Male germ cell-associated kinase is required for axoneme formation during ciliogenesis in zebrafish photoreceptors.
Hung-Ju ChiangYuko NishiwakiWei-Chieh Jerry ChiangIchiro MasaiPublished in: Disease models & mechanisms (2024)
Vertebrate photoreceptors are highly specialized retinal neurons that have cilium-derived membrane organelles called outer segments, 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 lacked outer segments and underwent apoptosis. Interestingly, zebrafish mak mutants failed to generate axonemes during photoreceptor ciliogenesis, whereas basal bodies were 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 was found to be critical in ciliary axoneme development and photoreceptor survival. Thus, Mak is required for ciliogenesis and outer segment formation in zebrafish photoreceptors to ensure intracellular protein transport and photoreceptor survival.
Keyphrases
- germ cell
- protein kinase
- tyrosine kinase
- wild type
- magnetic resonance
- computed tomography
- magnetic resonance imaging
- palliative care
- cell proliferation
- optical coherence tomography
- endoplasmic reticulum stress
- machine learning
- spinal cord
- adipose tissue
- electronic health record
- insulin resistance
- high fat diet induced
- artificial intelligence
- skeletal muscle
- loop mediated isothermal amplification