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The <jats:italic>Sox17\u2010<\/jats:italic>haploinsufficient (<jats:italic>Sox17<\/jats:italic><jats:sup>+\/\u2212<\/jats:sup>) mouse is an animal model of BA that encompasses bile duct injury and subsequent BA\u2010like inflammation by the neonatal stage. Most <jats:italic>Sox17<\/jats:italic><jats:sup>+\/\u2212<\/jats:sup> neonates die soon after birth, but some <jats:italic>Sox17<\/jats:italic><jats:sup>+\/\u2212<\/jats:sup> pups reach adulthood and have a normal life span, unlike human BA. However, the phenotype and BA\u2010derived scars in the hepatobiliary organs of surviving <jats:italic>Sox17<\/jats:italic><jats:sup>+\/\u2212<\/jats:sup> mice are unknown. Here, we examined the phenotypes of the hepatobiliary organs in post\u2010weaning and young adult <jats:italic>Sox17<\/jats:italic><jats:sup><jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> mice. The results confirmed the significant reduction in liver weight, together with peripheral calcinosis and aberrant vasculature in the hepatic lobule, in surviving <jats:italic>Sox17<\/jats:italic><jats:sup><jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> mice as compared with their wild\u2010type (WT) littermates. Such hepatic phenotypes may be sequelae of hepatobiliary damage at the fetal and neonatal stages, a notion supported by the slight, but significant, increases in the levels of serum markers of liver damage in adult <jats:italic>Sox17<\/jats:italic><jats:sup><jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> mice. The surviving <jats:italic>Sox17<\/jats:italic><jats:sup><jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> mice had a shorter gallbladder in which ectopic hepatic ducts were more frequent compared to WT mice. Also, the surviving <jats:italic>Sox17<\/jats:italic><jats:sup><jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> mice showed neither obstruction of the EHBDs nor atrophy or inflammation of hepatocytes or the intrahepatic ducts. These data suggest that some <jats:italic>Sox17<\/jats:italic><jats:sup><jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> pups with BA naturally escape lethality and recover from fetal hepatobiliary damages during the perinatal period, highlighting the usefulness of the in vivo model in understanding the hepatobiliary healing processes after surgical restoration of bile flow in human BA.<\/jats:p>","DOI":"10.1002\/ar.24466","type":"journal-article","created":{"date-parts":[[2020,6,1]],"date-time":"2020-06-01T07:29:22Z","timestamp":1590996562000},"page":"3096-3107","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":11,"title":["Anatomical and histological characteristics of the hepatobiliary system in adult <i>Sox17<\/i> heterozygote mice"],"prefix":"10.1002","volume":"303","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0975-5915","authenticated-orcid":false,"given":"Montri","family":"Pattarapanawan","sequence":"first","affiliation":[{"name":"Department of Veterinary Anatomy The University of Tokyo  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mami","family":"Uemura","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy The University of Tokyo  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nanae","family":"Miyazaki","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy The University of Tokyo  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shohei","family":"Takami","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy The University of Tokyo  Tokyo Japan"},{"name":"Department of Pediatric Surgery The University of Tokyo  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hirotaka","family":"Tomiyasu","sequence":"additional","affiliation":[{"name":"Department of Veterinary Internal Medicine The University of Tokyo  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naoki","family":"Tsunekawa","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy The University of Tokyo  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoshikazu","family":"Hirate","sequence":"additional","affiliation":[{"name":"Center of Experimental Animal Tokyo Medical and Dental University  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jun","family":"Fujishiro","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery The University of Tokyo  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masamichi","family":"Kurohmaru","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy The University of Tokyo  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masami","family":"Kanai\u2010Azuma","sequence":"additional","affiliation":[{"name":"Center of Experimental Animal Tokyo Medical and Dental University  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroki","family":"Higashiyama","sequence":"additional","affiliation":[{"name":"Department of Physiological Chemistry and Metabolism The University of Tokyo  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoshiakira","family":"Kanai","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy The University of Tokyo  Tokyo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,7,2]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3468(78)80462-X"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrgastro.2015.74"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/hep.510230652"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clinre.2012.03.017"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/hep.29905"},{"key":"e_1_2_7_7_1","first-page":"170","article-title":"The long\u2010term outcome of the Kasai operation in patients with biliary atresia: A systematic review","volume":"71","author":"Bijl E. 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