A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to cilia
Intraflagellar transport (IFT) is a highly conserved mechanism for motor-driven transport of cargo within cilia, but how this cargo is selectively transported to cilia is unclear. WDR35/IFT121 is a component of the IFT-A complex best known for its role in ciliary retrograde transport. In the absence...
| Main Authors: | , , , , , , , , , , , , |
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| Format: | Article |
| Language: | English |
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eLife Sciences Publications Ltd
2021-11-01
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| Series: | eLife |
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| Online Access: | https://elifesciences.org/articles/69786 |
| _version_ | 1811227502202322944 |
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| author | Tooba Quidwai Jiaolong Wang Emma A Hall Narcis A Petriman Weihua Leng Petra Kiesel Jonathan N Wells Laura C Murphy Margaret A Keighren Joseph A Marsh Esben Lorentzen Gaia Pigino Pleasantine Mill |
| author_facet | Tooba Quidwai Jiaolong Wang Emma A Hall Narcis A Petriman Weihua Leng Petra Kiesel Jonathan N Wells Laura C Murphy Margaret A Keighren Joseph A Marsh Esben Lorentzen Gaia Pigino Pleasantine Mill |
| author_sort | Tooba Quidwai |
| collection | DOAJ |
| description | Intraflagellar transport (IFT) is a highly conserved mechanism for motor-driven transport of cargo within cilia, but how this cargo is selectively transported to cilia is unclear. WDR35/IFT121 is a component of the IFT-A complex best known for its role in ciliary retrograde transport. In the absence of WDR35, small mutant cilia form but fail to enrich in diverse classes of ciliary membrane proteins. In Wdr35 mouse mutants, the non-core IFT-A components are degraded and core components accumulate at the ciliary base. We reveal deep sequence homology of WDR35 and other IFT-A subunits to α and ß′ COPI coatomer subunits and demonstrate an accumulation of ‘coat-less’ vesicles that fail to fuse with Wdr35 mutant cilia. We determine that recombinant non-core IFT-As can bind directly to lipids and provide the first in situ evidence of a novel coat function for WDR35, likely with other IFT-A proteins, in delivering ciliary membrane cargo necessary for cilia elongation. |
| first_indexed | 2024-04-12T09:42:33Z |
| format | Article |
| id | doaj.art-a1c4b4199b71449c8dc737a7d2b3ac26 |
| institution | Directory Open Access Journal |
| issn | 2050-084X |
| language | English |
| last_indexed | 2024-04-12T09:42:33Z |
| publishDate | 2021-11-01 |
| publisher | eLife Sciences Publications Ltd |
| record_format | Article |
| series | eLife |
| spelling | doaj.art-a1c4b4199b71449c8dc737a7d2b3ac262022-12-22T03:38:02ZengeLife Sciences Publications LtdeLife2050-084X2021-11-011010.7554/eLife.69786A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to ciliaTooba Quidwai0https://orcid.org/0000-0001-5248-9010Jiaolong Wang1Emma A Hall2Narcis A Petriman3https://orcid.org/0000-0002-3189-7530Weihua Leng4Petra Kiesel5Jonathan N Wells6https://orcid.org/0000-0003-3700-020XLaura C Murphy7Margaret A Keighren8Joseph A Marsh9Esben Lorentzen10https://orcid.org/0000-0001-6493-7220Gaia Pigino11Pleasantine Mill12https://orcid.org/0000-0001-5218-134XMRC Human Genetics Unit, Institute of Genetics and Cancer, University of Edinburgh, Edinburgh, United KingdomDepartment of Molecular Biology and Genetics, Aarhus University, Aarhus, DenmarkMRC Human Genetics Unit, Institute of Genetics and Cancer, University of Edinburgh, Edinburgh, United KingdomDepartment of Molecular Biology and Genetics, Aarhus University, Aarhus, DenmarkMax Planck Institute of Molecular Cell Biology and Genetics, Dresden, GermanyMax Planck Institute of Molecular Cell Biology and Genetics, Dresden, GermanyMRC Human Genetics Unit, Institute of Genetics and Cancer, University of Edinburgh, Edinburgh, United KingdomMRC Human Genetics Unit, Institute of Genetics and Cancer, University of Edinburgh, Edinburgh, United KingdomMRC Human Genetics Unit, Institute of Genetics and Cancer, University of Edinburgh, Edinburgh, United KingdomMRC Human Genetics Unit, Institute of Genetics and Cancer, University of Edinburgh, Edinburgh, United KingdomDepartment of Molecular Biology and Genetics, Aarhus University, Aarhus, DenmarkMax Planck Institute of Molecular Cell Biology and Genetics, Dresden, Germany; Human Technopole, Milan, ItalyMRC Human Genetics Unit, Institute of Genetics and Cancer, University of Edinburgh, Edinburgh, United KingdomIntraflagellar transport (IFT) is a highly conserved mechanism for motor-driven transport of cargo within cilia, but how this cargo is selectively transported to cilia is unclear. WDR35/IFT121 is a component of the IFT-A complex best known for its role in ciliary retrograde transport. In the absence of WDR35, small mutant cilia form but fail to enrich in diverse classes of ciliary membrane proteins. In Wdr35 mouse mutants, the non-core IFT-A components are degraded and core components accumulate at the ciliary base. We reveal deep sequence homology of WDR35 and other IFT-A subunits to α and ß′ COPI coatomer subunits and demonstrate an accumulation of ‘coat-less’ vesicles that fail to fuse with Wdr35 mutant cilia. We determine that recombinant non-core IFT-As can bind directly to lipids and provide the first in situ evidence of a novel coat function for WDR35, likely with other IFT-A proteins, in delivering ciliary membrane cargo necessary for cilia elongation.https://elifesciences.org/articles/69786ciliaintraflagellar transport ciliary pocketmembrane cargosvesicular trafficcoatomer |
| spellingShingle | Tooba Quidwai Jiaolong Wang Emma A Hall Narcis A Petriman Weihua Leng Petra Kiesel Jonathan N Wells Laura C Murphy Margaret A Keighren Joseph A Marsh Esben Lorentzen Gaia Pigino Pleasantine Mill A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to cilia eLife cilia intraflagellar transport ciliary pocket membrane cargos vesicular traffic coatomer |
| title | A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to cilia |
| title_full | A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to cilia |
| title_fullStr | A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to cilia |
| title_full_unstemmed | A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to cilia |
| title_short | A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to cilia |
| title_sort | wdr35 dependent coat protein complex transports ciliary membrane cargo vesicles to cilia |
| topic | cilia intraflagellar transport ciliary pocket membrane cargos vesicular traffic coatomer |
| url | https://elifesciences.org/articles/69786 |
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