RRC ID 60057
Author Yanagi K, Sone R, Ohga R, Kawahara A.
Title Involvement of the centrosomal protein 55 (cep55) gene in zebrafish head formation.
Journal Genes Cells
Abstract Mammalian CEP55 (centrosomal protein 55 kDa) is a coiled-coil protein localized to the centrosome in interphase cells and is required for cytokinesis. A homozygous non-sense mutation in human CEP55 has been recently identified in perinatal lethal MARCH (multinucleated neurons, anhydramnios, renal dysplasia, cerebellar hypoplasia and hydranencephaly) syndrome. We have isolated zebrafish cep55 mutants defective in head morphology. The zebrafish cep55 gene was expressed in the head including the retina and the pectoral fin at 1 day post-fertilization (dpf), and extensive cell death was widely observed in the head and tail of the cep55 mutant. In the cep55 mutant, the anterior-posterior distance of the ventral pharyngeal arches was short, and retinal lamination was disorganized. Neural cells, such as islet1-positive cells and pax2-positive cells, and fli1b-positive vascular cells were reduced in the head of the cep55 mutant. Thus, we propose that the zebrafish cep55 mutant is a model organism for human MARCH syndrome.
Volume 24(10)
Pages 642-649
Published 2019-10
DOI 10.1111/gtc.12715
PMID 31365163
MeSH Animals Cell Cycle Proteins / genetics* Cell Cycle Proteins / metabolism Centrosome / metabolism Cytokinesis / genetics Head / abnormalities Head / embryology Mutation Nuclear Proteins / genetics* Nuclear Proteins / metabolism Transcription Factors / genetics Transcription Factors / metabolism Zebrafish / abnormalities Zebrafish / embryology* Zebrafish / genetics Zebrafish Proteins / genetics* Zebrafish Proteins / metabolism
IF 1.922
Times Cited 0
Zebrafish Tg(isl-GFP) rw0