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This study reports the inaugural human infection of echinococcosis caused by the Mongolian genotype of <jats:italic>E. multilocularis<\/jats:italic> in China, also the first reported indigenous AE case in Inner Mongolia. <\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Case presentation<\/jats:title>\n            <jats:p>A 58-year-old female pastoralist from Inner Mongolia, who had no endemic region exposure history but prolonged occupational contact with dogs, presented with severe AE. Clinical examinations revealed a massive hepatic lesion exceeding 10\u00a0cm in diameter, accompanied by elevated eosinophils (0.90\u2009\u00d7\u200910<jats:sup>9<\/jats:sup>\/L) and basophils (0.08\u2009\u00d7\u200910<jats:sup>9<\/jats:sup>\/L). Despite undergoing liver transplantation, the patient succumbed postoperatively. Histopathological confirmation and molecular phylogenetics identified the Mongolian genotype of <jats:italic>E. multilocularis<\/jats:italic> infection, distinct from the predominant Asian genotype in China. Potential evidence of zoonotic transmission was discovered through genotype-matched <jats:italic>E. multilocularis<\/jats:italic> detection in corsac fox (<jats:italic>Vulpes corsac<\/jats:italic>) feces from the grasslands along the shores of Hulun Lake (Hulun Buir City, northeastern Inner Mongolia, China).<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Conclusions<\/jats:title>\n            <jats:p>This report provides the primary evidence of a locally acquired human AE infection in China caused by the Mongolian genotype of <jats:italic>Echinococcus multilocularis<\/jats:italic>. The discovery of this case challenges historical classifications of echinococcosis endemic areas. The findings call for revised AE-endemic identification criteria, improved AE diagnostic protocols, and enhanced AE surveillance in the Inner Mongolia region to generate further epidemiological evidence and information on disease progression.<\/jats:p>\n          <\/jats:sec>","DOI":"10.1186\/s40249-025-01342-4","type":"journal-article","created":{"date-parts":[[2025,7,24]],"date-time":"2025-07-24T07:52:18Z","timestamp":1753343538000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Clinical confirmation of an infection\u00a0with Echinococcus multilocularis\u00a0(Mongolian genotype): first case report of human alveolar echinococcosis in Inner Mongolia, China"],"prefix":"10.1186","volume":"14","author":[{"given":"Xu","family":"Wang","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhan-Jun","family":"Xiao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chui-Zhao","family":"Xue","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wen-Ting","family":"Wu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jiang-Hui","family":"Yang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chun","family":"Yan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ying","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yan","family":"Kui","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wen-Bo","family":"Luo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xi","family":"Du","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Run-Na","family":"Zan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rong-Jian","family":"Shang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sa","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rigen","family":"Na","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-2729-9642","authenticated-orcid":false,"given":"Shuai","family":"Han","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shi-Zhu","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2025,7,24]]},"reference":[{"issue":"11","key":"1342_CR1","doi-asserted-by":"publisher","first-page":"1243","DOI":"10.4254\/wjh.v16.i11.1243","volume":"16","author":"QD Jing","year":"2024","unstructured":"Jing QD, Ji-De A, Liu LX, Fan HN. 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