Reference - Detail
| RRC ID | 84126 |
|---|---|
| Author | Barbelanne M, Lu Y, Kumar K, Zhang X, Li C, Park K, Warner A, Xu XZS, Shaham S, Leroux MR. |
| Title | C. elegans PPEF-type phosphatase (Retinal degeneration C ortholog) functions in diverse classes of cilia to regulate nematode behaviors. |
| Journal | Sci Rep |
| Abstract |
Primary (non-motile) cilia represent structurally and functionally diverse organelles whose roles as specialized cellular antenna are central to animal cell signaling pathways, sensory physiology and development. An ever-growing number of ciliary proteins, including those found in vertebrate photoreceptors, have been uncovered and linked to human disorders termed ciliopathies. Here, we demonstrate that an evolutionarily-conserved PPEF-family serine-threonine phosphatase, not functionally linked to cilia in any organism but associated with rhabdomeric (non-ciliary) photoreceptor degeneration in the Drosophila rdgC (retinal degeneration C) mutant, is a bona fide ciliary protein in C. elegans. The nematode protein, PEF-1, depends on transition zone proteins, which make up a 'ciliary gate' in the proximal-most region of the cilium, for its compartmentalization within cilia. Animals lacking PEF-1 protein function display structural defects to several types of cilia, including potential degeneration of microtubules. They also exhibit anomalies to cilium-dependent behaviors, including impaired responses to chemical, temperature, light, and noxious CO2 stimuli. Lastly, we demonstrate that PEF-1 function depends on conserved myristoylation and palmitoylation signals. Collectively, our findings broaden the role of PPEF proteins to include cilia, and suggest that the poorly-characterized mammalian PPEF1 and PPEF2 orthologs may also have ciliary functions and thus represent ciliopathy candidates. |
| Volume | 14(1) |
| Pages | 28347 |
| Published | 2024-11-16 |
| DOI | 10.1038/s41598-024-79057-z |
| PII | 10.1038/s41598-024-79057-z |
| PMID | 39550471 |
| PMC | PMC11569196 |
| MeSH | Animals Behavior, Animal Caenorhabditis elegans* / genetics Caenorhabditis elegans* / metabolism Caenorhabditis elegans Proteins* / genetics Caenorhabditis elegans Proteins* / metabolism Cilia* / metabolism Retinal Degeneration / genetics Retinal Degeneration / metabolism Retinal Degeneration / pathology |
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| Total number of mentions | 1 |
| Altmetric score changes over past 6months | 0.0 |
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| C.elegans | tm2816 |