Potential risk factors associated with human alveolar echinococcosis: Systematic review and meta-analysis.

BACKGROUND: Human alveolar echinococcosis (AE) is a severe zoonotic disease caused by the metacestode stage of Echinococcus multilocularis. AE is commonly associated with a long incubation period that may last for more than ten years. The objective of this systematic literature review was to identify and summarize the current knowledge on statistically relevant potential risk factors (PRFs) associated with AE in humans. METHODOLOGY/PRINCIPAL FINDINGS: Six bibliographic databases were searched, generating a total of 1,009 publications. Following the removal of duplicate records and the exclusion of papers that failed to meet the criteria of a previously agreed a priori protocol, 23 publications were retained; however, 6 of these did not contain data in a format that allowed their inclusion in the meta-analysis. The remaining 17 publications (6 case-control and 11 cross-sectional studies) were meta-analysed to investigate associations between AE and PRFs. Pooled odds ratios (OR) were used as a measure of effect and separately analysed for case-control and cross-sectional studies. In the case-control studies, the following PRFs for human AE showed higher odds of outcome: "dog ownership", "cat ownership", "have a kitchen garden", "occupation: farmer", "haymaking in meadows not adjacent to water", "went to forests for vocational reasons", "chewed grass" and "hunting / handling foxes". In the cross-sectional studies, the following PRFs showed higher odds of outcome: "dog ownership", "play with dogs", "gender: female", "age over 20 years", "ethnic group: Tibetan", "low income", "source of drinking water other than well or tap", "occupation: herding" and "low education". Our meta-analysis confirmed that the chance of AE transmission through ingestion of food and water contaminated with E. multilocularis eggs exists, but showed also that food- and water-borne PRFs do not significantly increase the risk of infection. CONCLUSIONS/SIGNIFICANCE: This systematic review analysed international peer-reviewed articles that have over the years contributed to our current understanding of the epidemiology of human AE. The identification of potential risk factors may help researchers and decision makers improve surveillance and/or preventive measures that aim at decreasing human infection with E. multilocularis. More primary studies are needed to confirm potential risk factors and their role in the epidemiology of human AE.

Is there echinococcosis in West Africa? A refugee from Niger with a liver cyst.

BACKGROUND: Italy is presently facing an increase in immigration from sub-Saharan Africa through the Mediterranean Sea. Case reports of human cystic echinococcosis (CE) have been reported from most sub-Saharan countries. Therefore, an increase in the number of patients with CE coming from these areas in the Italian and European centers for infectious diseases is expected. Unfortunately, the epidemiology of CE in sub-Saharan countries is poorly known, which makes clinical suspicion and diagnosis of such infection difficult in patients coming from these areas. RESULTS: Here we report a case of hepatic CE in a patient from Niger who arrived in Italy through Libya and visited in a Tropical Medicine referral center in Northern Italy. The parasite was identified molecularly as the G6 "camel" strain of Echinococcus granulosus (E. canadensis). The diagnosis and management of a chronic and clinically complex infection like CE in such situation is difficult. Only 40 cases of CE from Niger have been reported; of these, 75% had extra-hepatic localization. To our knowledge, no strain characterization of human isolates from Niger has been reported so far. The CE cyst of the patient was in CE3a stage, indicating active transmission from the area in which the patient came. However, prevalence data from Niger, and from any other country in West Africa, are almost inexistent. CONCLUSIONS: We argue that population epidemiology surveys with ultrasound are warranted in Sahelian countries, including Niger. These studies could improve the knowledge of CE epidemiology, provide health authorities with important information for public health interventions targeting this zoonosis, and shed light on any difference between tissue tropism and clinical manifestations caused by the different E. granulosus strains.

Transmission ecology of taeniid larval cestodes in rodents in Sweden, a low endemic area for Echinococcus multilocularis.

Although local prevalence of Echinococcus multilocularis may be high, this zoonotic parasite has an overall low prevalence in foxes and rodents in Sweden. To better understand opportunities for E. multilocularis transmission in the Swedish environment, the aim of this study was to investigate other taeniid cestodes and to relate observed patterns to E. multilocularis. Cestode parasites were examined in fox feces and rodents caught in different habitats from four regions of Sweden. Arvicola amphibius and Microtus agrestis were parasitized with Versteria mustelae, Hydatigera taeniaeformis s. l., and E. multilocularis, whereas Myodes glareolus and Apodemus spp. were parasitized with V. mustelae, Taenia polyacantha, H. taeniaeformis s.l., and Mesocestoides spp. Rodents caught in field habitat (Ar. amphibius, Mi. agrestis) were more likely (OR 10, 95% CI 5-19) to be parasitized than rodents caught in forest habitat (My. glareolus, Apodemus spp.). The parasite preference for each rodent species was present regardless of the type of background contamination from fox feces. These results further support the importance of both ecological barriers and individual species susceptibility in parasite transmission, and indicate that future monitoring for E. multilocularis in the Swedish environment should focus in field habitats where Mi. agrestis and Ar. amphibius are abundant.

Global Distribution of Alveolar and Cystic Echinococcosis.

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.

Autochthonous human alveolar echinococcosis in a Hungarian patient.

BACKGROUND: Alveolar echinococcosis is a zoonotic parasitic disease causing a severe clinical condition and is known as the most deadly of all helminth infections. Moreover, this disease is also an increasing concern in Northern and Eastern Europe due to its spread in the wildlife animal host. CASE PRESENTATION: An asymptomatic 70-year-old woman from south-western Hungary was diagnosed with multiple liver lesions. Imaging techniques (ultrasound, computed tomography and magnetic resonance imaging), serology (ELISA, indirect hemagglutination and Western blot), and conventional staining methods (hematoxylin-eosin and periodic acid-Schiff) were used for the detection of the disease. A histopathological re-evaluation of formalin-fixed paraffin block by immunohistochemical staining with the monoclonal antibody Em2G11 definitively confirmed the diagnosis of alveolar echinococcosis. CONCLUSIONS: To our knowledge, this is the first confirmed autochthonous case of human alveolar echinococcosis in Hungary. To what extent diagnostic difficulties may contribute to underestimate this zoonosis in Eastern Europe is unknown. Differential diagnosis with alveolar echinococcosis should be considered for patients with multiple, tumor-like cystic lesions of the liver, in countries where this parasite is emerging.

Fresh fruits, vegetables and mushrooms as transmission vehicles for Echinococcus multilocularis in highly endemic areas of Poland: reply to concerns.

Echinococcus multilocularis is a tapeworm that may cause alveolar echinococcosis (AE), one of the most dangerous parasitic zoonoses. As in the case of other foodborne diseases, unwashed fruits and vegetables, contaminated with dispersed forms of E. multilocularis, may serve as an important transmission route for this parasite. In this article, we reply to the incorrect interpretation of results of our study concerning the detection of E. multilocularis DNA in fresh fruit, vegetable and mushroom samples collected from the highly endemic areas of the Warmia-Masuria Province, Poland, to dispel any doubts. The accusations formulated by the commentators concerning our paper are unfounded; moreover, these commentators demand information which was beyond the purview of our study. Making generalisations and drawing far-reaching conclusions from our work is also unjustified. The majority of positive samples were found in only a few hyperendemic communities; this information corresponds with the highest number of both infected foxes and AE cases in humans recorded in this area. Our findings indicate that E. multilocularis is present in the environment and may create a potential risk for the inhabitants. These people should simply be informed to wash fruits and vegetables before eating. No additional far-reaching conclusions should be drawn from our data. We believe these commentators needlessly misinterpreted our results and disseminated misleading information. Nevertheless, we would like to encourage any readers simply to contact us if any aspects of our study are unclear.

A Newly Discovered Epidemic Area of Echinococcus multilocularis in West Gansu Province in China.

Alveolar echinococcosis (AE) is a lethal parasitic disease. In Gansu Province of China, all AE cases reported in literature were from Zhang and Min Counties, the southern part of the province. Here, we report the discovery of nine AE cases and one cystic echinococcosis (CE) case from Nanfeng Town of Minle County, in the middle of Hexi Corridor in west Gansu Province. The diagnosis of these cases were confirmed by serology, histopathology, computed tomography, B-ultrasound, immunohistochemistry method, DNA polymerase chain reaction and sequencing analysis. Because eight of nine AE cases came from First Zhanglianzhuang (FZLZ) village, we conducted preliminary epidemiological analyses of 730 persons on domestic water, community and ecology such as 356 dogs' faeces of FZLZ, in comparison with those of other five villages surrounding FZLZ. Our studies indicate that Nanfeng Town of Minle County is a newly discovered focus of AE in China as a CE and AE co-epidemic area. Further research of Echinococcus multilocularis transmission pattern in the area should be carried for prevention of this parasitic disease.

Echinococcus multilocularis in North America: the great unknown.

Over the last decade, studies have begun to shed light on the distribution and genetic characterization of Echinococcus multilocularis, the causative agent of alveolar echinococcosis (AE), in North America. Recent findings indicate that the parasite is likely expanding its range in the central region of the United States and Canada and that invasions of European strains might have occurred. In our review, we present the available data on E. multilocularis infections in wild and domestic animals and humans in North America and emphasize the lack of knowledge on the distribution of the parasite in wild and domestic hosts. Furthermore, we stress the need to better understand the complexity of host communities and their roles in shaping the transmission and distribution of the parasite. We hypothesize that a lack of knowledge about AE by North American physicians might result in the misdiagnosis of cases and an underestimation of disease incidence. The endemic presence of the parasite in urban areas and a recent human case in Alberta, Canada, suggest that the scientific community may need to reconsider the local public health risks, re-assess past cases that might have been overlooked and increase surveillance efforts to identify new cases of human AE.

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

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.

Guía provincial para el manejo de la hidatidosis hepática: Neuquén, 2012 / Provincial guide for the management of hepatic hydatidosis: Neuquén, 2012

La hidatidosis es una parasitosis zoonótica, causada por el cestode Echinococcus (E.) granulosus. En nuestro país es un importante problema de salud pública al ser considerada como enfermedad endémica, especialmente en regiones como la provincia de Neuquén, donde uno de sus pilares económicos es la actividad ganadera, especialmente la cría de ovinos y caprinos.(AU)

Guía provincial para el manejo de la hidatidosis hepática: Neuquén, 2012 / Provincial guide for the management of hepatic hydatidosis: Neuquén, 2012

La hidatidosis es una parasitosis zoonótica, causada por el cestode Echinococcus (E.) granulosus. En nuestro país es un importante problema de salud pública al ser considerada como enfermedad endémica, especialmente en regiones como la provincia de Neuquén, donde uno de sus pilares económicos es la actividad ganadera, especialmente la cría de ovinos y caprinos.

Alveolar echinococcosis-spreading disease challenging clinicians: a case report and literature review.

Human alveolar echinococcosis (AE) is a potentially deadly disease; recent studies have shown that the endemic area of Echinococcus multilocularis, its causative agent, is larger than previously known. This disease has low prevalence and remains underreported in Europe. Emerging clinical data show that diagnostic difficulties are still common. We report on a 76-year old patient suffering from AE lesions restricted to the left lobe of the liver who underwent a curative extended left hemihepatectomy. Prior to the resection a liver biopsy under the suspicion of an atypical malignancy was performed. After the intervention he developed a pseudoaneurysm of the hepatic artery that was successfully coiled. Surprisingly, during surgery, the macroscopic appearance of the tumour revealed a growth pattern that was rather typical for cystic echinococcosis (CE), i.e., a gross tumour composed of multiple large vesicles with several centimeters in diameter. In addition, there were neither extensive adhesions nor infiltrations of the neighboring pancreas and diaphragm as was expected from previous imaging results. The unexpected diagnosis of AE was confirmed by definite histopathology, specific polymerase chain reaction and serology results. This is a rare case of unusual macroscopic presentation of AE that posed immense diagnostic challenges and had an eventful course. To our knowledge this is the first case of an autochthonous infection in this particular geographic area of Germany, the federal state of Saxony. This report may provide new hints for an expanding area of risk for AE and emphasizes the risk of complications in the scope of diagnostic procedures and the limitations of modern radiological imaging.

Transmission ecosystems of Echinococcus multilocularis in China and Central Asia.

From continental to regional scales, the zoonosis alveolar echinococcosis (AE) (caused by Echinococcus multilocularis) forms discrete patches of endemicity within which transmission hotspots of much larger prevalence may occur. Since the late 80s, a number of hotspots have been identified in continental Asia, mostly in China, wherein the ecology of intermediate host communities has been described. This is the case in south Gansu, at the eastern border of the Tibetan plateau, in south Ningxia, in the western Tian Shan of Xinjiang, and in the Alay valley of south Kyrgyzstan. Here we present a comparative natural history and characteristics of transmission ecosystems or ecoscapes. On this basis, regional types of transmission and their ecological characteristics have been proposed in a general framework. Combining climatic, land cover and intermediate host species distribution data, we identified and mapped 4 spatially distinct types of transmission ecosystems typified by the presence of one of the following small mammal 'flagship' species: Ellobius tancrei, Ochotona curzoniae, Lasiopodomys brandtii or Eospalax fontanierii. Each transmission ecosystem had its own characteristics which can serve as a reference for further in-depth research in the transmission ecology of E. multilocularis. This approach may be used at fine spatial scales to characterize other poorly known transmission systems of the large Eurasian endemic zone, and help in consideration of surveillance systems and interventions.

Populations at risk for alveolar echinococcosis, France.

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.

The emergence of echinococcosis in central Asia.

SUMMARY Following the collapse of the Soviet Union in 1991, there was an increase in the number of cases of human echinococcosis recorded throughout central Asia. Between 1991 and 2001 incidence rates of cystic echinococcosis (CE) increased by 4 fold or more. There also appeared to be increases in prevalence of CE in livestock and prevalences of Echinococcus granulosus reported in dogs. The increase in human echinococcosis was associated with changes in livestock husbandry, decline in veterinary public health services, increases in dog populations and increased poverty, all of which served to promote transmission of E. granulosus. A few years after reports of increased transmission of E. granulosus, the first reports of E. multilocularis infection in dogs were recorded. Further studies indicated that in both Kazakhstan and Kyrgyzstan prevalences of up to 18% were present. Recently there has been a dramatic increase in the number of cases of human alveolar echinococcosis recorded in Kyrgyzstan with over 60 cases reported in 2011.

A new intermediate host for Echinococcus multilocularis: the southern red-backed vole (Myodes gapperi) in urban landscape in Calgary, Canada.

Human Alveolar Echinococcosis (HAE) is a potentially fatal parasitic disease caused by Echinococcus multilocularis, a cestode characterized by a sylvatic life-cycle involving several species of rodents and lagomorphs as intermediate hosts and canids as definitive hosts. Despite the wide distribution of the parasite in North America, the number of competent intermediate host species identified to date is still relatively small, and mainly includes the northern vole (Microtus oeconomus), brown lemming (Lemmus sibiricus), northern red-backed vole (Myodes rutilus), deer mouse (Peromyscus maniculatus) and meadow vole (Microtus pennsylvanicus). By monitoring the infections in rodents in the city of Calgary (Alberta, Canada), we have detected a case of severe alveolar echinococcosis in a southern red-backed vole (Myodes gapperi), a species never reported before as an intermediate host for this parasite. Observation of protoscolices in the intra-abdominal multilocular cysts indicates that M. gapperi could act as a competent intermediate host for the transmission of E. multilocularis. Since M. gapperi can be found in close proximity to, and within metropolitan areas, this species could play a role in the establishment and maintenance of the sylvatic life-cycle of E. multilocularis in urban landscapes, where the potential for zoonotic transmission is higher. The new intermediate host reported needs to be taken into account in future surveys and transmission models for this parasite.