{"@context":{"@vocab":"https://cir.nii.ac.jp/schema/1.0/","rdfs":"http://www.w3.org/2000/01/rdf-schema#","dc":"http://purl.org/dc/elements/1.1/","dcterms":"http://purl.org/dc/terms/","foaf":"http://xmlns.com/foaf/0.1/","prism":"http://prismstandard.org/namespaces/basic/2.0/","cinii":"http://ci.nii.ac.jp/ns/1.0/","datacite":"https://schema.datacite.org/meta/kernel-4/","ndl":"http://ndl.go.jp/dcndl/terms/","jpcoar":"https://github.com/JPCOAR/schema/blob/master/2.0/"},"@id":"https://cir.nii.ac.jp/crid/1360565169045028992.json","@type":"Article","productIdentifier":[{"identifier":{"@type":"DOI","@value":"10.1242/dev.086702"}},{"identifier":{"@type":"URI","@value":"http://journals.biologists.com/dev/article-pdf/140/3/639/1163030/639.pdf"}},{"identifier":{"@type":"PMID","@value":"23293295"}}],"resourceType":"学術雑誌論文(journal article)","dc:title":[{"@value":"<i>Sox17</i> haploinsufficiency results in perinatal biliary atresia and hepatitis in C57BL/6 background mice"}],"description":[{"type":"abstract","notation":[{"@value":"<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 ‘embryonic hepatitis’, 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+/− 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+/− 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>"}]}],"creator":[{"@id":"https://cir.nii.ac.jp/crid/1380569413806161159","@type":"Researcher","foaf:name":[{"@value":"Aisa Ozawa"}],"jpcoar:affiliationName":[{"@value":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}]},{"@id":"https://cir.nii.ac.jp/crid/1380569413806160902","@type":"Researcher","foaf:name":[{"@value":"Takumi Nagata"}],"jpcoar:affiliationName":[{"@value":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}]},{"@id":"https://cir.nii.ac.jp/crid/1380569413806161163","@type":"Researcher","foaf:name":[{"@value":"Kaoruko Kurasawa"}],"jpcoar:affiliationName":[{"@value":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}]},{"@id":"https://cir.nii.ac.jp/crid/1420845751150490752","@type":"Researcher","personIdentifier":[{"@type":"KAKEN_RESEARCHERS","@value":"40332879"},{"@type":"NRID","@value":"1000040332879"},{"@type":"NRID","@value":"9000001709348"},{"@type":"NRID","@value":"9000019090304"},{"@type":"NRID","@value":"9000261008348"},{"@type":"RESEARCHMAP","@value":"https://researchmap.jp/read0068912"}],"foaf:name":[{"@value":"Ikuo Nobuhisa"}],"jpcoar:affiliationName":[{"@value":"Department of Stem Cell Regulation, Medical Research Institute, Tokyo Medical and Dental University, Yushima 1-5-45, Bunkyo-ku, Tokyo 113-8510, 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Tokyo Medical and Dental University, Yushima 1-5-45, Bunkyo-ku, Tokyo 113-8510, Japan"}]},{"@id":"https://cir.nii.ac.jp/crid/1420564276176378752","@type":"Researcher","personIdentifier":[{"@type":"KAKEN_RESEARCHERS","@value":"60551546"},{"@type":"NRID","@value":"1000060551546"},{"@type":"NRID","@value":"9000410476081"},{"@type":"NRID","@value":"9000258010305"},{"@type":"NRID","@value":"9000410151268"},{"@type":"NRID","@value":"9000001499207"},{"@type":"NRID","@value":"9000410476542"},{"@type":"NRID","@value":"9000403545426"},{"@type":"NRID","@value":"9000413480396"},{"@type":"NRID","@value":"9000345399803"},{"@type":"NRID","@value":"9000006861835"},{"@type":"NRID","@value":"9000283894112"},{"@type":"NRID","@value":"9000392065593"},{"@type":"NRID","@value":"9000398558587"},{"@type":"NRID","@value":"9000413359160"},{"@type":"NRID","@value":"9000392061143"},{"@type":"NRID","@value":"9000402262285"},{"@type":"NRID","@value":"9000392061175"},{"@type":"NRID","@value":"9000392058912"},{"@type":"NRID","@value":"9000392067267"},{"@type":"NRID","@value":"9000347224630"},{"@type":"NRID","@value":"9000347162128"},{"@type":"NRID","@value":"9000368937095"},{"@type":"NRID","@value":"9000347224819"},{"@type":"NRID","@value":"9000392069061"},{"@type":"NRID","@value":"9000411513936"},{"@type":"NRID","@value":"9000347362018"},{"@type":"NRID","@value":"9000021078193"},{"@type":"RESEARCHMAP","@value":"https://researchmap.jp/haraken"}],"foaf:name":[{"@value":"Kenshiro Hara"}],"jpcoar:affiliationName":[{"@value":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"},{"@value":"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"}]},{"@id":"https://cir.nii.ac.jp/crid/1380569413806161040","@type":"Researcher","foaf:name":[{"@value":"Akihiko Kudo"}],"jpcoar:affiliationName":[{"@value":"Department of Anatomy, Kyorin University School of Medicine, Mitaka, Tokyo 181-8611, Japan"}]},{"@id":"https://cir.nii.ac.jp/crid/1380569413806160773","@type":"Researcher","foaf:name":[{"@value":"Hayato Kawakami"}],"jpcoar:affiliationName":[{"@value":"Department of Anatomy, Kyorin University School of Medicine, Mitaka, Tokyo 181-8611, Japan"}]},{"@id":"https://cir.nii.ac.jp/crid/1380569413806161036","@type":"Researcher","foaf:name":[{"@value":"Yukio Saijoh"}],"jpcoar:affiliationName":[{"@value":"Department of Neurobiology and Anatomy, The University of Utah, Salt Lake City, UT 84132-3401, USA"}]},{"@id":"https://cir.nii.ac.jp/crid/1380569413806161039","@type":"Researcher","foaf:name":[{"@value":"Masamichi Kurohmaru"}],"jpcoar:affiliationName":[{"@value":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}]},{"@id":"https://cir.nii.ac.jp/crid/1380569413806161044","@type":"Researcher","foaf:name":[{"@value":"Masami Kanai-Azuma"}],"jpcoar:affiliationName":[{"@value":"Center for Experimental Animal, Tokyo Medical and Dental University, Yushima 1-5-45, Bunkyo-ku, Tokyo 113-8510, Japan"}]},{"@id":"https://cir.nii.ac.jp/crid/1420845751138209792","@type":"Researcher","personIdentifier":[{"@type":"KAKEN_RESEARCHERS","@value":"30260326"},{"@type":"NRID","@value":"1000030260326"},{"@type":"NRID","@value":"9000000810924"},{"@type":"NRID","@value":"9000253282317"},{"@type":"NRID","@value":"9000009971527"},{"@type":"NRID","@value":"9000415337645"},{"@type":"NRID","@value":"9000415338236"},{"@type":"RESEARCHMAP","@value":"https://researchmap.jp/read0123258"}],"foaf:name":[{"@value":"Yoshiakira Kanai"}],"jpcoar:affiliationName":[{"@value":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"}]},{"@id":"https://cir.nii.ac.jp/crid/1380569413806160909","@type":"Researcher","foaf:name":[{"@value":"Mami Uemura"}],"jpcoar:affiliationName":[{"@value":"Department of Veterinary Anatomy, The University of Tokyo, Yayoi 1-1-1, Bunkyo-ku, Tokyo 113-8657, Japan"},{"@value":"Center for Experimental Animal, Tokyo Medical and Dental University, Yushima 1-5-45, Bunkyo-ku, Tokyo 113-8510, Japan"}]}],"publication":{"publicationIdentifier":[{"@type":"EISSN","@value":"14779129"},{"@type":"PISSN","@value":"09501991"}],"prism:publicationName":[{"@value":"Development"}],"dc:publisher":[{"@value":"The Company of Biologists"}],"prism:publicationDate":"2013-02-01","prism:volume":"140","prism:number":"3","prism:startingPage":"639","prism:endingPage":"648"},"reviewed":"false","url":[{"@id":"http://journals.biologists.com/dev/article-pdf/140/3/639/1163030/639.pdf"}],"createdAt":"2013-01-04","modifiedAt":"2021-04-26","foaf:topic":[{"@id":"https://cir.nii.ac.jp/all?q=Male","dc:title":"Male"},{"@id":"https://cir.nii.ac.jp/all?q=Heterozygote","dc:title":"Heterozygote"},{"@id":"https://cir.nii.ac.jp/all?q=Time%20Factors","dc:title":"Time Factors"},{"@id":"https://cir.nii.ac.jp/all?q=Haploinsufficiency","dc:title":"Haploinsufficiency"},{"@id":"https://cir.nii.ac.jp/all?q=Hepatitis,%20Animal","dc:title":"Hepatitis, Animal"},{"@id":"https://cir.nii.ac.jp/all?q=Epithelium","dc:title":"Epithelium"},{"@id":"https://cir.nii.ac.jp/all?q=Mice","dc:title":"Mice"},{"@id":"https://cir.nii.ac.jp/all?q=Biliary%20Atresia","dc:title":"Biliary Atresia"},{"@id":"https://cir.nii.ac.jp/all?q=Pregnancy","dc:title":"Pregnancy"},{"@id":"https://cir.nii.ac.jp/all?q=HMGB%20Proteins","dc:title":"HMGB Proteins"},{"@id":"https://cir.nii.ac.jp/all?q=SOXF%20Transcription%20Factors","dc:title":"SOXF Transcription Factors"},{"@id":"https://cir.nii.ac.jp/all?q=Animals","dc:title":"Animals"},{"@id":"https://cir.nii.ac.jp/all?q=Cell%20Proliferation","dc:title":"Cell Proliferation"},{"@id":"https://cir.nii.ac.jp/all?q=Mice,%20Inbred%20ICR","dc:title":"Mice, Inbred ICR"},{"@id":"https://cir.nii.ac.jp/all?q=Cholestasis","dc:title":"Cholestasis"},{"@id":"https://cir.nii.ac.jp/all?q=Gallbladder","dc:title":"Gallbladder"},{"@id":"https://cir.nii.ac.jp/all?q=Gene%20Expression%20Regulation,%20Developmental","dc:title":"Gene Expression Regulation, Developmental"},{"@id":"https://cir.nii.ac.jp/all?q=Embryo,%20Mammalian","dc:title":"Embryo, Mammalian"},{"@id":"https://cir.nii.ac.jp/all?q=Endoplasmic%20Reticulum%20Stress","dc:title":"Endoplasmic Reticulum Stress"},{"@id":"https://cir.nii.ac.jp/all?q=Immunohistochemistry","dc:title":"Immunohistochemistry"},{"@id":"https://cir.nii.ac.jp/all?q=Mice,%20Inbred%20C57BL","dc:title":"Mice, Inbred C57BL"},{"@id":"https://cir.nii.ac.jp/all?q=Animals,%20Newborn","dc:title":"Animals, Newborn"},{"@id":"https://cir.nii.ac.jp/all?q=Liver","dc:title":"Liver"},{"@id":"https://cir.nii.ac.jp/all?q=Hepatocytes","dc:title":"Hepatocytes"},{"@id":"https://cir.nii.ac.jp/all?q=Female","dc:title":"Female"},{"@id":"https://cir.nii.ac.jp/all?q=Bile%20Ducts","dc:title":"Bile Ducts"}],"project":[{"@id":"https://cir.nii.ac.jp/crid/1040000782066441600","@type":"Project","projectIdentifier":[{"@type":"KAKEN","@value":"21248034"},{"@type":"JGN","@value":"JP21248034"},{"@type":"URI","@value":"https://kaken.nii.ac.jp/grant/KAKENHI-PROJECT-21248034/"}],"notation":[{"@language":"ja","@value":"内胚葉形成因子SOX17の肝臓、胆管、膵臓発生における役割"},{"@language":"en","@value":"Crucial roles of SOX17 in liver-gallbladder-pancreas development"}]},{"@id":"https://cir.nii.ac.jp/crid/1040000782191965056","@type":"Project","projectIdentifier":[{"@type":"KAKEN","@value":"24228005"},{"@type":"JGN","@value":"JP24228005"},{"@type":"URI","@value":"https://kaken.nii.ac.jp/grant/KAKENHI-PROJECT-24228005/"}],"notation":[{"@language":"ja","@value":"胆嚢・胆管の形態形成・再生能と先天性疾患の分子機構の解明"},{"@language":"en","@value":"Morphogenesis, regeneration and congenital disease in gallbladder and bile duct system in 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