{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T00:21:07Z","timestamp":1790814067728,"version":"4.1.0"},"reference-count":18,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2019,9,1]],"date-time":"2019-09-01T00:00:00Z","timestamp":1567296000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100007449","name":"Takeda Science Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007449","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Genes to Cells"],"published-print":{"date-parts":[[2019,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Mammalian CEP55 (centrosomal protein 55\u00a0kDa) is a coiled\u2010coil protein localized to the centrosome in interphase cells and is required for cytokinesis. A homozygous non\u2010sense mutation in human <jats:italic>CEP55<\/jats:italic> has been recently identified in perinatal lethal MARCH (multinucleated neurons, anhydramnios, renal dysplasia, cerebellar hypoplasia and hydranencephaly) syndrome. We have isolated zebrafish <jats:italic>cep55<\/jats:italic> mutants defective in head morphology. The zebrafish <jats:italic>cep55<\/jats:italic> gene was expressed in the head including the retina and the pectoral fin at 1\u00a0day post\u2010fertilization (dpf), and extensive cell death was widely observed in the head and tail of the <jats:italic>cep55<\/jats:italic> mutant. In the <jats:italic>cep55<\/jats:italic> mutant, the anterior\u2013posterior distance of the ventral pharyngeal arches was short, and retinal lamination was disorganized. Neural cells, such as <jats:italic>islet1<\/jats:italic>\u2010positive cells and <jats:italic>pax2<\/jats:italic>\u2010positive cells, and <jats:italic>fli1b<\/jats:italic>\u2010positive vascular cells were reduced in the head of the <jats:italic>cep55<\/jats:italic> mutant. Thus, we propose that the zebrafish <jats:italic>cep55<\/jats:italic> mutant is a model organism for human MARCH syndrome.<\/jats:p>","DOI":"10.1111\/gtc.12715","type":"journal-article","created":{"date-parts":[[2019,7,31]],"date-time":"2019-07-31T14:49:13Z","timestamp":1564584553000},"page":"642-649","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Involvement of the <i>centrosomal protein 55<\/i> (<i>cep55<\/i>) gene in zebrafish head formation"],"prefix":"10.1111","volume":"24","author":[{"given":"Kanoko","family":"Yanagi","sequence":"first","affiliation":[{"name":"Laboratory for Developmental Biology, Center for Medical Education and Sciences, Graduate School of Medical Science University of Yamanashi Chuo Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ryota","family":"Sone","sequence":"additional","affiliation":[{"name":"Laboratory for Developmental Biology, Center for Medical Education 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