4.7 Article

Fam151b, the mouse homologue of C.elegans menorin gene, is essential for retinal function

期刊

SCIENTIFIC REPORTS
卷 10, 期 1, 页码 -

出版社

NATURE PUBLISHING GROUP
DOI: 10.1038/s41598-019-57398-4

关键词

-

资金

  1. MRC [A410]
  2. NIH [5UM1HG006348-08]
  3. Medical Research Council
  4. MRC [MC_PC_U127561112, MC_UU_00007/15, MC_UU_00007/4, MC_UP_1502/1] Funding Source: UKRI

向作者/读者索取更多资源

Fam151b is a mammalian homologue of the C. elegans menorin gene, which is involved in neuronal branching. The International Mouse Phenotyping Consortium (IMPC) aims to knock out every gene in the mouse and comprehensively phenotype the mutant animals. This project identified Fam151b homozygous knock-out mice as having retinal degeneration. We show they have no photoreceptor function from eye opening, as demonstrated by a lack of electroretinograph (ERG) response. Histological analysis shows that during development of the eye the correct number of cells are produced and that the layers of the retina differentiate normally. However, after eye opening at P14, Fam151b mutant eyes exhibit signs of retinal stress and rapidly lose photoreceptor cells. We have mutated the second mammalian menorin homologue, Fam151a, and homozygous mutant mice have no discernible phenotype. Sequence analysis indicates that the FAM151 proteins are members of the PLC-like phosphodiesterase superfamily. However, the substrates and function of the proteins remains unknown.

作者

我是这篇论文的作者
点击您的名字以认领此论文并将其添加到您的个人资料中。

评论

主要评分

4.7
评分不足

次要评分

新颖性
-
重要性
-
科学严谨性
-
评价这篇论文

推荐

暂无数据
暂无数据