Reference - Detail
| RRC ID | 53317 |
|---|---|
| Author | Wei Q, Zhang Y, Schouteden C, Zhang Y, Zhang Q, Dong J, Wonesch V, Ling K, Dammermann A, Hu J. |
| Title | The hydrolethalus syndrome protein HYLS-1 regulates formation of the ciliary gate. |
| Journal | Nat Commun |
| Abstract |
Transition fibres (TFs), together with the transition zone (TZ), are basal ciliary structures thought to be crucial for cilium biogenesis and function by acting as a ciliary gate to regulate selective protein entry and exit. Here we demonstrate that the centriolar and basal body protein HYLS-1, the C. elegans orthologue of hydrolethalus syndrome protein 1, is required for TF formation, TZ organization and ciliary gating. Loss of HYLS-1 compromises the docking and entry of intraflagellar transport (IFT) particles, ciliary gating for both membrane and soluble proteins, and axoneme assembly. Additional depletion of the TF component DYF-19 in hyls-1 mutants further exacerbates TZ anomalies and completely abrogates ciliogenesis. Our data support an important role for HYLS-1 and TFs in establishment of the ciliary gate and underline the importance of selective protein entry for cilia assembly. |
| Volume | 7 |
| Pages | 12437 |
| Published | 2016-8-18 |
| DOI | 10.1038/ncomms12437 |
| PII | ncomms12437 |
| PMID | 27534274 |
| PMC | PMC4992140 |
| MeSH | Animals Caenorhabditis elegans Caenorhabditis elegans Proteins / metabolism* Cilia / metabolism* Cilia / ultrastructure Flagella / metabolism Gene Deletion Hand Deformities, Congenital / metabolism* Heart Defects, Congenital / metabolism* Hydrocephalus / metabolism* Models, Biological Organogenesis |
| IF | 12.121 |
| Times Cited | 10 |
| Altmetric score |
オルトメトリクス指標項目
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| The most frequently cited source | News |
| Total number of mentions | 9 |
| Altmetric score changes over past 6months | 0.0 |
| Resource | |
| C.elegans | tm3067 tm2705 tm2547 tm3100 |