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The developmental genes expressed in the elements of the brain ventricular system such as the ependyma and circumventricular organs act as molecular determinants of cell adhesion critical for the formation of brain ventricular system. They control brain development and function, including the flow of cerebrospinal fluid. Here, we describe the novel distantly related member of the zebrafish L1-CAM family of genes\u2014<jats:italic>camel<\/jats:italic>. Whereas its maternal transcripts distributed uniformly, the zygotic transcripts demonstrate clearly defined expression patterns, in particular in the axial structures: floor plate, hypochord, and roof plate. <jats:italic>camel<\/jats:italic> expresses in several other cell lineages with access to the brain ventricular system, including the midbrain roof plate, subcommissural organ, organum vasculosum lamina terminalis, median eminence, paraventricular organ, flexural organ, and inter-rhombomeric boundaries. This expression pattern suggests a role of Camel in neural development. Several isoforms of Camel generated by differential splicing of exons encoding the sixth fibronectin type III domain enhance cell adhesion differentially. The antisense oligomer morpholino-mediated loss-of-function of Camel affects cell adhesion and causes hydrocephalus and scoliosis manifested via the tail curled down phenotype. The subcommissural organ\u2019s derivative\u2014the Reissner fiber\u2014participates in the flow of cerebrospinal fluid. The Reissner fiber fails to form upon morpholino-mediated Camel loss-of-function. The Camel mRNA\u2013mediated gain-of-function causes the Reissner fiber misdirection. This study revealed a link between Chl1a\/Camel and Reissner fiber formation, and this supports the idea that CHL1 is one of the scoliosis factors.<\/jats:p>","DOI":"10.1007\/s00441-020-03270-1","type":"journal-article","created":{"date-parts":[[2020,9,10]],"date-time":"2020-09-10T10:51:40Z","timestamp":1599735100000},"page":"835-852","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Camel regulates development of the brain ventricular system"],"prefix":"10.1007","volume":"383","author":[{"given":"Shulan","family":"Yang","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alexander","family":"Emelyanov","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"May-Su","family":"You","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Melvin","family":"Sin","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6580-7986","authenticated-orcid":false,"given":"Vladimir","family":"Korzh","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2020,9,9]]},"reference":[{"issue":"1","key":"3270_CR1","doi-asserted-by":"publisher","first-page":"9368","DOI":"10.1038\/s41598-017-09599-y","volume":"7","author":"WF Alsanie","year":"2017","unstructured":"Alsanie WF, Penna V, Schachner M, Thompson LH, Parish CL (2017) Homophilic binding of the neural cell adhesion molecule CHL1 regulates development of ventral midbrain dopaminergic pathways. 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