Involvement of the centrosomal protein 55 (cep55) gene in zebrafish head formation

Genes to Cells : Devoted to Molecular & Cellular Mechanisms
Kanoko YanagiAtsuo Kawahara

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.

References

Jul 27, 2002·Developmental Biology·Atsuo KawaharaIgor B Dawid
Aug 9, 2002·Developmental Biology·Nathan D Lawson, Brant M Weinstein
Mar 23, 2013·Biology Open·Katy SchmidtDavid J Stephens
Apr 12, 2013·Genes to Cells : Devoted to Molecular & Cellular Mechanisms·Satoshi OtaAtsuo Kawahara
Jan 7, 2014·Tumour Biology : the Journal of the International Society for Oncodevelopmental Biology and Medicine·Jinqiu TaoZekuan Xu
Feb 11, 2015·FASEB Journal : Official Publication of the Federation of American Societies for Experimental Biology·Jessie JefferyKum Kum Khanna

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