論文 - 詳細
| RRC ID | 60057 |
|---|---|
| 著者 | Yanagi K, Sone R, Ohga R, Kawahara A. |
| タイトル | Involvement of the centrosomal protein 55 (cep55) gene in zebrafish head formation. |
| ジャーナル | 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. |
| 巻・号 | 24(10) |
| ページ | 642-649 |
| 公開日 | 2019-10-1 |
| 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 |
| 引用数 | 0 |
| オルトメトリクス指標 |
オルトメトリクス指標項目
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| 最多言及媒体 | |
| 各媒体での言及数の合計 | 1 |
| 過去6か月間でのオルトメトリクス指標の変動値 | 0.0 |
| リソース情報 | |
| ゼブラフィッシュ | Tg(isl-GFP) rw0 |