Date Published: February 24, 2016
Publisher: Public Library of Science
Author(s): Marija Stojkovic, Thomas Junghanss, Mira Veeser, Tim F. Weber, Peter Sauer, Hector H Garcia. http://doi.org/10.1371/journal.pntd.0004278
Abstract: Background and AimsBiliary 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.MethodsRetrospective 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.ResultsSix 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.ConclusionsSerial 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.
Partial Text: Alveolar echinococcosis (AE) is a parasitic disease characterised by liver lesions with infiltrative growth comparable to malignancies. On radiological imaging microcystic honeycomb like lesions are considered characteristic but solid tumors and necrotic cavities (‘pseudocysts’) are also frequently seen which brings solid and cystic differential diagnoses into play. With continuous benzimindazole treatment the 10 year survival rate is excellent for patients with non-curatively resectable AE lesions.
Between 2006 and 2015 seven consecutive patients with hepatic AE and biliary infiltration with cholangitis were referred for endoscopic treatment. All patients presented with jaundice. Patient characteristics, pre-referral treatment and biliary duct pathology is summarized in Table 1. Table 2 and Fig 1 summarize laboratory results, types and frequency of interventions, outcome and follow-up. Figs 2–4 illustrate imaging and ERC findings.
Alveolar echinococcosis (AE) of the biliary tree is characterised by destruction of biliary vessels due to infiltrative, malignancy like growth of AE liver lesions. In general, growth of AE can be halted with albendazole, a benzimidazole with parasitostatic effect. ERC is the treatment of choice for benign biliary strictures and general ERC treatment recommendations are being applied in AE patients . Our series shows that serial endoscopic balloon dilation and stenting combined with benzimidazole treatment can re-establish and maintain biliary duct patency for many years. Thus, interventions with a more profound impact on the quality of life such as percutaneous bile drainage can be avoided or postponed. Well established ERC management for AE patients could possibly even postpone liver transplantation which is highly problematic in this entity as immunosuppression favors re-growth of non-resected or non-resectable AE components and spread with distant metastases.