PUBLICATION
Heme-binding protein CYB5D1 is a radial spoke component required for coordinated ciliary beating
- Authors
- Zhao, L., Xie, H., Kang, Y., Lin, Y., Liu, G., Sakato-Antoku, M., Patel-King, R.S., Wang, B., Wan, C., King, S.M., Zhao, C., Huang, K.
- ID
- ZDB-PUB-210421-1
- Date
- 2021
- Source
- Proceedings of the National Academy of Sciences of the United States of America 118(17): (Journal)
- Registered Authors
- Zhao, Chengtian
- Keywords
- Chlamydomonas, cilia, motility, redox, zebrafish
- MeSH Terms
-
- Animals
- Axoneme/metabolism
- Chlamydomonas/metabolism
- Chlamydomonas/physiology
- Cilia/metabolism*
- Cilia/physiology*
- Cytochromes b5/metabolism*
- Cytochromes b5/physiology
- Dyneins/metabolism
- Flagella/metabolism
- Flagella/physiology
- Heme-Binding Proteins/metabolism
- Heme-Binding Proteins/physiology
- Microtubules/metabolism
- Mutation
- Zebrafish/metabolism
- PubMed
- 33875586 Full text @ Proc. Natl. Acad. Sci. USA
Citation
Zhao, L., Xie, H., Kang, Y., Lin, Y., Liu, G., Sakato-Antoku, M., Patel-King, R.S., Wang, B., Wan, C., King, S.M., Zhao, C., Huang, K. (2021) Heme-binding protein CYB5D1 is a radial spoke component required for coordinated ciliary beating. Proceedings of the National Academy of Sciences of the United States of America. 118(17):.
Abstract
Coordinated beating is crucial for the function of multiple cilia. However, the molecular mechanism is poorly understood. Here, we characterize a conserved ciliary protein CYB5D1 with a heme-binding domain and a cordon-bleu ubiquitin-like domain. Mutation or knockdown of Cyb5d1 in zebrafish impaired coordinated ciliary beating in the otic vesicle and olfactory epithelium. Similarly, the two flagella of an insertional mutant of the CYB5D1 ortholog in Chlamydomonas (Crcyb5d1) showed an uncoordinated pattern due to a defect in the cis-flagellum. Biochemical analyses revealed that CrCYB5D1 is a radial spoke stalk protein that binds heme only under oxidizing conditions. Lack of CrCYB5D1 resulted in a reductive shift in flagellar redox state and slowing down of the phototactic response. Treatment of Crcyb5d1 with oxidants restored coordinated flagellar beating. Taken together, these data suggest that CrCYB5D1 may integrate environmental and intraciliary signals and regulate the redox state of cilia, which is crucial for the coordinated beating of multiple cilia.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping