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However, little is known about the molecular and cellular mechanisms regulating CSF flow in the developing brain. Through whole-genome sequencing analysis, we report that a homozygous splice site mutation in coiled-coil domain containing 39 (Ccdc39) is responsible for early postnatal hydrocephalus in the progressive hydrocephalus (prh) mouse mutant. Ccdc39 is selectively expressed in embryonic choroid plexus and ependymal cells on the medial wall of the forebrain ventricle, and the protein is localized to the axoneme of motile cilia. The Ccdc39prh\/prh ependymal cells develop shorter cilia with disorganized microtubules lacking the axonemal inner arm dynein. Using high-speed video microscopy, we show that an orchestrated ependymal ciliary beating pattern controls unidirectional CSF flow on the ventricular surface, which generates bulk CSF flow in the developing brain. Collectively, our data provide the first evidence for involvement of Ccdc39 in hydrocephalus and suggest that the proper development of medial wall ependymal cilia is crucial for normal mouse brain development.<\/jats:p>","DOI":"10.1242\/dev.154500","type":"journal-article","created":{"date-parts":[[2018,1,10]],"date-time":"2018-01-10T01:20:35Z","timestamp":1515547235000},"source":"Crossref","is-referenced-by-count":87,"title":["A mutation in <i>Ccdc39<\/i> causes neonatal hydrocephalus with abnormal motile cilia development in mice"],"prefix":"10.1242","volume":"145","author":[{"given":"Zakia","family":"Abdelhamed","sequence":"first","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Pediatric Neurosurgery , , 3333 Burnet Ave, Cincinnati, OH 45242 , USA"},{"name":"Al-Azhar University 2 Department of Anatomy and Embryology, Faculty of Medicine (Girls\u2019 Section) , , Cairo 11651, Egypt"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shawn M.","family":"Vuong","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Pediatric Neurosurgery , , 3333 Burnet Ave, Cincinnati, OH 45242 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lauren","family":"Hill","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Pediatric Neurosurgery , , 3333 Burnet Ave, Cincinnati, OH 45242 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Crystal","family":"Shula","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Pediatric Neurosurgery , , 3333 Burnet Ave, Cincinnati, OH 45242 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew","family":"Timms","sequence":"additional","affiliation":[{"name":"Center for Developmental Biology and Regenerative Medicine, Seattle Children's Hospital 3 , 4800 Sand Point Way NE, Seattle, WA 98105 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Beier","sequence":"additional","affiliation":[{"name":"Center for Developmental Biology and Regenerative Medicine, Seattle Children's Hospital 3 , 4800 Sand Point Way NE, Seattle, WA 98105 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kenneth","family":"Campbell","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Pediatric Neurosurgery , , 3333 Burnet Ave, Cincinnati, OH 45242 , USA"},{"name":"Cincinnati Children's Hospital Medical Center 4 Division of Developmental Biology , , 3333 Burnet Ave, Cincinnati, OH 45242 \u2008 USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francesco T.","family":"Mangano","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Pediatric Neurosurgery , , 3333 Burnet Ave, Cincinnati, OH 45242 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4512-6806","authenticated-orcid":false,"given":"Rolf W.","family":"Stottmann","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 4 Division of Developmental Biology , , 3333 Burnet Ave, Cincinnati, OH 45242 \u2008 USA"},{"name":"Cincinnati Children's Hospital Medical Center 5 Division of Human Genetics , , 3333 Burnet Ave, Cincinnati, OH 45242 \u2008 USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4000-9750","authenticated-orcid":false,"given":"June","family":"Goto","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Pediatric Neurosurgery , , 3333 Burnet Ave, Cincinnati, OH 45242 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2018,1,9]]},"reference":[{"key":"2024061216103730200_DEV154500C1","doi-asserted-by":"publisher","first-page":"403","DOI":"10.1007\/s00335-015-9579-6","article-title":"The mouse genomes project: a repository of inbred laboratory mouse strain genomes","volume":"26","author":"Adams","year":"2015","journal-title":"Mamm. 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