論文 - 詳細
| RRC ID | 88138 |
|---|---|
| 著者 | Wang S, Li M, Chen G, Chen Z, Lei K, Ökten Z, Xie S, Zhou T, Li W, Ou G. |
| タイトル | Joubert syndrome 26 protein enforces compartmentalized motility of a ciliary kinesin. |
| ジャーナル | Proc Natl Acad Sci U S A |
| Abstract |
Cilia are essential cellular antennae that rely on precise motor-driven transport to assemble and function. Two kinesin-2 motors-kinesin-II and OSM-3 in Caenorhabditis elegans-cooperate to transport cargo along cilia, with kinesin-II operating in the middle segment and OSM-3 taking over distally. However, how kinesin-II is spatially confined to prevent its invasion into distal regions remains unclear. Here, we identify Joubert syndrome 26 protein (JBTS-26) as a critical regulator of this motor handover. JBTS-26 localizes to axonemal doublet microtubules in the ciliary middle segment, where it competes with kinesin-II for binding to the IFT-B subunit OSM-5/IFT88. This competition displaces kinesin-II from IFT particles, enabling OSM-3 to assume distal transport. Loss of JBTS-26 allows kinesin-II to invade the distal cilium and slows down IFT. Conversely, JBTS-26 overexpression accelerates IFT by prematurely releasing kinesin-II. Our findings reveal a mechanism for compartmentalized motor regulation and link defective motor handover to ciliopathy pathogenesis. |
| 巻・号 | 122(47) |
| ページ | e2504374122 |
| 公開日 | 2025-11-25 |
| DOI | 10.1073/pnas.2504374122 |
| PMID | 41264249 |
| PMC | PMC12663925 |
| MeSH | Abnormalities, Multiple Animals Caenorhabditis elegans / genetics Caenorhabditis elegans / metabolism Caenorhabditis elegans Proteins* / genetics Caenorhabditis elegans Proteins* / metabolism Cerebellum / abnormalities Cerebellum / metabolism Cilia* / metabolism Eye Abnormalities* / genetics Eye Abnormalities* / metabolism Humans Kidney Diseases, Cystic* / genetics Kidney Diseases, Cystic* / metabolism Kinesins* / genetics Kinesins* / metabolism Microtubules / metabolism Retina* / abnormalities Retina* / metabolism |
| オルトメトリクス指標 |
オルトメトリクス指標項目
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| 最多言及媒体 | X(Twitter) |
| 各媒体での言及数の合計 | 3 |
| 過去6か月間でのオルトメトリクス指標の変動値 | 0.0 |
| リソース情報 | |
| 線虫 | tm324 |