Primary ciliogenesis requires the distal appendage component Cep123
Summary Primary cilium formation is initiated at the distal end of the mother centriole in a highly co-ordinated manner. This requires the capping of the distal end of the mother centriole with a ciliary vesicle and the anchoring of the basal body (mother centriole) to the cell cortex, both of which...
| Main Authors: | , , , , , |
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| Format: | Article |
| Language: | English |
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The Company of Biologists
2013-04-01
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| Series: | Biology Open |
| Subjects: | |
| Online Access: | http://bio.biologists.org/content/2/6/535 |
| _version_ | 1818656654593359872 |
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| author | James E. Sillibourne Ilse Hurbain Thierry Grand-Perret Bruno Goud Phong Tran Michel Bornens |
| author_facet | James E. Sillibourne Ilse Hurbain Thierry Grand-Perret Bruno Goud Phong Tran Michel Bornens |
| author_sort | James E. Sillibourne |
| collection | DOAJ |
| description | Summary
Primary cilium formation is initiated at the distal end of the mother centriole in a highly co-ordinated manner. This requires the capping of the distal end of the mother centriole with a ciliary vesicle and the anchoring of the basal body (mother centriole) to the cell cortex, both of which are mediated by the distal appendages. Here, we show that the distal appendage protein Cep123 (Cep89/CCDC123) is required for the assembly, but not the maintenance, of a primary cilium. In the absence of Cep123 ciliary vesicle formation fails, suggesting that it functions in the early stages of primary ciliogenesis. Consistent with such a role, Cep123 interacts with the centriolar satellite proteins PCM-1, Cep290 and OFD1, all of which play a role in primary ciliogenesis. These interactions are mediated by a domain in the C-terminus of Cep123 (400–783) that overlaps the distal appendage-targeting domain (500–600). Together, the data implicate Cep123 as a new player in the primary ciliogenesis pathway and expand upon the role of the distal appendages in this process. |
| first_indexed | 2024-12-17T03:29:01Z |
| format | Article |
| id | doaj.art-2c60f4a629c04eba8af73723d9788796 |
| institution | Directory Open Access Journal |
| issn | 2046-6390 |
| language | English |
| last_indexed | 2024-12-17T03:29:01Z |
| publishDate | 2013-04-01 |
| publisher | The Company of Biologists |
| record_format | Article |
| series | Biology Open |
| spelling | doaj.art-2c60f4a629c04eba8af73723d97887962022-12-21T22:05:18ZengThe Company of BiologistsBiology Open2046-63902013-04-012653554510.1242/bio.2013445720134457Primary ciliogenesis requires the distal appendage component Cep123James E. Sillibourne0Ilse Hurbain1Thierry Grand-Perret2Bruno Goud3Phong Tran4Michel Bornens5 Institut Curie, Centre de Recherche/UMR144 du Centre Nationale de la Recherche Scientifique, 26 rue d'Ulm, F-75248 Paris Cedex 05, France Cell and Tissue Imaging Facility-IBiSA, CNRS UMR 144, Paris F-75248, France Oncology Discovery, Janssen Research and Development, Turnhoutseweg 30, B-2340 Beerse, Belgium Institut Curie, Centre de Recherche/UMR144 du Centre Nationale de la Recherche Scientifique, 26 rue d'Ulm, F-75248 Paris Cedex 05, France Institut Curie, Centre de Recherche/UMR144 du Centre Nationale de la Recherche Scientifique, 26 rue d'Ulm, F-75248 Paris Cedex 05, France Institut Curie, Centre de Recherche/UMR144 du Centre Nationale de la Recherche Scientifique, 26 rue d'Ulm, F-75248 Paris Cedex 05, France Summary Primary cilium formation is initiated at the distal end of the mother centriole in a highly co-ordinated manner. This requires the capping of the distal end of the mother centriole with a ciliary vesicle and the anchoring of the basal body (mother centriole) to the cell cortex, both of which are mediated by the distal appendages. Here, we show that the distal appendage protein Cep123 (Cep89/CCDC123) is required for the assembly, but not the maintenance, of a primary cilium. In the absence of Cep123 ciliary vesicle formation fails, suggesting that it functions in the early stages of primary ciliogenesis. Consistent with such a role, Cep123 interacts with the centriolar satellite proteins PCM-1, Cep290 and OFD1, all of which play a role in primary ciliogenesis. These interactions are mediated by a domain in the C-terminus of Cep123 (400–783) that overlaps the distal appendage-targeting domain (500–600). Together, the data implicate Cep123 as a new player in the primary ciliogenesis pathway and expand upon the role of the distal appendages in this process.http://bio.biologists.org/content/2/6/535Centriolar satellitesCentrosomeDistal appendagesPrimary cilium |
| spellingShingle | James E. Sillibourne Ilse Hurbain Thierry Grand-Perret Bruno Goud Phong Tran Michel Bornens Primary ciliogenesis requires the distal appendage component Cep123 Biology Open Centriolar satellites Centrosome Distal appendages Primary cilium |
| title | Primary ciliogenesis requires the distal appendage component Cep123 |
| title_full | Primary ciliogenesis requires the distal appendage component Cep123 |
| title_fullStr | Primary ciliogenesis requires the distal appendage component Cep123 |
| title_full_unstemmed | Primary ciliogenesis requires the distal appendage component Cep123 |
| title_short | Primary ciliogenesis requires the distal appendage component Cep123 |
| title_sort | primary ciliogenesis requires the distal appendage component cep123 |
| topic | Centriolar satellites Centrosome Distal appendages Primary cilium |
| url | http://bio.biologists.org/content/2/6/535 |
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