Calaxin is required for cilia-driven determination of vertebrate laterality
Calaxin is required for cilia-driven determination of vertebrate laterality
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DOI：
10.1038/s42003-019-0462-y
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发表时间：
2019-06-20
影响因子：
5.9
通讯作者：
Inaba, Kazuo
中科院分区：
文献类型：
--
作者：
Sasaki, Keita;Shiba, Kogiku;Inaba, Kazuo
Calaxin是一种钙结合动力蛋白相关蛋白，调节鞭毛和纤毛运动。在海鞘中，calaxin在精子的趋化性中起重要作用。然而，还没有人知道calaxin在脊椎动物中的功能。在这里，我们表明，Efcab1，编码calaxin的无效突变的小鼠，显示典型的表型原发性纤毛运动障碍，包括脑积水，反位，和异常运动的气管纤毛和精子鞭毛。引人注目的是，雄性和雌性都是可行的和可生育的，表明calaxin不是小鼠受精所必需的。上皮多纤毛和精子鞭毛的9 + 2轴丝结构正常，但9 + 0节纤毛的形成明显中断。在斑马鱼中敲除calaxin也会由于库普弗囊泡纤毛的不规则纤毛跳动而引起内脏逆位，尽管9 + 2轴丝结构似乎保持正常。
Calaxin is a Ca2(+)-binding dynein-associated protein that regulates flagellar and ciliary movement. In ascidians, calaxin plays essential roles in chemotaxis of sperm. However, nothing has been known for the function of calaxin in vertebrates. Here we show that the mice with a null mutation in Efcab1, which encodes calaxin, display typical phenotypes of primary ciliary dyskinesia, including hydrocephalus, situs inversus, and abnormal motility of trachea cilia and sperm flagella. Strikingly, both males and females are viable and fertile, indicating that calaxin is not essential for fertilization in mice. The 9 + 2 axonemal structures of epithelial multicilia and sperm flagella are normal, but the formation of 9 + 0 nodal cilia is significantly disrupted. Knockout of calaxin in zebrafish also causes situs inversus due to the irregular ciliary beating of Kupffer's vesicle cilia, although the 9 + 2 axonemal structure appears to remain normal.