PUBLICATION
Calaxin stabilizes the docking of outer arm dyneins onto ciliary doublet microtubule in vertebrates
- Authors
- Yamaguchi, H., Morikawa, M., Kikkawa, M.
- ID
- ZDB-PUB-230415-50
- Date
- 2023
- Source
- eLIFE 12: (Journal)
- Registered Authors
- Kikkawa, Masahide
- Keywords
- cell biology, molecular biophysics, structural biology, zebrafish
- MeSH Terms
-
- Animals
- Axoneme/metabolism
- Cilia/metabolism
- Dyneins*/metabolism
- Humans
- Male
- Microtubules/metabolism
- Mutation
- Zebrafish*/genetics
- PubMed
- 37057896 Full text @ Elife
Citation
Yamaguchi, H., Morikawa, M., Kikkawa, M. (2023) Calaxin stabilizes the docking of outer arm dyneins onto ciliary doublet microtubule in vertebrates. eLIFE. 12:.
Abstract
Outer arm dynein (OAD) is the main force generator of ciliary beating. Although OAD loss is the most frequent cause of human primary ciliary dyskinesia, the docking mechanism of OAD onto the ciliary doublet microtubule (DMT) remains elusive in vertebrates. Here, we analyzed the functions of Calaxin/Efcab1 and Armc4, the two of five components of vertebrate OAD-DC (docking complex), using zebrafish spermatozoa and cryo-electron tomography. Mutation of armc4 caused complete loss of OAD, whereas mutation of calaxin caused only partial loss of OAD. Detailed structural analysis revealed that calaxin-/- OADs are tethered to DMT through DC components other than Calaxin, and that recombinant Calaxin can autonomously rescue the deficient DC structure and the OAD instability. Our data demonstrate the discrete roles of Calaxin and Armc4 in the OAD-DMT interaction, suggesting the stabilizing process of OAD docking onto DMT in vertebrates.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping