• Echinococcus multilocularis
• alveolar echinococcosis
• diagnostic techniques
• parasite

• 2017YFA0205201 the Major State Basic Research Development Program of China
• 81925019 the National Natural Science Foundation of China
• U1705281 the National Natural Science Foundation of China
• 20720190088 the Fundamental Research Funds for the Central Universities
• 20720200019 the Fundamental Research Funds for the Central Universities
• 2020Y4003 the Science Foundation of Fujian Province
• 201812631010 the Science Foundation of Fujian Province
• NCET-13-0502 the Program for New Century Excellent Talents in University, China
• GZC20231414 the Postdoctoral Fellowship Program of CPSF

• Alveolar echinococcosis
• Cyst infection
• Echinococcus multilocularis
• Interventional radiology
• Percutaneous cyst drainage
• Virtual non-enhanced dual-energy computed tomography

• Classification
• Hepatic echinococcosis
• PET/CT
• Semi-quantitative parameters

• Humans
• Fluorodeoxyglucose F18
• Positron Emission Tomography Computed Tomography/methods
• Male
• Echinococcosis, Hepatic/diagnostic imaging
• Female
• Adult
• Middle Aged
• Radiopharmaceuticals
• Tibet
• Aged
• Young Adult
• ROC Curve
• Retrospective Studies
• Sensitivity and Specificity

• National Natural Science Foundation of China

• Cerebral alveolar echinococcosis
• Cerebral cystic echinococcosis
• Clinicopathological Features

• Humans
• Adult
• Male
• Female
• Middle Aged
• China/epidemiology
• Echinococcosis/pathology
• Young Adult
• Magnetic Resonance Imaging
• Diagnosis, Differential
• Brain Diseases/parasitology
• Brain Diseases/pathology
• Adolescent
• Brain/pathology
• Brain/parasitology

• 2022D01C244 Natural Science Foundation of Xinjiang Uyghur Autonomous Region

• computed tomography
• hepatic alveolar echinococcosis
• nomogram
• perilesional
• radiomics

• alveoläre echinokokkose
• echinococcus multilocularis
• epidemiologie
• prävalenz
• deutschland
• alveolar echinococcosis
• epidemiology
• prevalence
• germany

• Male
• Humans
• Female
• Infant, Newborn
• Germany/epidemiology
• Retrospective Studies
• Echinococcosis/epidemiology
• Berlin
• Prevalence

• Alveolar echinococcosis
• Interventional
• Liver
• Radiology
• Treatment

• Alveolar echinococcosis
• Contrast-enhanced ultrasound
• EMUC-US
• Echinococcus multilocularis
• Hepatic hemangioma
• Ultrasonography

• Echinococcus multilocularis
• MGB probe.
• Nested PCR
• cfDNA

• Alveolar echinococcosis
• Cystic Echinococcosis
• Diagnostic imaging
• Echinococcosis
• Radiologists
• Radiology (Interventional)

• CT
• MRI
• US
• alveolar echinococcosis
• imaging

Human alveolar echinococcosis (AE), which is caused by the cestode Echinococcus (E.) multilocularis, is an epidemiologically relevant issue in modern medicine and still poses a diagnostic and therapeutic challenge. Since diagnosis mainly relies on imaging procedures and serological testing, we retrospectively and comparatively analyzed the performance of an Echinococcus IgG screening ELISA, whole serum IgE, and two specific confirmatory ELISA platforms using the defined E. multilocularis antigens Em2-Em18 (Em2+) and recombinant Em18 (recEm18). With special emphasis on the clinical usefulness of recEm18, we correlated the laboratory results with clinical characteristics and imaging findings in a large and well-characterized cohort of N = 124 AE patients, who were followed over several years after either surgical plus subsequent pharmacological treatment or pharmacotherapy alone. All patients had routinely received PET-CTI every two years. Our data reveal strong correlations for both Echinococcus IgG and recEm18 with tracer uptake in PET-CTI and parasitic lesion size and number, suggesting additional clinical usefulness of recEm18 for certain constellations only, while IgG and Em2+ still appear reasonable and sensitive screening methods for initial diagnosis of AE. With this study, we aim to contribute to further optimizing medical care of AE patients. For instance, it might be reasonable to consider the replacement of some PET-CTI follow-ups by imaging procedures with less radiation exposure or serological means alone. Further studies that clarify the correlation of serological markers with ultrasound criteria might be particularly useful, and further retrospective as well as prospective investigations are justified in this context.

Echinococcus granulosus sensu lato and Echinococcus multilocularis are responsible for serious health and economic implications for humans and animals. This study was designed to conduct a bibliometric analysis of global research on echinococcosis/hydatidosis included in the Web of Science Core Collection databases from 2000 to 2019. A total of 7066 relevant articles between 2000 and 2019 were identified. Most articles were published in 2015 (502 articles), 2017 (492 articles) and 2018 (493 articles), with the Veterinary Parasitology journal publishing the largest number of articles (237). Researchers from Xinjiang Medical University, China authored the most articles (388) in the field. Authors Craig, P.S. and Deplazes, P. were the most active in publishing143 and 126 hydatid cyst research papers, respectively. The most echinococcosis/hydatidosis publications originated from Turkey, China and Iran, with 1210, 708 and 531 articles, respectively. The highest levels of research collaboration were evident between China- England, China-France, England-France, China-Australia, and China-Japan. Also, the top researchers in this field had relatively extensive collaborations with each other. Our bibliometric analysis provides a picture of the scientific research into the echinococcosis/hydatidosis field. Further multi-national collaborative research efforts in this field should show promising progress in the future.

• Bibliometric
• Echinococcosis
• Echinococcus granulosus sensu lato
• Echinococcus multilocularis
• Hydatidosis
• Scientific collaboration
• Web of science

Alveolar echinococcosis (AE) is considered as a fatal zoonosis caused by the larvae of Echinococcus multilocularis. The lungs and brain are the most common metastatic organs. We report a human case of hepatic alveolar echinococcosis accompanied by lung and brain metastasis. In particular, the patient had a history of tuberculosis and the lung lesions were easily misdiagnosed as lung abscesses. The lesions of liver and lung underwent radical resection and confirmed as alveolar echinococcosis by pathological examination. The patient had no surgical complications after operation and was discharged after symptomatic treatment. Unfortunately, the patient later developed multiple intracerebral AE metastases. We required the patient to take albendazole orally for life and follow up.

• Echinococcus multilocularis
• alveolar echinococcosis
• human
• misdiagnosis

• Alveolar echinococcosis
• Laparoscopic approach
• Open surgery

• echinococcosis
• retroperitoneal
• surgical treatment

• Adult
• Albendazole/therapeutic use
• Anthelmintics/therapeutic use
• Echinococcosis/pathology
• Echinococcosis/surgery
• Echinococcosis, Hepatic/pathology
• Echinococcosis, Hepatic/surgery
• Female
• Humans
• Male
• Middle Aged
• Retroperitoneal Space/pathology
• Retroperitoneal Space/surgery
• Retrospective Studies
• Severity of Illness Index
• Tomography, X-Ray Computed

Human alveolar echinococcosis (AE) caused by Echinococcus multilocularis is an underreported, often misdiagnosed and mistreated parasitic disease mainly due to its low incidence. The aim of this study was to describe the epidemiological and clinical characteristics of human AE patients in Hungary for the first time.

Between 2003 and 2018, epidemiological and clinical data of suspected AE patients were collected retrospectively from health database management systems.

This case series included a total of 16 AE patients. The mean age of patients was 53 years (range: 24–78 years). The sex ratio was 1:1. Four patients (25%) revealed no recurrence after radical surgery and adjuvant albendazole (ABZ) therapy. For five patients (31.3%) with unresectable lesions, a stabilization of lesions with ABZ treatment was achieved. In seven patients (43.8%), progression of AE was documented. The mean diagnostic delay was 33 months (range: 1–122 months). Three AE related deaths (fatality rate 18.8%) were recorded.

AE is an emerging infectious disease in Hungary with a high fatality rate since based on our results, almost every fifth AE patient died in the study period. Differential diagnosis and appropriate surgical and medical therapy for AE is an urging challenge for clinicians in Hungary, as well as in some other European countries where E. multilocularis is prevalent.

• Case series
• Clinical epidemiology
• Echinococcus multilocularis
• Human alveolar echinococcosis
• Hungary

Background: At present, laparoscopy is relatively mature as a minimally invasive technique, but there are few reports on this approach for the radical treatment of hepatic alveolar echinococcosis (AE). In this study, we aimed to evaluate the safety and feasibility of laparoscopic hepatectomy (LH) for AE treatment. Results: A retrospective review of medical records obtained from 13 patients diagnosed with AE between January 2018 and December 2019 and treated with laparoscopic hepatectomy was conducted at the First Affiliated Hospital of Xinjiang Medical University. All patients (n = 13) underwent hepatic resection using laparoscopy and none were transferred to open surgery. The average duration of surgery was 285 min (145–580 min). Intraoperative bleeding was 305 mL (20–2000 mL). The mean duration of postoperative catheterization was 6.9 days (3–21 days), and postoperative hospital stay was 7.2 days (4–14 days). No complication of Clavien-Dindo grade III or above occurred, except for the second patient with acute liver failure post-surgically. No recurrences or deaths were observed at 9–30 months of follow-up. Conclusions: Laparoscopic hepatectomy appears to be safe and effective in selected AE patients. The advantages of this technique for AE treatment need to be further compared with the classical open approach.

• Alveolar echinococcosis
• Laparoscopic hepatectomy
• Minimally invasive

Cystic echinococcosis (CE) is a neglected zoonotic disease caused by Echinococcus granulosus sensu lato. Diagnosis and monitoring of CE rely primarily on imaging while serology is used as a confirmatory test. However, imaging is not always conclusive and currently available serological assays have suboptimal sensitivity and specificity, lack standardization, and are not useful for patients´ follow-up. Seroassays for CE are usually based on hydatid fluid (HF), a complex, variable antigenic mixture, and cross-reactivity exists especially with alveolar echinococcosis. Recombinant proteins based on immunogenic antigens most abundant in HF, such as AgB1, AgB2 and Ag5, have been used to overcome these limitations. None of them so far showed potential to replace HF; however, their performance have been largely tested on a limited number of samples, and comparison of different antigens using the same cohort has been rarely performed. The combination of several immunogenic epitopes in a single recombinant protein could enhance test sensitivity. For the diagnosis and follow-up of patients with CE, we compared the performance of the crude HF, previously described recombinant 2B2t antigen, and GST-tagged version of 2B2t, and novel designed recombinants (GST-Ag5t and the GST-DIPOL chimera containing AgB1, AgBB2 and Ag5 epitopes) by IgG-ELISA format. Samples belong to a retrospective cohort of 253 well-characterized patients with CE, previously described for the evaluation of the 2B2t antigen, 92 patients with alveolar echinococcosis, and 82 healthy donors. The reference standard for CE diagnosis was the presence of a CE lesion as diagnosed by ultrasonography. The highest sensitivity was obtained with HF [86.7%, 95% confidence interval (CI): 81.2–91.0], followed by GST-2B2t (70.0%, 95% CI: 63.1–76.2), 2B2t (65.5%, 95% CI: 58.5–72.0), GST-Ag5t (64.5%, 95% CI: 57.5–71.1) and GST-DIPOL (63.1%, 95% CI: 56.0–69.7). The GST-2B2t had the best specificity (95.8%, 95% CI: 88.3–99.1) and the lowest cross-reactivity (38.7%, 95% CI: 27.6–50.6). Good response to treatment also correlated to negative test results in the GST-2B2t ELISA. While none of the tested recombinant antigen appears suitable to replace HF for the diagnosis of CE, GST-2B2t should be further explored as a confirmation test, based on its high specificity and low cross-reactivity, and for the follow-up after treatment in those patients with positive serology for this antigen.

Cystic echinococcosis (CE) is a neglected parasitic zoonosis. Its diagnosis and follow-up require evaluation with imaging. Currently available serological tests are applied to confirm the diagnosis in doubtful cases, although having limitations in diagnostic accuracy, and they are not useful for patients’ follow-up. Seroassays for CE are usually based on hydatid fluid (HF) obtained from infected animals, with consequent problems of heterogeneity and low specificity. The use of semi-purified HF derivatives or recombinant antigens has been attempted to improve these aspects, but with an unacceptable loss in sensitivity. Most newly developed antigens have been tested on a limited number of samples, not always well characterized, and have been rarely compared using the same samples cohort. Here, we tested and compared three recombinant antigens (2B2t, GST-2B2t and GST-Ag5t), and a recombinant chimeric antigen (DIPOL) based on three highly immunogenic components of HF (B1, B2 and Ag5), in an attempt to increase the sensitivity of recombinant antigen-based seroassays for the diagnosis and follow-up of patients with CE. We found that GST-2B2t had higher sensitivity than the other antigenic preparations, but still not as high as HF, and that GST-2B2t and GST-DIPOL had statistically higher specificity than any of the other tested antigens. GST-2B2t also showed potential for the follow-up of patients with CE after drug treatment.

• Animals
• Antibodies, Helminth/blood
• Antigens, Helminth/genetics
• Antigens, Helminth/immunology
• Cohort Studies
• Cross Reactions
• Echinococcosis/diagnosis
• Echinococcosis/immunology
• Echinococcus granulosus/immunology
• Enzyme-Linked Immunosorbent Assay/methods
• Female
• Humans
• Immunoassay
• Italy
• Male
• Recombinant Proteins/immunology
• Retrospective Studies
• Sensitivity and Specificity
• Serologic Tests/methods

• FP7 International Cooperation
• Ministerio de Ciencia, Innovación y Universidades

Positron emission tomography-computed tomography (PET-CT) with 18F-fluorodesoxyglucose (FDG) is the imaging modality of choice for assessing inflammation surrounding hepatic alveolar echinococcosis (AE) lesions. This study is the first to evaluate FDG uptake in hepatic AE (n = 51) based on the standardized uptake value (SUV) and to correlate the SUVs with primary morphology and calcification patterns, based on the Echinococcus multilocularis Ulm Classification for Computed-Tomography (EMUC-CT). Our results show that the SUVs were increased for lesions with EMUC-CT types I-IV primary morphology, compared to the surrounding healthy liver tissue (SUV = 2.5 ± 0.4; p < 0.05). Type IV lesions included, by far, the highest number of PET-negative lesions. A comparison of lesions with different primary morphologies showed clear differences. The highest SUVs were found for types I and III, and the lowest was found for type IV. Type IV lesions (SUV, 3.8 ± 1.5) showed significantly lower uptake compared to type I (SUV, 6.9 ± 3.5; p = 0.030) and type III (SUV, 7.4 ± 3.9; p = 0.031) lesions. For type II lesions, the results showed only a statistical trend (SUV, 6.1 ± 3.1; p = 0.073). Due to the small number of cases, an evaluation of type V (n = 1) lesions was not possible. The different SUVs of lesions with different primary morphologies, particularly the lower FDG uptake observed in type IV lesions, suggested that these SUVs might reflect different stages of the disease.

Echinococcosis multilocularis (Hydatid Disease - HD) is a zoonotic disease caused by the larval form of Echinococcus multilocularis (EM). The main sites for this zoonosis are the Middle East, China, India, Alaska, and Siberia. It is rather rare in Europe. In Poland, the Warmian-Masurian Province is the endemic region for Echinococcus multilocularis. The clinical manifestation of the disease is dependent on the location, the size of the cyst and the development stage of the parasite. Considering the uncommon character of echinococcosis in Central Europe, especially such located in the areas outside the liver and lungs, the authors would like to present a case of coexistence in one patient of two EM foci in the liver and the head of the pancreas.

We present a clinical case of a 32-year-old man who was diagnosed with a cystic lesion with septa and calcification in the sixth segment of the liver and a suspicious change in the head of the pancreas. ELISA Em 2 plus test was positive, Western Blot method - the P-5 pattern showed an image that is characteristic of an EM infection. The sixth liver segment with a tumour and a tumour from the head of pancreas were excised by means of laparotomy. On the 6th day after the surgery the patient was discharged from hospital without complications and in good condition. Currently, he is under the control of a parasitic and zoonotic clinic. He takes an 800 mg daily dosage of Albendazole.

The presented clinical case shows that if we have a patient with cystic / tumour change in the pancreas and positive immunological tests, CT and MRI of the abdominal cavity are usually sufficient in order to fully diagnose and to qualify such a person for surgery. The most effective treatment is surgical treatment supplemented with pre- and postsurgical treatment with Albendazole.

• Echinococcus multilocularis
• Hydatis dsease
• Pancreas

Alveolar echinococcosis is a severe and rare helminthic disease with increasing incidence in endemic regions. Herein, available evidence on curative surgical and potential palliative approaches was reviewed. Such strategies have to be applied in the context of available resources in different health-care systems. Complete resection followed by adjuvant therapy remains the only curative treatment available. Curative surgery is performed by open or laparoscopic approach depending on the extent of the disease and the experience of the surgical team. Palliative resections are typically not indicated, because the availability of endoscopic treatments of biliary complications and long-term benzimidazoles represent efficient alternatives to surgery. Liver transplantation as an alternative to palliative surgery has not been shown to be superior to long-term conservative therapy. Immunosuppressive therapy might additionally contribute to fatal disease recurrence after transplantation.

•

Alveolar echinococcosis is an aggressive zoonotic infection caused by the parasite Echinococcus multilocularis.

• Alveolar echinococcosis
• Echinococcus multilocularis
• Interventional treatment
• Liver transplantation
• Resection
• Surgical strategies

• Echinococcus
• Echinococcus granulosus
• Echinococcus multilocularis
• alveolar echinococcosis
• cystic echinococcosis
• echinococcosis
• genetic epidemiology
• genome
• strains/genotypes
• transcriptome
• zoonosis

• Albendazole/therapeutic use
• Animals
• China/epidemiology
• Clinical Trials as Topic
• Cystectomy
• Disease Management
• Echinococcosis/epidemiology
• Echinococcosis/therapy
• Humans
• Quality of Life
• Transplantation, Autologous
• Zoonoses/epidemiology
• Zoonoses/parasitology
• Zoonoses/therapy

• ELISA
• alveolar echinococcosis
• immunoblotting
• rEm18
• serodiagnosis

• Alveolar echinococcosis
• Biomarker
• Circulating cell-free DNA
• Digital PCR

• Animals
• Cell-Free Nucleic Acids/blood
• Cell-Free Nucleic Acids/genetics
• DNA, Helminth/blood
• DNA, Helminth/genetics
• Echinococcosis/blood
• Echinococcosis/diagnosis
• Echinococcosis/parasitology
• Echinococcus multilocularis/genetics
• Echinococcus multilocularis/isolation & purification
• Gerbillinae
• Humans
• Real-Time Polymerase Chain Reaction/methods

• Alveolar echinococcosis
• Distribution
• Echinococcus multilocularis
• National database
• Prevalence

• Adult
• Aged
• Animals
• Databases, Factual
• Disease Notification
• Echinococcosis/epidemiology
• Echinococcus multilocularis
• Female
• Germany/epidemiology
• Humans
• Male
• Middle Aged
• Prevalence
• Young Adult

• No. 440/15 Deutsche Forschungsgemeinschaft

• Liver
• alveolar echinococcosis
• computed tomography perfusion imaging

• Alveolar echinococcosis
• Apparent diffusion coefficient (ADC)
• Diffusion magnetic resonance imaging
• Hepatocellular carcinoma
• Intrahepatic cholangiocarcinoma

• Adult
• Aged
• Carcinoma, Hepatocellular/diagnostic imaging
• Case-Control Studies
• Cholangiocarcinoma/diagnostic imaging
• Diffusion Magnetic Resonance Imaging
• Echinococcosis, Hepatic/diagnostic imaging
• Female
• Humans
• Liver Neoplasms/diagnostic imaging
• Male
• Middle Aged

• Algorithms
• Animals
• Biopsy
• Disease Models, Animal
• Echinococcosis, Hepatic/diagnostic imaging
• Echinococcosis, Hepatic/parasitology
• Echinococcosis, Hepatic/pathology
• Image Processing, Computer-Assisted
• Immunohistochemistry
• Male
• Models, Statistical
• Rats
• X-Ray Microtomography/methods

• National Natural Science Foundation of China

• Amoebic abscesses
• Echinococcal cyst
• Echinococcosis
• Liver abscesses

• Echinococcosis, Hepatic/classification
• Echinococcosis, Hepatic/diagnostic imaging
• Echinococcosis, Hepatic/therapy
• Humans
• Liver/diagnostic imaging
• Liver Abscess/classification
• Liver Abscess/diagnostic imaging
• Liver Abscess/therapy
• Ultrasonography/methods

• Alveolar echinococcosis
• Benzimidazoles
• Burden of echinococcoses
• Cystic echinococcosis
• Imaging procedures
• Liver transplantation
• Metacestode diseases
• Neotropical echinococcoses
• Percutaneous sterilization techniques
• Surgery
• Watch-and-wait

• Animals
• Echinococcosis/diagnosis
• Echinococcosis/parasitology
• Echinococcosis, Hepatic/diagnosis
• Echinococcosis, Hepatic/parasitology
• Echinococcus/isolation & purification
• Humans

• Echinococcus multilocularis
• Endoscopic retrograde cholangio-pancreatography (ERCP)
• alveolar echinococcosis (AE)
• biliary stent
• biliary stricture
• cholangitis

• Alveolar echinococcosis
• cerebral cystic lesions
• head-ache
• spinal

Background: This study retrospectively analyzed the clinical data, laboratory results, imaging findings, and histopathological features of 28 patients who underwent ultrasound-guided core-needle biopsy from a hepatic lesion and were diagnosed with alveolar echinococcosis. Results: Among 28 patients included in the study, 16 were females and 12 were males. The mean age of the studied population was 53 ± 16 years, and the age range was 18–79 years. The most common presenting symptom was abdominal pain, which was observed in 14 patients. A total of 36 lesions were detected in the patients’ livers, out of which 7 had a cystic appearance. Hepatic vascular involvement, bile duct involvement, and other organ involvement were depicted in 14, 5, and 7 patients, respectively. The average number of cores taken from the lesions was 2.7, ranging between 2 and 5. In histopathological evaluation, PAS+ parasitic membrane structures were visualized on a necrotic background in all cases. Regarding seven patients, who were operated, the pathological findings of preoperative percutaneous biopsies were in perfect agreement with the pathological examinations after surgical resections. None of the patients developed major complications after biopsy. Conclusion: Ultrasound-guided core-needle biopsy is a minimally invasive, reliable, and effective diagnostic tool for the definitive diagnosis of hepatic alveolar echinococcosis.

AIM: To establish a computed tomography (CT)-morphological classification for hepatic alveolar echinococcosis was the aim of the study.

METHODS: The CT morphology of hepatic lesions in 228 patients with confirmed alveolar echinococcosis (AE) drawn from the Echinococcus Databank of the University Hospital of Ulm was reviewed retrospectively. For this reason, CT datasets of combined positron emission tomography (PET)-CT examinations were evaluated. The diagnosis of AE was made in patients with unequivocal seropositivity; positive histological findings following diagnostic puncture or partial resection of the liver; and/or findings typical for AE at either ultrasonography, CT, magnetic resonance imaging or PET-CT. The CT-morphological findings were grouped into the new classification scheme.

RESULTS: Within the classification a lesion was dedicated to one out of five “primary morphologies” as well as to one out of six “patterns of calcification”. “primary morphology” and “pattern of calcification” are primarily focussed on separately from each other and combined, whereas the “primary morphology” V is not further characterized by a “pattern of calcification”. Based on the five primary morphologies, further descriptive sub-criteria were appended to types I-III. An analysis of the calcification pattern in relation to the primary morphology revealed the exclusive association of the central calcification with type IV primary morphology. Similarly, certain calcification patterns exhibited a clear predominance for other primary morphologies, which underscores the delimitation of the individual primary morphological types from each other. These relationships in terms of calcification patterns extend into the primary morphological sub-criteria, demonstrating the clear subordination of those criteria.

CONCLUSION: The proposed CT-morphological classification (EMUC-CT) is intended to facilitate the recognition and interpretation of lesions in hepatic alveolar echinococcosis. This could help to interpret different clinical courses better and shall assist in the context of scientific studies to improve the comparability of CT findings.

• Hepatic alveolar echinococcosis
• Diagnosis
• Echinococcus multilocularis
• Classification
• Computed tomography
• Alveolar echinococcosis

• Adult
• Aged
• Databases, Factual
• Echinococcosis, Hepatic/classification
• Echinococcosis, Hepatic/diagnostic imaging
• Echinococcosis, Hepatic/parasitology
• Female
• Germany
• Hospitals, University
• Humans
• Liver/diagnostic imaging
• Liver/parasitology
• Male
• Middle Aged
• Positron Emission Tomography Computed Tomography
• Predictive Value of Tests
• Prognosis
• Retrospective Studies
• Terminology as Topic

Biliary vessel pathology due to alveolar echicococcosis (AE) results in variable combinations of stenosis, necrosis and inflammation. Modern management strategies for patients with cholestasis are desperately needed. The aim is proof of principle of serial ERC (endoscopic retrograde cholangiography) balloon dilation for AE biliary pathology.

Retrospective case series of seven consecutive patients with AE-associated biliary pathology and ERC treatment in an interdisciplinary endoscopy unit at a University Hospital which hosts a national echinococcosis treatment center. The AE patient cohort consists of 106 patients with AE of the liver of which 13 presented with cholestasis. 6/13 received bilio-digestive anastomosis and 7/13 patients were treated by ERC and are reported here. Biliary stricture balloon dilation was performed with 18-Fr balloons at the initial and with 24-Fr balloons at subsequent interventions. If indicated 10 Fr plastic stents were placed.

Six patients were treated by repeated balloon dilation and stenting, one by stenting only. After an acute phase of 6 months with repeated balloon dilation, three patients showed “sustained clinical success” and four patients “assisted therapeutic success,” of which one has not yet reached the six month endpoint. In one patient, sustained success could not be achieved despite repeated insertion of plastic stents and balloon dilation, but with temporary insertion of a fully covered self-expanding metal stent (FCSEMS). There was no loss to follow up. No major complications were observed.

Serial endoscopic dilation is a standard tool in the treatment of benign biliary strictures. Serial endoscopic intervention with balloon dilation combined with benzimidazole treatment can re-establish and maintain biliary duct patency in AE associated pathology and probably contributes to avoid or postpone bilio-digestive anastomosis. This approach is in accordance with current ERC guidelines and is minimally disruptive for patients.

Alveolar echinococcosis (AE) is a zoonosis causing infiltrative liver lesions. A subgroup of patients presents with central liver lesions and biliary obstruction. At present there is no clear concept for the treatment of biliary obstruction in AE of the liver, and data from high quality trials to base treatment decisions on evidence are missing. In rare neglected infectious diseases with very low prevalence, clinical data can mainly be generated from case series. In our study we aim at the proof of principle of serial ERC balloon dilation for biliary pathology associated to alveolar echinococcosis. This approach is in accordance with current ERC guidelines and is minimally disruptive for patients.

• Adult
• Aged
• Angioplasty, Balloon
• Cholangiography
• Cholangiopancreatography, Endoscopic Retrograde
• Cholestasis/diagnosis
• Cholestasis/etiology
• Cholestasis/pathology
• Cholestasis/therapy
• Constriction, Pathologic/diagnosis
• Constriction, Pathologic/etiology
• Constriction, Pathologic/therapy
• Echinococcosis
• Echinococcosis, Hepatic/complications
• Female
• Humans
• Male
• Middle Aged
• Retrospective Studies