{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,18]],"date-time":"2026-09-18T21:03:11Z","timestamp":1789765391043,"version":"4.0.1"},"reference-count":28,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2024,6,1]],"date-time":"2024-06-01T00:00:00Z","timestamp":1717200000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2024,6,1]],"date-time":"2024-06-01T00:00:00Z","timestamp":1717200000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2024,4,11]],"date-time":"2024-04-11T00:00:00Z","timestamp":1712793600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100007449","name":"Takeda Science Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007449","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["20H03644"],"award-info":[{"award-number":["20H03644"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["23K07336"],"award-info":[{"award-number":["23K07336"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Biochemical and Biophysical Research Communications"],"published-print":{"date-parts":[[2024,6]]},"DOI":"10.1016\/j.bbrc.2024.149932","type":"journal-article","created":{"date-parts":[[2024,4,11]],"date-time":"2024-04-11T21:15:35Z","timestamp":1712870135000},"page":"149932","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":10,"special_numbering":"C","title":["DHCR7 links cholesterol synthesis with neuronal development and axonal integrity"],"prefix":"10.1016","volume":"712-713","author":[{"ORCID":"https:\/\/orcid.org\/0009-0009-6494-6675","authenticated-orcid":false,"given":"Shuya","family":"Miyazaki","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8100-0330","authenticated-orcid":false,"given":"Nobuyuki","family":"Shimizu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroaki","family":"Miyahara","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2106-2564","authenticated-orcid":false,"given":"Hitoshi","family":"Teranishi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5829-9533","authenticated-orcid":false,"given":"Ryohei","family":"Umeda","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shinji","family":"Yano","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tatsuo","family":"Shimada","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroshi","family":"Shiraishi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kosaku","family":"Komiya","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Akira","family":"Katoh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5676-8949","authenticated-orcid":false,"given":"Akihiko","family":"Yoshimura","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Reiko","family":"Hanada","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3317-6366","authenticated-orcid":false,"given":"Toshikatsu","family":"Hanada","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.bbrc.2024.149932_bib1","doi-asserted-by":"crossref","first-page":"107","DOI":"10.1056\/NEJM199401133300205","article-title":"Defective cholesterol biosynthesis associated with the Smith-Lemli-Opitz syndrome","volume":"330","author":"Tint","year":"1994","journal-title":"N. Engl. J. Med."},{"key":"10.1016\/j.bbrc.2024.149932_bib2","doi-asserted-by":"crossref","first-page":"138","DOI":"10.1016\/j.plipres.2016.09.003","article-title":"DHCR7: a vital enzyme switch between cholesterol and vitamin D production","volume":"64","author":"Prabhu","year":"2016","journal-title":"Prog. Lipid Res."},{"key":"10.1016\/j.bbrc.2024.149932_bib3","article-title":"Cholesterol: its regulation and role in central nervous system disorders","author":"Orth","year":"2012","journal-title":"Cholesterol 2012"},{"key":"10.1016\/j.bbrc.2024.149932_bib4","doi-asserted-by":"crossref","first-page":"2965","DOI":"10.1016\/S0006-3495(04)74347-7","article-title":"Role of cholesterol in the formation and nature of lipid rafts in planar and spherical model membranes","volume":"86","author":"Crane","year":"2004","journal-title":"Biophys. J."},{"key":"10.1016\/j.bbrc.2024.149932_bib5","doi-asserted-by":"crossref","first-page":"1249","DOI":"10.1038\/ncomms2221","article-title":"Cholesterol modulates cell signaling and protein networking by specifically interacting with PDZ domain-containing scaffold proteins","volume":"3","author":"Sheng","year":"2012","journal-title":"Nat. Commun."},{"key":"10.1016\/j.bbrc.2024.149932_bib6","doi-asserted-by":"crossref","first-page":"4393","DOI":"10.1038\/ncomms5393","article-title":"Cholesterol selectively activates canonical Wnt signalling over non-canonical Wnt signalling","volume":"5","author":"Sheng","year":"2014","journal-title":"Nat. Commun."},{"key":"10.1016\/j.bbrc.2024.149932_bib7","doi-asserted-by":"crossref","first-page":"329","DOI":"10.1086\/301982","article-title":"Smith-Lemli-Opitz syndrome is caused by mutations in the 7-dehydrocholesterol reductase gene","volume":"63","author":"Waterham","year":"1998","journal-title":"Am. J. Hum. Genet."},{"key":"10.1016\/j.bbrc.2024.149932_bib8","doi-asserted-by":"crossref","first-page":"250","DOI":"10.1002\/ajmg.c.31343","article-title":"Smith-Lemli-Opitz syndrome: phenotype, natural history, and epidemiology","volume":"160C","author":"Nowaczyk","year":"2012","journal-title":"Am J Med Genet C Semin Med Genet"},{"key":"10.1016\/j.bbrc.2024.149932_bib9","doi-asserted-by":"crossref","first-page":"905","DOI":"10.1172\/JCI200112103","article-title":"7-Dehydrocholesterol\u2013dependent proteolysis of HMG-CoA reductase suppresses sterol biosynthesis in a mouse model of Smith-Lemli-Opitz\/RSH syndrome","volume":"108","author":"Fitzky","year":"2001","journal-title":"J. Clin. Invest."},{"key":"10.1016\/j.bbrc.2024.149932_bib10","doi-asserted-by":"crossref","first-page":"839","DOI":"10.1093\/hmg\/ddl003","article-title":"Development and characterization of a hypomorphic Smith-Lemli-Opitz syndrome mouse model and efficacy of simvastatin therapy","volume":"15","author":"Correa-Cerro","year":"2006","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.bbrc.2024.149932_bib11","doi-asserted-by":"crossref","first-page":"465","DOI":"10.1038\/cr.2013.45","article-title":"Genome editing with RNA-guided Cas9 nuclease in zebrafish embryos","volume":"23","author":"Chang","year":"2013","journal-title":"Cell Res."},{"key":"10.1016\/j.bbrc.2024.149932_bib12","doi-asserted-by":"crossref","first-page":"237","DOI":"10.1007\/978-1-4939-3771-4_16","article-title":"Studying lipid metabolism and transport during zebrafish development","volume":"1451","author":"Zeituni","year":"2016","journal-title":"Methods Mol. Biol."},{"key":"10.1016\/j.bbrc.2024.149932_bib13","doi-asserted-by":"crossref","first-page":"2063","DOI":"10.1194\/jlr.D800017-JLR200","article-title":"Highly sensitive analysis of sterol profiles in human serum by LC-ESI-MS\/MS","volume":"49","author":"Honda","year":"2008","journal-title":"J. Lipid Res."},{"key":"10.1016\/j.bbrc.2024.149932_bib14","first-page":"400","article-title":"A method for the combined staining of cells and fibers in the nervous system, J. Neuropathol","volume":"12","author":"Kluver","year":"1953","journal-title":"Exp. Neurol."},{"key":"10.1016\/j.bbrc.2024.149932_bib15","doi-asserted-by":"crossref","first-page":"10","DOI":"10.1016\/j.bbrc.2023.07.005","article-title":"Behavioral and neurological effects of Vrk1 deficiency in zebrafish","volume":"675","author":"Carrasco Apolinario","year":"2023","journal-title":"Biochem. Biophys. Res. Commun."},{"key":"10.1016\/j.bbrc.2024.149932_bib16","doi-asserted-by":"crossref","first-page":"38","DOI":"10.1016\/j.bbr.2009.06.022","article-title":"Understanding behavioral and physiological phenotypes of stress and anxiety in zebrafish","volume":"205","author":"Egan","year":"2009","journal-title":"Behav. Brain Res."},{"key":"10.1016\/j.bbrc.2024.149932_bib17","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1007\/s10641-006-9122-4","article-title":"The effects of early and adult social environment on zebrafish (Danio rerio) behavior","volume":"80","author":"Moretz","year":"2006","journal-title":"Environ. Biol. Fish."},{"key":"10.1016\/j.bbrc.2024.149932_bib18","doi-asserted-by":"crossref","first-page":"136","DOI":"10.1002\/gene.20196","article-title":"HuC:Kaede, a useful tool to label neural morphologies in networks in vivo","volume":"44","author":"Sato","year":"2006","journal-title":"Genesis"},{"key":"10.1016\/j.bbrc.2024.149932_bib19","doi-asserted-by":"crossref","first-page":"1771","DOI":"10.1523\/JNEUROSCI.5500-11.2012","article-title":"Generation of multiple classes of V0 neurons in zebrafish spinal cord: progenitor heterogeneity and temporal control of neuronal diversity","volume":"32","author":"Satou","year":"2012","journal-title":"J. Neurosci."},{"key":"10.1016\/j.bbrc.2024.149932_bib20","doi-asserted-by":"crossref","first-page":"475","DOI":"10.1002\/dvdy.21853","article-title":"Gfap and nestin reporter lines reveal characteristics of neural progenitors in the adult zebrafish brain","volume":"238","author":"Lam","year":"2009","journal-title":"Dev. Dynam."},{"key":"10.1016\/j.bbrc.2024.149932_bib21","doi-asserted-by":"crossref","first-page":"263","DOI":"10.1016\/j.ydbio.2006.03.040","article-title":"Neural stem cells and neurogenesis in the adult zebrafish brain: origin, proliferation dynamics, migration and cell fate","volume":"295","author":"Grandel","year":"2006","journal-title":"Dev. Biol."},{"key":"10.1016\/j.bbrc.2024.149932_bib22","doi-asserted-by":"crossref","first-page":"71","DOI":"10.1016\/j.ydbio.2012.07.016","article-title":"Visualization and exploration of Tcf\/Lef function using a highly responsive Wnt\/beta-catenin signaling-reporter transgenic zebrafish","volume":"370","author":"Shimizu","year":"2012","journal-title":"Dev. Biol."},{"key":"10.1016\/j.bbrc.2024.149932_bib23","doi-asserted-by":"crossref","DOI":"10.3390\/ijms23147550","article-title":"Modelling autism spectrum disorder (ASD) and attention-deficit\/hyperactivity disorder (ADHD) using mice and zebrafish","volume":"23","author":"Dougnon","year":"2022","journal-title":"Int. J. Mol. Sci."},{"key":"10.1016\/j.bbrc.2024.149932_bib24","doi-asserted-by":"crossref","DOI":"10.1016\/j.celrep.2021.110008","article-title":"Sterols lower energetic barriers of membrane bending and fission necessary for efficient clathrin-mediated endocytosis","volume":"37","author":"Anderson","year":"2021","journal-title":"Cell Rep."},{"key":"10.1016\/j.bbrc.2024.149932_bib25","doi-asserted-by":"crossref","DOI":"10.7554\/eLife.67141","article-title":"7-Dehydrocholesterol-derived oxysterols cause neurogenic defects in Smith-Lemli-Opitz syndrome","volume":"11","author":"Tomita","year":"2022","journal-title":"Elife"},{"key":"10.1016\/j.bbrc.2024.149932_bib26","doi-asserted-by":"crossref","first-page":"388","DOI":"10.1038\/nm.4067","article-title":"Modeling Smith-Lemli-Opitz syndrome with induced pluripotent stem cells reveals a causal role for Wnt\/beta-catenin defects in neuronal cholesterol synthesis phenotypes","volume":"22","author":"Francis","year":"2016","journal-title":"Nat. Med."},{"key":"10.1016\/j.bbrc.2024.149932_bib27","doi-asserted-by":"crossref","first-page":"119","DOI":"10.1089\/cap.2009.0076","article-title":"Impulsive aggression in attention-deficit\/hyperactivity disorder: symptom severity, co-morbidity, and attention-deficit\/hyperactivity disorder subtype","volume":"20","author":"Connor","year":"2010","journal-title":"J. Child Adolesc. Psychopharmacol."},{"key":"10.1016\/j.bbrc.2024.149932_bib28","doi-asserted-by":"crossref","first-page":"e145","DOI":"10.1016\/j.biopsych.2011.02.036","article-title":"The roles of dopamine and noradrenaline in the pathophysiology and treatment of attention-deficit\/hyperactivity disorder","volume":"69","author":"Del Campo","year":"2011","journal-title":"Biol. Psychiatr."}],"container-title":["Biochemical and Biophysical Research Communications"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0006291X24004686?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0006291X24004686?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,9,18]],"date-time":"2026-09-18T20:14:09Z","timestamp":1789762449000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S0006291X24004686"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,6]]},"references-count":28,"alternative-id":["S0006291X24004686"],"URL":"https:\/\/doi.org\/10.1016\/j.bbrc.2024.149932","relation":{},"ISSN":["0006-291X"],"issn-type":[{"value":"0006-291X","type":"print"}],"subject":[],"published":{"date-parts":[[2024,6]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"DHCR7 links cholesterol synthesis with neuronal development and axonal integrity","name":"articletitle","label":"Article Title"},{"value":"Biochemical and Biophysical Research Communications","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.bbrc.2024.149932","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2024 The Author(s). Published by Elsevier Inc.","name":"copyright","label":"Copyright"}],"article-number":"149932"}}