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Abrogation of Stem Loop Binding Protein (Slbp) function leads to a failure of cells to transition from proliferation to differentiation, retinal coloboma and midline axon guidance deficits

Turner, Katherine J.; Hoyle, Jacqueline; Valdivia, Leonardo E.; Cerveny, Kara L.; Hart, Wendy; Mangoli, Maryam; Geisler, Robert; Rees, Michele; Houart, Corinne; Poole, Richard J.; Wilson, Stephen W.; Gestri, Gaia; Moens, Cecilia [Hrsg.]

Abstract:
Through forward genetic screening for mutations affecting visual system development, we identified prominent coloboma and cell-autonomous retinal neuron differentiation, lamination and retinal axon projection defects in eisspalte (ele) mutant zebrafish. Additional axonal deficits were present, most notably at midline axon commissures. Genetic mapping and cloning of the ele mutation showed that the affected gene is slbp, which encodes a conserved RNA stem-loop binding protein involved in replication dependent histone mRNA metabolism. Cells throughout the central nervous system remained in the cell cycle in ele mutant embryos at stages when, and locations where, post-mitotic cells have differentiated in wild-type siblings. Indeed, RNAseq analysis showed down-regulation of many genes associated with neuronal differentiation. This was coincident with changes in the levels and spatial localisation of expression of various genes implicated, for instance, in axon guidance, that likely underlie specific ele phenotypes. These results suggest that many of the cell and tissue specific phenotypes in ele mutant embryos are secondary to altered e ... mehr

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Verlagsausgabe §
DOI: 10.5445/IR/1000091157
Veröffentlicht am 18.02.2019
Zugehörige Institution(en) am KIT Institut für Toxikologie und Genetik (ITG)
Publikationstyp Zeitschriftenaufsatz
Jahr 2019
Sprache Englisch
Identifikator ISSN: 1932-6203
urn:nbn:de:swb:90-911576
KITopen-ID: 1000091157
HGF-Programm 47.01.01 (POF III, LK 01)
Erschienen in PLOS ONE
Band 14
Heft 1
Seiten Article: e0211073
Vorab online veröffentlicht am 29.01.2019
Nachgewiesen in Web of Science
Scopus
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