| RRC ID |
53317
|
| 著者 |
Wei Q, Zhang Y, Schouteden C, Zhang Y, Zhang Q, Dong J, Wonesch V, Ling K, Dammermann A, Hu J.
|
| タイトル |
The hydrolethalus syndrome protein HYLS-1 regulates formation of the ciliary gate.
|
| ジャーナル |
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.
|
| 巻・号 |
7
|
| ページ |
12437
|
| 公開日 |
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
|
| 引用数 |
10
|
| リソース情報 |
| 線虫 |
tm3067
tm2705
tm2547
tm3100 |
