PUBLICATION
Male germ cell-associated kinase (MAK) is required for axoneme formation during ciliogenesis in zebrafish photoreceptors
- Authors
- Chiang, H.J., Nishiwaki, Y., Chiang, W.C., Masai, I.
- ID
- ZDB-PUB-240530-15
- Date
- 2024
- Source
- Disease models & mechanisms 17(7): (Journal)
- Registered Authors
- Chiang, Hung-Ju, Chiang, Wei-Chieh, Masai, Ichiro, Nishiwaki, Yuko
- Keywords
- Ciliogenesis, Ciliopathy, MAK, Photoreceptor degeneration, Zebrafish
- MeSH Terms
-
- Animals
- Apoptosis
- Axoneme*/metabolism
- Basal Bodies/metabolism
- Cell Survival
- Cilia*/metabolism
- Male
- Mutation*/genetics
- Photoreceptor Cells, Vertebrate/metabolism
- Protein Serine-Threonine Kinases*/metabolism
- Retinal Rod Photoreceptor Cells/metabolism
- Serine-Threonine Kinase 3
- Zebrafish*/embryology
- Zebrafish Proteins*/genetics
- Zebrafish Proteins*/metabolism
- PubMed
- 38813692 Full text @ Dis. Model. Mech.
Citation
Chiang, H.J., Nishiwaki, Y., Chiang, W.C., Masai, I. (2024) Male germ cell-associated kinase (MAK) is required for axoneme formation during ciliogenesis in zebrafish photoreceptors. Disease models & mechanisms. 17(7):.
Abstract
Vertebrate photoreceptors are highly specialized retinal neurons that have cilium-derived membrane organelles called outer segments (OS), which function as platforms for phototransduction. Male germ cell-associated kinase (MAK) is a cilium-associated serine/threonine kinase, and its genetic mutation causes photoreceptor degeneration in mice and retinitis pigmentosa in humans. However, the role of MAK in photoreceptors is not fully understood. Here, we report that zebrafish mak mutants show rapid photoreceptor degeneration during embryonic development. In mak mutants, both cone and rod photoreceptors completely lack OSs and undergo apoptosis. Interestingly, zebrafish mak mutants fail to generate axonemes during photoreceptor ciliogenesis, whereas basal bodies are specified. These data suggest that MAK contributes to axoneme development in zebrafish, in contrast to mouse Mak mutants, which have elongated photoreceptor axonemes. Furthermore, the kinase activity of MAK is critical in ciliary axoneme development and photoreceptor survival. Thus, MAK is required for ciliogenesis and OS formation in zebrafish photoreceptors to ensure intracellular protein transport and photoreceptor survival.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping