{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,20]],"date-time":"2026-09-20T08:14:25Z","timestamp":1789892065657,"version":"4.0.1"},"update-to":[{"DOI":"10.1371\/journal.pntd.0010146","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2022,2,9]],"date-time":"2022-02-09T00:00:00Z","timestamp":1644364800000}}],"reference-count":25,"publisher":"Public Library of Science (PLoS)","issue":"1","license":[{"start":{"date-parts":[[2022,1,28]],"date-time":"2022-01-28T00:00:00Z","timestamp":1643328000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Fontana Foundation"}],"content-domain":{"domain":["www.plosntds.org"],"crossmark-restriction":false},"short-container-title":["PLoS Negl Trop Dis"],"abstract":"<jats:sec id=\"sec001\">\n<jats:title>Objectives<\/jats:title>\n<jats:p>Alveolar echinococcosis (AE) is an orphan zoonosis of increasing concern in endemic areas, including Europe. It frequently presents in an advanced, inoperable stage, that requires life-long parasitostatic benzimidazole therapy. In some patients, long-term therapy leads to negative anti-Em18 antibody ELISA and PET. It is disputed, whether these patients are truly cured and treatment can be safely discontinued. Our aim was to retrospectively assess long-term outcome of 34 patients with inoperable AE who participated in a previous study to determine feasibility of benzimidazole treatment cessation.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec002\">\n<jats:title>Methods<\/jats:title>\n<jats:p>Retrospective analysis of medical charts was undertaken in all 34 AE patients who participated in our previous study. Of particular interest were AE recurrence or other reasons for re-treatment in patients who stopped benzimidazole therapy and whether baseline clinical and laboratory parameters help identify of patients that might qualifiy for treatment cessation. Additionally, volumetric measurement of AE lesions on contrast-enhanced cross-sectional imaging was performed at baseline and last follow-up in order to quantify treatment response.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec003\">\n<jats:title>Results<\/jats:title>\n<jats:p>12 of 34 patients stopped benzimidazole therapy for a median of 131 months. 11 of these patients showed stable or regressive AE lesions as determined by volumetric measurement. One patient developed progressive lesions with persistently negative anti-Em18 antibody ELISA but slight FDG-uptake in repeated PET imaging. At baseline, patients who met criteria for treatment cessation demonstrated higher lymphocyte count and lower total IgE.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec004\">\n<jats:title>Conclusion<\/jats:title>\n<jats:p>Treatment cessation is feasible in inoperable AE patients, who demonstrate negative anti-Em18 antibody ELISA and PET on follow-up. Close monitoring including sectional imaging is strongly advised.<\/jats:p>\n<\/jats:sec>","DOI":"10.1371\/journal.pntd.0010146","type":"journal-article","created":{"date-parts":[[2022,1,28]],"date-time":"2022-01-28T18:36:11Z","timestamp":1643394971000},"page":"e0010146","update-policy":"https:\/\/doi.org\/10.1371\/journal.pntd.corrections_policy","source":"Crossref","is-referenced-by-count":18,"title":["Evaluation of a structured treatment discontinuation in patients with inoperable alveolar echinococcosis on long-term benzimidazole therapy: A\u00a0retrospective cohort study"],"prefix":"10.1371","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6208-9694","authenticated-orcid":true,"given":"Ansgar","family":"Deibel","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1230-092X","authenticated-orcid":true,"given":"Daniel","family":"Stocker","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cordula","family":"Meyer zu Schwabedissen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lars","family":"Husmann","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1968-6543","authenticated-orcid":true,"given":"Philipp Andreas","family":"Kronenberg","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2685-1167","authenticated-orcid":true,"given":"Felix","family":"Grimm","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter","family":"Deplazes","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C\u00e4cilia S.","family":"Reiner","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9020-8192","authenticated-orcid":true,"given":"Beat","family":"M\u00fcllhaupt","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"340","published-online":{"date-parts":[[2022,1,28]]},"reference":[{"issue":"3","key":"pntd.0010146.ref001","doi-asserted-by":"crossref","first-page":"343","DOI":"10.3201\/eid0903.020341","article-title":"European echinococcosis registry: human alveolar echinococcosis, Europe, 1982\u20132000","volume":"9","author":"P Kern","year":"2003","journal-title":"Emerging infectious diseases"},{"issue":"9","key":"pntd.0010146.ref002","doi-asserted-by":"crossref","first-page":"407","DOI":"10.1016\/j.pt.2015.06.001","article-title":"Threat of alveolar echinococcosis to public health\u2014a challenge for Europe","volume":"31","author":"B Gottstein","year":"2015","journal-title":"Trends in parasitology"},{"issue":"1","key":"pntd.0010146.ref003","doi-asserted-by":"crossref","first-page":"107","DOI":"10.1128\/CMR.17.1.107-135.2004","article-title":"Biological, epidemiological, and clinical aspects of echinococcosis, a zoonosis of increasing concern","volume":"17","author":"J Eckert","year":"2004","journal-title":"Clinical microbiology reviews"},{"issue":"3","key":"pntd.0010146.ref004","doi-asserted-by":"crossref","first-page":"655","DOI":"10.1016\/S0889-8553(05)70268-5","article-title":"Cestodes. 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