PUBLICATION
Involvement of the centrosomal protein 55 (cep55) gene in zebrafish head formation
- Authors
- Yanagi, K., Sone, R., Ohga, R., Kawahara, A.
- ID
- ZDB-PUB-190801-24
- Date
- 2019
- Source
- Genes to cells : devoted to molecular & cellular mechanisms 24(10): 642-649 (Journal)
- Registered Authors
- Kawahara, Atsuo
- Keywords
- cep55, MARCH syndrome, cell death, centrosomal protein, cytokinesis
- MeSH Terms
-
- 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
- PubMed
- 31365163 Full text @ Genes Cells
Citation
Yanagi, K., Sone, R., Ohga, R., Kawahara, A. (2019) Involvement of the centrosomal protein 55 (cep55) gene in zebrafish head formation. Genes to cells : devoted to molecular & cellular mechanisms. 24(10):642-649.
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 nonsense 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. Therefore, we propose that the zebrafish cep55 mutant is a model organism for human MARCH syndrome. This article is protected by copyright. All rights reserved.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping