Cep131-Cep162 and Cby-Fam92 complexes cooperatively maintain Cep290 at the basal body and contribute to ciliogenesis initiation
Copyright: © 2024 Wu et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited..
Cilia play critical roles in cell signal transduction and organ development. Defects in cilia function result in a variety of genetic disorders. Cep290 is an evolutionarily conserved ciliopathy protein that bridges the ciliary membrane and axoneme at the basal body (BB) and plays critical roles in the initiation of ciliogenesis and TZ assembly. How Cep290 is maintained at BB and whether axonemal and ciliary membrane localized cues converge to determine the localization of Cep290 remain unknown. Here, we report that the Cep131-Cep162 module near the axoneme and the Cby-Fam92 module close to the membrane synergistically control the BB localization of Cep290 and the subsequent initiation of ciliogenesis in Drosophila. Concurrent deletion of any protein of the Cep131-Cep162 module and of the Cby-Fam92 module leads to a complete loss of Cep290 from BB and blocks ciliogenesis at its initiation stage. Our results reveal that the first step of ciliogenesis strictly depends on cooperative and retroactive interactions between Cep131-Cep162, Cby-Fam92 and Cep290, which may contribute to the complex pathogenesis of Cep290-related ciliopathies.
| Errataetall: |
UpdateOf: PLoS Biol. 2020 Dec 28;18(12):e3001034. - PMID 33370260 |
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| Medienart: |
E-Artikel |
| Erscheinungsjahr: |
2024 |
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| Erschienen: |
2024 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:22 |
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| Enthalten in: |
PLoS biology - 22(2024), 3 vom: 05. März, Seite e3002330 |
| Sprache: |
Englisch |
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| Beteiligte Personen: |
Wu, Zhimao [VerfasserIn] |
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Date Completed 07.03.2024 Date Revised 07.03.2024 published: Electronic-eCollection UpdateOf: PLoS Biol. 2020 Dec 28;18(12):e3001034. - PMID 33370260 Citation Status MEDLINE |
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| doi: |
10.1371/journal.pbio.3002330 |
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NLM369319486 |
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| 520 | |a Copyright: © 2024 Wu et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. | ||
| 520 | |a Cilia play critical roles in cell signal transduction and organ development. Defects in cilia function result in a variety of genetic disorders. Cep290 is an evolutionarily conserved ciliopathy protein that bridges the ciliary membrane and axoneme at the basal body (BB) and plays critical roles in the initiation of ciliogenesis and TZ assembly. How Cep290 is maintained at BB and whether axonemal and ciliary membrane localized cues converge to determine the localization of Cep290 remain unknown. Here, we report that the Cep131-Cep162 module near the axoneme and the Cby-Fam92 module close to the membrane synergistically control the BB localization of Cep290 and the subsequent initiation of ciliogenesis in Drosophila. Concurrent deletion of any protein of the Cep131-Cep162 module and of the Cby-Fam92 module leads to a complete loss of Cep290 from BB and blocks ciliogenesis at its initiation stage. Our results reveal that the first step of ciliogenesis strictly depends on cooperative and retroactive interactions between Cep131-Cep162, Cby-Fam92 and Cep290, which may contribute to the complex pathogenesis of Cep290-related ciliopathies | ||
| 650 | 4 | |a Journal Article | |
| 700 | 1 | |a Chen, Huicheng |e verfasserin |4 aut | |
| 700 | 1 | |a Zhang, Yingying |e verfasserin |4 aut | |
| 700 | 1 | |a Wang, Yaru |e verfasserin |4 aut | |
| 700 | 1 | |a Wang, Qiaoling |e verfasserin |4 aut | |
| 700 | 1 | |a Augière, Céline |e verfasserin |4 aut | |
| 700 | 1 | |a Hou, Yanan |e verfasserin |4 aut | |
| 700 | 1 | |a Fu, Yuejun |e verfasserin |4 aut | |
| 700 | 1 | |a Peng, Ying |e verfasserin |4 aut | |
| 700 | 1 | |a Durand, Bénédicte |e verfasserin |4 aut | |
| 700 | 1 | |a Wei, Qing |e verfasserin |4 aut | |
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