Camel regulates development of the brain ventricular system
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Camel regulates development of the brain ventricular system Shulan Yang 1,2 & Alexander Emelyanov 1,3 & May-Su You 1,4 & Melvin Sin 1 & Vladimir Korzh 1,5 Received: 8 April 2020 / Accepted: 29 July 2020 # The Author(s) 2020
Abstract Development of the brain ventricular system of vertebrates and the molecular mechanisms involved are not fully understood. 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—camel. 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. camel 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’s derivative—the Reissner fiber— participates in the flow of cerebrospinal fluid. The Reissner fiber fails to form upon morpholino-mediated Camel loss-of-function. The Camel mRNA–mediated 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. Keywords Subcommissural organ . Flexural organ . Floor plate . Roof plate . Hypochord . Ependyma
Introduction The subcommissural organ (SCO) is an ependymal brain gland found in the diencephalic roof of the third ventricle at the entrance to the cerebral aqueduct. It synthesizes and Electronic supplementary material The online version of this article (https://doi.org/10.1007/s00441-020-03270-1) contains supplementary material, which is available to authorized users. * Vladimir Korzh [email protected] 1
Institute of Molecular and Cell Biology, Agency for Science, Technology and Research, Singapore, Singapore
2
Translational Medicine Centre, The First Affiliated Hospital, Sun Yat-sen University, Guangzhou, China
3
Institute for Research on Cancer and Aging, Nice, France
4
National Health Research Institutes, Zhunan, Taiwan
5
International Institute of Molecular and Cell Biology, Warsaw, Poland
releases into the third ventricle (vIII) large g
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