A WDR47 homolog facilitates ciliogenesis by modulating intraflagellar transport

Cilia are conserved organelles found in many cell types in eukaryotes, and their dysfunction causes defects in environmental sensing and signaling transduction; such defects are termed ciliopathies. Distinct cilia have cell-specific morphologies and exert distinct functions. However, the underlying...

Full description

Saved in:
Bibliographic Details
Published inJournal of cell science Vol. 135; no. 24
Main Authors Song, Chun-Xue, Zeng, Xian-Ting, Zeng, Wan-Xin, Liu, Rong, Tong, Xia-Jing, Li, Qian
Format Journal Article
LanguageEnglish
Published England 15.12.2022
Subjects
Animals
Biological Transport
Caenorhabditis elegans
Cilia - metabolism
Ciliopathies - metabolism
Mammals
Neurons - metabolism
Chemosensory neuron
Intraflagellar transport
WD40-repeat protein
Cilia
Online AccessGet full text

Cover

More Information
Summary:Cilia are conserved organelles found in many cell types in eukaryotes, and their dysfunction causes defects in environmental sensing and signaling transduction; such defects are termed ciliopathies. Distinct cilia have cell-specific morphologies and exert distinct functions. However, the underlying mechanisms of cell-specific ciliogenesis and regulation are unclear. Here, we identified a WD40-repeat (WDR) protein, NMTN-1 (the homolog of mammalian WDR47), and show that it is specifically required for ciliogenesis of AWB chemosensory neurons in C. elegans. NMTN-1 is expressed in the AWB chemosensory neuron pair, and is enriched at the basal body (BB) of the AWB cilia. Knockout of nmtn-1 causes abnormal AWB neuron cilia morphology, structural integrity, and induces aberrant AWB-mediated aversive behaviors. We further demonstrate that nmtn-1 deletion affects movement of intraflagellar transport (IFT) particles and their cargo delivery in AWB neurons. Our results indicate that NMTN-1 is essential for AWB neuron ciliary morphology and function, which reveal a novel mechanism for cell-specific ciliogenesis. Given that WDR47/NMTN-1 is conserved in mammals, our findings may help understanding of the process of cell-specific ciliogenesis and provide insights for treating ciliopathies.
ISSN:0021-9533
1477-9137
DOI:10.1242/jcs.260303