized by obstructive lung disease in adults. Genetic variants within DNAH6, DNAH14, and DNAAF3 are associated with variation in lung function among persons with cystic fibrosis.

Isolation, motivation and balance: living with type 1 or cystic fibrosis-related diabetes.

PubMed

Tierney, Stephanie; Deaton, Christi; Webb, Kevin; Jones, Andrew; Dodd, Mary; McKenna, Diane; Rowe, Rachel

2008-04-01

To explore patients' responses to developing and managing cystic fibrosis-related diabetes and to contrast their views with those of individuals with type 1 diabetes mellitus. The incidence of diabetes among people with cystic fibrosis has increased with improvement in life expectancy. However, little is known about how patients respond to and manage cystic fibrosis-related diabetes, and how this compares with people living with type 1 diabetes mellitus. Qualitative research was undertaken in order to fully explore meanings and views. Semi-structured telephone or face-to-face interviews were conducted with patients who had cystic fibrosis-related diabetes or type 1 diabetes mellitus, during which, they discussed diagnosis and management of diabetes. Framework analysis was employed to identify themes and to consider similarities and differences between the two groups. Eleven cystic fibrosis-related diabetes and 12 type 1 diabetes mellitus patients were interviewed in 2006. Patients with cystic fibrosis-related diabetes described their diabetes diagnosis as a progression of their primary illness, management of which was important owing to the benefits it brought to their cystic fibrosis. Those with type 1 diabetes mellitus were more likely to report feeling psychologically low because of diabetes and to list long-term complications as a key factor motivating self-management. Both groups struggled to balance the demands of diabetes with other life and health obligations, and experienced isolation because of diabetes. Conclusions. Variation in perceptions recalled during interviews stemmed from diabetes being part of an existing life-threatening chronic illness in people with cystic fibrosis-related diabetes. Similarities and differences in attitudes and management practices were found, with less urgency regarding glucose monitoring and fewer information resources available for those with cystic fibrosis-related diabetes. Both groups need support for optimal diabetes

Complementary and alternative medicine use in children with cystic fibrosis.

PubMed

Giangioppo, Sandra; Kalaci, Odion; Radhakrishnan, Arun; Fleischer, Erin; Itterman, Jennifer; Lyttle, Brian; Price, April; Radhakrishnan, Dhenuka

2016-11-01

To estimate the overall prevalence of complementary and alternative medicine use among children with cystic fibrosis, determine specific modalities used, predictors of use and subjective helpfulness or harm from individual modalities. Of 53 children attending the cystic fibrosis clinic in London, Ontario (100% recruitment), 79% had used complementary and alternative medicine. The most commonly used modalities were air purifiers, humidifiers, probiotics, and omega-3 fatty acids. Family complementary and alternative medicine use was the only independent predictor of overall use. The majority of patients perceived benefit from specific modalities for cystic fibrosis symptoms. Given the high frequency and number of modalities used and lack of patient and disease characteristics predicting use, we recommend that health care providers should routinely ask about complementary and alternative medicine among all pediatric cystic fibrosis patients and assist patients in understanding the potential benefits and risks to make informed decisions about its use. Copyright © 2016 Elsevier Ltd. All rights reserved.

Mycobacterium chimaera pulmonary infection complicating cystic fibrosis: a case report.

PubMed

Cohen-Bacrie, Stéphan; David, Marion; Stremler, Nathalie; Dubus, Jean-Christophe; Rolain, Jean-Marc; Drancourt, Michel

2011-09-22

Mycobacterium chimaera is a recently described species within the Mycobacterium avium complex. Its pathogenicity in respiratory tract infection remains disputed. It has never been isolated during cystic fibrosis respiratory tract infection. An 11-year-old boy of Asian ethnicity who was born on Réunion Island presented to our hospital with cystic fibrosis after a decline in his respiratory function over the course of seven years. We found that the decline in his respiratory function was correlated with the persistent presence of a Mycobacterium avium complex organism further identified as M. chimaera. Using sequencing-based methods of identification, we observed that M. chimaera organisms contributed equally to respiratory tract infections in patients with cystic fibrosis when compared with M. avium subsp. hominissuis isolates. We believe that M. chimaera should be regarded as an emerging opportunistic respiratory pathogen in patients with cystic fibrosis, including young children, and that its detection warrants long-lasting appropriate anti-mycobacterial treatment to eradicate it.

Not All Children with Cystic Fibrosis Have Abnormal Esophageal Neutralization during Chemical Clearance of Acid Reflux.

PubMed

Woodley, Frederick W; Moore-Clingenpeel, Melissa; Machado, Rodrigo Strehl; Nemastil, Christopher J; Jadcherla, Sudarshan R; Hayes, Don; Kopp, Benjamin T; Kaul, Ajay; Di Lorenzo, Carlo; Mousa, Hayat

2017-09-01

Acid neutralization during chemical clearance is significantly prolonged in children with cystic fibrosis, compared to symptomatic children without cystic fibrosis. The absence of available reference values impeded identification of abnormal findings within individual patients with and without cystic fibrosis. The present study aimed to test the hypothesis that significantly more children with cystic fibrosis have acid neutralization durations during chemical clearance that fall outside the physiological range. Published reference value for acid neutralization duration during chemical clearance (determined using combined impedance/pH monitoring) was used to assess esophageal acid neutralization efficiency during chemical clearance in 16 children with cystic fibrosis (3 to <18 years) and 16 age-matched children without cystic fibrosis. Duration of acid neutralization during chemical clearance exceeded the upper end of the physiological range in 9 of 16 (56.3%) children with and in 3 of 16 (18.8%) children without cystic fibrosis ( p =0.0412). The likelihood ratio for duration indicated that children with cystic fibrosis are 2.1-times more likely to have abnormal acid neutralization during chemical clearance, and children with abnormal acid neutralization during chemical clearance are 1.5-times more likely to have cystic fibrosis. Significantly more (but not all) children with cystic fibrosis have abnormally prolonged esophageal clearance of acid. Children with cystic fibrosis are more likely to have abnormal acid neutralization during chemical clearance. Additional studies involving larger sample sizes are needed to address the importance of genotype, esophageal motility, composition and volume of saliva, and gastric acidity on acid neutralization efficiency in cystic fibrosis children.

Oral calorie supplements for cystic fibrosis.

PubMed

Smyth, Rosalind L; Rayner, Oli

2017-05-04

Poor nutrition occurs frequently in people with cystic fibrosis and is associated with other adverse outcomes. Oral calorie supplements are used to increase total daily calorie intake and improve weight gain. However, they are expensive and there are concerns they may reduce the amount of food eaten and not improve overall energy intake. This is an update of a previously published review. To establish whether in people with cystic fibrosis, oral calorie supplements: increase daily calorie intake; and improve overall nutritional intake, nutritional indices, lung function, survival and quality of life. To assess adverse effects associated with using these supplements. We searched the Cochrane Cystic Fibrosis Trials Register comprising references from comprehensive electronic database searches, handsearches of relevant journals and abstract books of conference proceedings. We contacted companies marketing oral calorie supplements.Last search: 18 October 2016. Randomised or quasi-randomised controlled trials comparing use of oral calorie supplements for at least one month to increase calorie intake with no specific intervention or additional nutritional advice in people with cystic fibrosis. We independently selected the included trials, assessed risk of bias and extracted data. We contacted the authors of included trials and obtained additional information for two trials. We identified 21 trials and included three, reporting results from 131 participants lasting between three months and one year. Two trials compared supplements to additional nutritional advice and one to no intervention. Two of the included trials recruited only children. In one trial the risk of bias was low across all domains, in a second trial the risk of bias was largely unclear and in the third mainly low. Blinding of participants was unclear in two of the trials. Also, in one trial the clinical condition of groups appeared to be unevenly balanced at baseline and in another trial there were

US Cystic Fibrosis Foundation and European Cystic Fibrosis Society consensus recommendations for the management of non-tuberculous mycobacteria in individuals with cystic fibrosis