Calaxin is required for cilia-driven determination of vertebrate laterality

Keita Sasaki, Kogiku Shiba, Akihiro Nakamura, Natsuko Kawano, Yuhkoh Satouh, Hiroshi Yamaguchi, Motohiro Morikawa, Daisuke Shibata, Ryuji Yanase, Kei Jokura, Mami Nomura, Mami Miyado, Shuji Takada, Hironori Ueno, Shigenori Nonaka, Tadashi Baba, Masahito Ikawa, Masahide Kikkawa, Kenji Miyado, Kazuo Inaba

Research output: Contribution to journalArticlepeer-review

14 Citations (Scopus)

Abstract

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.

Original languageEnglish
Number of pages1
JournalCommunications biology
Volume2
Issue number1
DOIs
Publication statusPublished - 20 Jun 2019

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