{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,18]],"date-time":"2026-09-18T22:45:31Z","timestamp":1789771531030,"version":"4.0.1"},"reference-count":53,"publisher":"The Company of Biologists","issue":"3","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2013,2,1]]},"abstract":"<jats:p>Congenital biliary atresia is an incurable disease of newborn infants, of unknown genetic causes, that results in congenital deformation of the gallbladder and biliary duct system. Here, we show that during mouse organogenesis, insufficient SOX17 expression in the gallbladder and bile duct epithelia results in congenital biliary atresia and subsequent acute \u2018embryonic hepatitis\u2019, leading to perinatal death in ~95% of the Sox17 heterozygote neonates in C57BL\/6 (B6) background mice. During gallbladder and bile duct development, Sox17 was expressed at the distal edge of the gallbladder primordium. In the Sox17+\/\u2212 B6 embryos, gallbladder epithelia were hypoplastic, and some were detached from the luminal wall, leading to bile duct stenosis or atresia. The shredding of the gallbladder epithelia is probably caused by cell-autonomous defects in proliferation and maintenance of the Sox17+\/\u2212 gallbladder\/bile duct epithelia. Our results suggest that Sox17 plays a dosage-dependent function in the morphogenesis and maturation of gallbladder and bile duct epithelia during the late-organogenic stages, highlighting a novel entry point to the understanding of the etiology and pathogenesis of human congenital biliary atresia.<\/jats:p>","DOI":"10.1242\/dev.086702","type":"journal-article","created":{"date-parts":[[2013,1,4]],"date-time":"2013-01-04T23:06:55Z","timestamp":1357340815000},"page":"639-648","source":"Crossref","is-referenced-by-count":70,"title":["<i>Sox17<\/i> haploinsufficiency results in perinatal biliary atresia and hepatitis in C57BL\/6 background mice"],"prefix":"10.1242","volume":"140","author":[{"given":"Mami","family":"Uemura","sequence":"first","affiliation":[{"name":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"},{"name":"Center for Experimental Animal, Tokyo Medical and Dental University, Yushima 1-5-45, Bunkyo-ku, Tokyo 113-8510, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aisa","family":"Ozawa","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takumi","family":"Nagata","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kaoruko","family":"Kurasawa","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naoki","family":"Tsunekawa","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ikuo","family":"Nobuhisa","sequence":"additional","affiliation":[{"name":"Department of Stem Cell Regulation, Medical Research Institute, Tokyo Medical and Dental University, Yushima 1-5-45, Bunkyo-ku, Tokyo 113-8510, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tetsuya","family":"Taga","sequence":"additional","affiliation":[{"name":"Department of Stem Cell Regulation, Medical Research Institute, Tokyo Medical and Dental University, Yushima 1-5-45, Bunkyo-ku, Tokyo 113-8510, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kenshiro","family":"Hara","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"},{"name":"Division of Germ Cell Biology, National Institute for Basic Biology and Department of Basic Biology, School of Life Science, Graduate University for Advanced Studies (SOKENDAI), Okazaki, Aichi 444-8585, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Akihiko","family":"Kudo","sequence":"additional","affiliation":[{"name":"Department of Anatomy, Kyorin University School of Medicine, Mitaka, Tokyo 181-8611, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hayato","family":"Kawakami","sequence":"additional","affiliation":[{"name":"Department of Anatomy, Kyorin University School of Medicine, Mitaka, Tokyo 181-8611, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yukio","family":"Saijoh","sequence":"additional","affiliation":[{"name":"Department of Neurobiology and Anatomy, The University of Utah, Salt Lake City, UT 84132-3401, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masamichi","family":"Kurohmaru","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masami","family":"Kanai-Azuma","sequence":"additional","affiliation":[{"name":"Center for Experimental Animal, Tokyo Medical and Dental University, Yushima 1-5-45, Bunkyo-ku, Tokyo 113-8510, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoshiakira","family":"Kanai","sequence":"additional","affiliation":[{"name":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","reference":[{"key":"2021042607343912700_R1","doi-asserted-by":"crossref","first-page":"393","DOI":"10.1016\/j.ydbio.2010.12.007","article-title":"The primitive endoderm lineage of the mouse blastocyst: sequential transcription factor activation and regulation of differentiation by Sox17","volume":"350","author":"Artus","year":"2011","journal-title":"Dev. 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