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A novel knockout mouse for the small EDRK-rich factor 2 ( Serf2 ) showing developmental and other deficits
Mammalian Genome ( IF 2.5 ) Pub Date : 2021-03-13 , DOI: 10.1007/s00335-021-09864-6
Karen Cleverley 1 , Weaverly Colleen Lee 1 , Paige Mumford 1, 2 , Toby Collins 1 , Matthew Rickman 1 , Thomas J Cunningham 3 , James Cleak 3 , Joffrey Mianne 4 , Zsombor Szoke-Kovacs 4 , Michelle Stewart 3 , Lydia Teboul 3 , Cheryl Maduro 1 , Sara Wells 4 , Frances K Wiseman 1, 2 , Elizabeth M C Fisher 1
Affiliation  

The small EDRK-rich factor 2 (SERF2) is a highly conserved protein that modifies amyloid fibre assembly in vitro and promotes protein misfolding. However, the role of SERF2 in regulating age-related proteotoxicity remains largely unexplored due to a lack of in vivo models. Here, we report the generation of Serf2 knockout mice using an ES cell targeting approach, with Serf2 knockout alleles being bred onto different defined genetic backgrounds. We highlight phenotyping data from heterozygous Serf2+/− mice, including unexpected male-specific phenotypes in startle response and pre-pulse inhibition. We report embryonic lethality in Serf2−/− null animals when bred onto a C57BL/6 N background. However, homozygous null animals were viable on a mixed genetic background and, remarkably, developed without obvious abnormalities. The Serf2 knockout mice provide a powerful tool to further investigate the role of SERF2 protein in previously unexplored pathophysiological pathways in the context of a whole organism.



中文翻译:

一种新型的富含 EDRK 因子 2 (Serf2) 的新型基因敲除小鼠,表现出发育和其他缺陷

富含 EDRK 的小因子 2 (SERF2) 是一种高度保守的蛋白质,可在体外修饰淀粉样蛋白纤维组装并促进蛋白质错误折叠。然而,由于缺乏体内模型,SERF2 在调节与年龄相关的蛋白毒性中的作用在很大程度上仍未得到探索。在这里,我们使用 ES 细胞靶向方法报告了Serf2敲除小鼠的产生,其中Serf2敲除等位基因被培育到不同的定义遗传背景。我们强调了来自杂合Serf2 +/-小鼠的表型数据,包括惊吓反应和脉冲前抑制中意想不到的雄性特异性表型。我们报告了Serf2的胚胎致死率-/-在 C57BL/6 N 背景上饲养时的空动物。然而,纯合的无效动物在混合遗传背景下是可行的,并且值得注意的是,它们发育时没有明显的异常。Serf2除小鼠提供了一个强大的工具,可以进一步研究 SERF2 蛋白在整个生物体背景下在以前未探索的病理生理途径中的作用。

更新日期:2021-03-15
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