{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T08:51:31Z","timestamp":1791967891534,"version":"4.3.4"},"reference-count":48,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T00:00:00Z","timestamp":1790812800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T00:00:00Z","timestamp":1790812800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T00:00:00Z","timestamp":1787702400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100019533","name":"Moderna","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100019533","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000862","name":"Doris Duke Charitable Foundation Inc","doi-asserted-by":"publisher","award":["2019081"],"award-info":[{"award-number":["2019081"]}],"id":[{"id":"10.13039\/100000862","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000062","name":"National Institute of Diabetes and Digestive and Kidney Diseases","doi-asserted-by":"publisher","award":["DK099257"],"award-info":[{"award-number":["DK099257"]}],"id":[{"id":"10.13039\/100000062","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","thelancet.com","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["eBioMedicine"],"published-print":{"date-parts":[[2026,10]]},"DOI":"10.1016\/j.ebiom.2026.106474","type":"journal-article","created":{"date-parts":[[2026,9,11]],"date-time":"2026-09-11T21:15:59Z","timestamp":1789161359000},"page":"106474","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":0,"special_numbering":"C","title":["Liver disease reveals KIF12 as a regulator of mitochondria, lysosome and cilia localisation in human cholangiocytes"],"prefix":"10.1016","volume":"132","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4316-5177","authenticated-orcid":false,"given":"Anubha","family":"Seth","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1948-5103","authenticated-orcid":false,"given":"Joseph","family":"Brancale","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3529-8025","authenticated-orcid":false,"given":"Nina","family":"Dashti-Gibson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rodrigo M.","family":"Florentino","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lanuza A.P.","family":"Faccioli","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhenghao","family":"Liu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chigoziri","family":"Konkwo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-0232-8971","authenticated-orcid":false,"given":"Hyunseok","family":"Hong","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alejandro","family":"Soto-Gutierrez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Silvia","family":"Vilarinho","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"issue":"6","key":"10.1016\/j.ebiom.2026.106474_bib1","doi-asserted-by":"crossref","first-page":"1627","DOI":"10.1002\/hep.32437","article-title":"Cholestatic liver diseases of genetic etiology: advances and controversies","volume":"75","author":"Ibrahim","year":"2022","journal-title":"Hepatology"},{"issue":"4","key":"10.1016\/j.ebiom.2026.106474_bib2","doi-asserted-by":"crossref","first-page":"471","DOI":"10.1002\/hep4.1320","article-title":"Recessive mutations in KIF12 cause high gamma-glutamyltransferase cholestasis","volume":"3","author":"Unlusoy Aksu","year":"2019","journal-title":"Hepatol Commun"},{"issue":"5","key":"10.1016\/j.ebiom.2026.106474_bib3","doi-asserted-by":"crossref","first-page":"1164","DOI":"10.1038\/s41436-018-0288-x","article-title":"Identification of novel loci for pediatric cholestatic liver disease defined by KIF12, PPM1F, USP53, LSR, and WDR83OS pathogenic variants","volume":"21","author":"Maddirevula","year":"2019","journal-title":"Genet Med"},{"key":"10.1016\/j.ebiom.2026.106474_bib4","doi-asserted-by":"crossref","first-page":"284","DOI":"10.1016\/j.jpeds.2021.09.019","article-title":"KIF12 variants and disturbed hepatocyte polarity in children with a phenotypic spectrum of cholestatic liver disease","volume":"240","author":"Stalke","year":"2022","journal-title":"J Pediatr"},{"issue":"7","key":"10.1016\/j.ebiom.2026.106474_bib5","doi-asserted-by":"crossref","first-page":"733","DOI":"10.1007\/s12098-023-04914-0","article-title":"Cholestatic liver disease in a child with KIF12 mutation","volume":"91","author":"Samanta","year":"2024","journal-title":"Indian J Pediatr"},{"issue":"1","key":"10.1016\/j.ebiom.2026.106474_bib6","doi-asserted-by":"crossref","first-page":"106","DOI":"10.1111\/cge.14444","article-title":"Clinical exome sequencing reveals a novel pathogenic variant in KIF12 underlying cholestasis with highly variable phenotypes","volume":"105","author":"Waheed","year":"2024","journal-title":"Clin Genet"},{"issue":"3","key":"10.1016\/j.ebiom.2026.106474_bib7","doi-asserted-by":"crossref","first-page":"224","DOI":"10.1111\/cge.14524","article-title":"Kinesin family member 12-related hepatopathy: a generally indolent disorder with elevated gamma-glutamyl-transferase activity","volume":"106","author":"Vogel","year":"2024","journal-title":"Clin Genet"},{"issue":"2","key":"10.1016\/j.ebiom.2026.106474_bib8","doi-asserted-by":"crossref","first-page":"661","DOI":"10.1007\/s12072-023-10553-6","article-title":"Diagnostic yield and novel candidate genes by next generation sequencing in 166 children with intrahepatic cholestasis","volume":"18","author":"Zheng","year":"2024","journal-title":"Hepatol Int"},{"key":"10.1016\/j.ebiom.2026.106474_bib9","article-title":"Transcript selection for the genetic diagnosis of KIF12-associated progressive familial intrahepatic cholestasis","volume":"12","author":"Cheng","year":"2024","journal-title":"Gastroenterol Rep (Oxf)"},{"issue":"5","key":"10.1016\/j.ebiom.2026.106474_bib10","doi-asserted-by":"crossref","DOI":"10.1111\/petr.14804","article-title":"Youngest living donor liver transplant for end-stage liver disease in a 6-Month-Old with a novel aggressive mutation in KIF12 gene","volume":"28","author":"Gautam","year":"2024","journal-title":"Pediatr Transplant"},{"key":"10.1016\/j.ebiom.2026.106474_bib11","article-title":"A case of progressive familial intrahepatic cholestasis 8 associated with KIF12 gene mutation in a 7-Year-Old girl","volume":"13","author":"Mangalath","year":"2023","journal-title":"J Clin Exp Hepatol"},{"issue":"7","key":"10.1016\/j.ebiom.2026.106474_bib12","doi-asserted-by":"crossref","first-page":"1049","DOI":"10.1111\/jpc.70072","article-title":"Neonatal cholestasis: exploring genetic causes and clinical outcomes","volume":"61","author":"Gurcan Kaya","year":"2025","journal-title":"J Paediatr Child Health"},{"issue":"2","key":"10.1016\/j.ebiom.2026.106474_bib13","doi-asserted-by":"crossref","first-page":"202","DOI":"10.1016\/j.devcel.2014.08.028","article-title":"Antioxidant signaling involving the microtubule motor KIF12 is an intracellular target of nutrition excess in beta cells","volume":"31","author":"Yang","year":"2014","journal-title":"Dev Cell"},{"issue":"6","key":"10.1016\/j.ebiom.2026.106474_bib14","doi-asserted-by":"crossref","first-page":"1608","DOI":"10.1038\/s44318-025-00366-8","article-title":"Mutations in the kinesin KIF12 promote MASH in humans and mice by disrupting lipogenic enzyme turnover","volume":"44","author":"Etemad","year":"2025","journal-title":"EMBO J"},{"issue":"1","key":"10.1016\/j.ebiom.2026.106474_bib15","doi-asserted-by":"crossref","first-page":"19","DOI":"10.1016\/j.stemcr.2016.05.004","article-title":"Tissue-engineered vascular rings from human iPSC-Derived smooth muscle cells","volume":"7","author":"Dash","year":"2016","journal-title":"Stem Cell Reports"},{"issue":"3","key":"10.1016\/j.ebiom.2026.106474_bib16","doi-asserted-by":"crossref","first-page":"972","DOI":"10.1002\/hep.29400","article-title":"Src kinase inhibition reduces inflammatory and cytoskeletal changes in DeltaF508 human cholangiocytes and improves cystic fibrosis transmembrane conductance regulator correctors efficacy","volume":"67","author":"Fiorotto","year":"2018","journal-title":"Hepatology"},{"issue":"2","key":"10.1016\/j.ebiom.2026.106474_bib17","doi-asserted-by":"crossref","first-page":"700","DOI":"10.1002\/hep.27165","article-title":"Generation of functional cholangiocyte-like cells from human pluripotent stem cells and HepaRG cells","volume":"60","author":"Dianat","year":"2014","journal-title":"Hepatology"},{"issue":"8","key":"10.1016\/j.ebiom.2026.106474_bib18","doi-asserted-by":"crossref","first-page":"853","DOI":"10.1038\/nbt.3294","article-title":"Directed differentiation of cholangiocytes from human pluripotent stem cells","volume":"33","author":"Ogawa","year":"2015","journal-title":"Nat Biotechnol"},{"issue":"8","key":"10.1016\/j.ebiom.2026.106474_bib19","doi-asserted-by":"crossref","first-page":"845","DOI":"10.1038\/nbt.3275","article-title":"Cholangiocytes derived from human induced pluripotent stem cells for disease modeling and drug validation","volume":"33","author":"Sampaziotis","year":"2015","journal-title":"Nat Biotechnol"},{"issue":"11","key":"10.1016\/j.ebiom.2026.106474_bib20","doi-asserted-by":"crossref","first-page":"5171","DOI":"10.1038\/s41596-021-00607-0","article-title":"Controlling the polarity of human gastrointestinal organoids to investigate epithelial biology and infectious diseases","volume":"16","author":"Co","year":"2021","journal-title":"Nat Protoc"},{"issue":"1","key":"10.1016\/j.ebiom.2026.106474_bib21","doi-asserted-by":"crossref","first-page":"219","DOI":"10.1016\/j.jhep.2021.03.005","article-title":"A single cell gene expression atlas of 28 human livers","volume":"75","author":"Brancale","year":"2021","journal-title":"J Hepatol"},{"issue":"5","key":"10.1016\/j.ebiom.2026.106474_bib22","doi-asserted-by":"crossref","first-page":"495","DOI":"10.1038\/nbt.3192","article-title":"Spatial reconstruction of single-cell gene expression data","volume":"33","author":"Satija","year":"2015","journal-title":"Nat Biotechnol"},{"key":"10.1016\/j.ebiom.2026.106474_bib23","doi-asserted-by":"crossref","first-page":"23","DOI":"10.1186\/s13630-016-0044-2","article-title":"The IN\/OUT assay: a new tool to study ciliogenesis","volume":"5","author":"Kukic","year":"2016","journal-title":"Cilia"},{"issue":"4","key":"10.1016\/j.ebiom.2026.106474_bib24","doi-asserted-by":"crossref","DOI":"10.1111\/acel.13584","article-title":"Acidic nanoparticles protect against alpha-synuclein-induced neurodegeneration through the restoration of lysosomal function","volume":"21","author":"Arotcarena","year":"2022","journal-title":"Aging Cell"},{"issue":"2","key":"10.1016\/j.ebiom.2026.106474_bib25","doi-asserted-by":"crossref","first-page":"E87","DOI":"10.1152\/ajpendo.00457.2019","article-title":"A pipeline for multidimensional confocal analysis of mitochondrial morphology, function, and dynamics in pancreatic beta-cells","volume":"318","author":"Chaudhry","year":"2020","journal-title":"Am J Physiol Endocrinol Metab"},{"key":"10.1016\/j.ebiom.2026.106474_bib26","doi-asserted-by":"crossref","first-page":"80","DOI":"10.1016\/j.ymeth.2016.09.016","article-title":"TrackMate: an open and extensible platform for single-particle tracking","volume":"115","author":"Tinevez","year":"2017","journal-title":"Methods"},{"key":"10.1016\/j.ebiom.2026.106474_bib27","doi-asserted-by":"crossref","DOI":"10.7554\/eLife.42288","article-title":"KymoButler, a deep learning software for automated kymograph analysis","volume":"8","author":"Jakobs","year":"2019","journal-title":"Elife"},{"key":"10.1016\/j.ebiom.2026.106474_bib28","doi-asserted-by":"crossref","DOI":"10.7554\/eLife.33125","article-title":"Robust model-based analysis of single-particle tracking experiments with Spot-On","volume":"7","author":"Hansen","year":"2018","journal-title":"Elife"},{"issue":"12","key":"10.1016\/j.ebiom.2026.106474_bib29","doi-asserted-by":"crossref","first-page":"550","DOI":"10.1186\/s13059-014-0550-8","article-title":"Moderated estimation of fold change and dispersion for RNA-seq data with DESeq2","volume":"15","author":"Love","year":"2014","journal-title":"Genome Biol"},{"issue":"1","key":"10.1016\/j.ebiom.2026.106474_bib30","doi-asserted-by":"crossref","first-page":"1816","DOI":"10.1038\/s41598-020-58766-1","article-title":"M3C: Monte carlo reference-based consensus clustering","volume":"10","author":"John","year":"2020","journal-title":"Sci Rep"},{"issue":"3","key":"10.1016\/j.ebiom.2026.106474_bib31","doi-asserted-by":"crossref","DOI":"10.1002\/imt2.43","article-title":"Complex heatmap visualization","volume":"1","author":"Gu","year":"2022","journal-title":"Imeta"},{"issue":"3","key":"10.1016\/j.ebiom.2026.106474_bib32","article-title":"clusterProfiler 4.0: a universal enrichment tool for interpreting omics data","volume":"2","author":"Wu","year":"2021","journal-title":"Innovation (Camb)"},{"key":"10.1016\/j.ebiom.2026.106474_bib33","article-title":"Fast gene set enrichment analysis","author":"Korotkevich","year":"2021","journal-title":"bioRxiv"},{"issue":"6531","key":"10.1016\/j.ebiom.2026.106474_bib34","doi-asserted-by":"crossref","first-page":"839","DOI":"10.1126\/science.aaz6964","article-title":"Cholangiocyte organoids can repair bile ducts after transplantation in the human liver","volume":"371","author":"Sampaziotis","year":"2021","journal-title":"Science"},{"key":"10.1016\/j.ebiom.2026.106474_bib35","doi-asserted-by":"crossref","first-page":"70","DOI":"10.1016\/j.crtox.2020.08.001","article-title":"Co-culture with mouse embryonic fibroblasts improves maintenance of metabolic function of human small hepatocyte progenitor cells","volume":"1","author":"Sengupta","year":"2020","journal-title":"Curr Res Toxicol"},{"key":"10.1016\/j.ebiom.2026.106474_bib36","doi-asserted-by":"crossref","DOI":"10.1016\/j.scr.2021.102523","article-title":"In vitro expansion of cirrhosis derived liver epithelial cells with defined small molecules","volume":"56","author":"Li","year":"2021","journal-title":"Stem Cell Res"},{"issue":"1","key":"10.1016\/j.ebiom.2026.106474_bib37","doi-asserted-by":"crossref","first-page":"5292","DOI":"10.1038\/s41467-020-19041-z","article-title":"Direct reprogramming of human umbilical vein- and peripheral blood-derived endothelial cells into hepatic progenitor cells","volume":"11","author":"Inada","year":"2020","journal-title":"Nat Commun"},{"issue":"12","key":"10.1016\/j.ebiom.2026.106474_bib38","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0168266","article-title":"Hedgehog signaling overcomes an EZH2-Dependent epigenetic barrier to promote cholangiocyte expansion","volume":"11","author":"Jalan-Sakrikar","year":"2016","journal-title":"PLoS One"},{"issue":"2","key":"10.1016\/j.ebiom.2026.106474_bib39","first-page":"342","article-title":"Neutrophils interact with cholangiocytes to cause cholestatic changes in alcoholic hepatitis","volume":"70","author":"Takeuchi","year":"2021","journal-title":"Gut"},{"issue":"7","key":"10.1016\/j.ebiom.2026.106474_bib40","doi-asserted-by":"crossref","first-page":"1147","DOI":"10.1016\/S0092-8674(00)81459-2","article-title":"Targeted disruption of mouse conventional kinesin heavy chain, kif5B, results in abnormal perinuclear clustering of mitochondria","volume":"93","author":"Tanaka","year":"1998","journal-title":"Cell"},{"issue":"12","key":"10.1016\/j.ebiom.2026.106474_bib41","doi-asserted-by":"crossref","first-page":"4137","DOI":"10.1529\/biophysj.106.100206","article-title":"Tracking single Kinesin molecules in the cytoplasm of mammalian cells","volume":"92","author":"Cai","year":"2007","journal-title":"Biophys J"},{"issue":"46","key":"10.1016\/j.ebiom.2026.106474_bib42","doi-asserted-by":"crossref","first-page":"E7185","DOI":"10.1073\/pnas.1611398113","article-title":"Transport efficiency of membrane-anchored kinesin-1 motors depends on motor density and diffusivity","volume":"113","author":"Grover","year":"2016","journal-title":"Proc Natl Acad Sci USA"},{"issue":"4 Pt B","key":"10.1016\/j.ebiom.2026.106474_bib43","doi-asserted-by":"crossref","first-page":"1245","DOI":"10.1016\/j.bbadis.2017.06.006","article-title":"The cholangiocyte primary cilium in health and disease","volume":"1864","author":"Mansini","year":"2018","journal-title":"Biochim Biophys Acta Mol Basis Dis"},{"issue":"5","key":"10.1016\/j.ebiom.2026.106474_bib44","doi-asserted-by":"crossref","first-page":"751","DOI":"10.1038\/modpathol.2011.212","article-title":"Cholangiocyte cilia are abnormal in syndromic and non-syndromic biliary atresia","volume":"25","author":"Chu","year":"2012","journal-title":"Mod Pathol"},{"issue":"1","key":"10.1016\/j.ebiom.2026.106474_bib45","doi-asserted-by":"crossref","first-page":"96","DOI":"10.1097\/MPG.0b013e318296e525","article-title":"Extrahepatic cholangiocyte cilia are abnormal in biliary atresia","volume":"57","author":"Karjoo","year":"2013","journal-title":"J Pediatr Gastroenterol Nutr"},{"key":"10.1016\/j.ebiom.2026.106474_bib46","doi-asserted-by":"crossref","DOI":"10.1016\/j.ebiom.2021.103530","article-title":"Identification of a wide spectrum of ciliary gene mutations in nonsyndromic biliary atresia patients implicates ciliary dysfunction as a novel disease mechanism","volume":"71","author":"Lam","year":"2021","journal-title":"EBioMedicine"},{"issue":"1","key":"10.1016\/j.ebiom.2026.106474_bib47","doi-asserted-by":"crossref","first-page":"89","DOI":"10.1002\/hep.32107","article-title":"Biliary organoids uncover delayed epithelial development and barrier function in biliary atresia","volume":"75","author":"Amarachintha","year":"2022","journal-title":"Hepatology"},{"issue":"12","key":"10.1016\/j.ebiom.2026.106474_bib48","doi-asserted-by":"crossref","first-page":"797","DOI":"10.1136\/jmedgenet-2019-106769","article-title":"'Kinesinopathies': emerging role of the kinesin family member genes in birth defects","volume":"57","author":"Kalantari","year":"2020","journal-title":"J Med Genet"}],"container-title":["eBioMedicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2352396426003580?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2352396426003580?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T08:22:57Z","timestamp":1791966177000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S2352396426003580"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,10]]},"references-count":48,"alternative-id":["S2352396426003580"],"URL":"https:\/\/doi.org\/10.1016\/j.ebiom.2026.106474","relation":{},"ISSN":["2352-3964"],"issn-type":[{"value":"2352-3964","type":"print"}],"subject":[],"published":{"date-parts":[[2026,10]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Liver disease reveals KIF12 as a regulator of mitochondria, lysosome and cilia localisation in human cholangiocytes","name":"articletitle","label":"Article Title"},{"value":"eBioMedicine","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.ebiom.2026.106474","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2026 The Author(s). Published by Elsevier B.V.","name":"copyright","label":"Copyright"}],"article-number":"106474"}}