PUBLICATION
Calaxin is required for cilia-driven determination of vertebrate laterality
- Authors
- Sasaki, K., Shiba, K., Nakamura, A., Kawano, N., Satouh, Y., Yamaguchi, H., Morikawa, M., Shibata, D., Yanase, R., Jokura, K., Nomura, M., Miyado, M., Takada, S., Ueno, H., Nonaka, S., Baba, T., Ikawa, M., Kikkawa, M., Miyado, K., Inaba, K.
- ID
- ZDB-PUB-190627-16
- Date
- 2019
- Source
- Communications biology 2: 226 (Journal)
- Registered Authors
- Keywords
- Ciliogenesis, Dynein
- MeSH Terms
-
- Animals
- Animals, Genetically Modified
- Brain/metabolism
- Brain/ultrastructure
- Cilia/metabolism*
- Cilia/ultrastructure
- Ciliary Motility Disorders/metabolism
- Ependyma/metabolism
- Ependyma/ultrastructure
- Flagella/metabolism
- Flagella/ultrastructure
- Mice, Inbred C57BL
- Movement/physiology
- Trachea/metabolism
- Trachea/ultrastructure
- Zebrafish
- PubMed
- 31240264 Full text @ Commun Biol
Citation
Sasaki, K., Shiba, K., Nakamura, A., Kawano, N., Satouh, Y., Yamaguchi, H., Morikawa, M., Shibata, D., Yanase, R., Jokura, K., Nomura, M., Miyado, M., Takada, S., Ueno, H., Nonaka, S., Baba, T., Ikawa, M., Kikkawa, M., Miyado, K., Inaba, K. (2019) Calaxin is required for cilia-driven determination of vertebrate laterality. Communications biology. 2:226.
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.
Errata / Notes
This article is corrected by ZDB-PUB-220906-161 .
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping