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A WDR35-dependent coat protein complex transports ciliary membrane cargo vesicles to cilia

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  • Tooba Quidwai, Molecular Medicine Centre, MRC Institute of Genetics and Molecular Medicine, Western General Hospital, Edinburgh, United Kingdom.
  • ,
  • Jiaolong Wang
  • Emma A Hall, University of Edinburgh
  • ,
  • Narcis A Petriman
  • Weihua Leng, Max Planck Institute of Molecular Cell Biology and Genetics
  • ,
  • Petra Kiesel, Max Planck Institute of Molecular Cell Biology and Genetics
  • ,
  • Jonathan N Wells, Molecular Medicine Centre, MRC Institute of Genetics and Molecular Medicine, Western General Hospital, Edinburgh, United Kingdom.
  • ,
  • Laura C Murphy, The University of Edinburgh
  • ,
  • Margaret A Keighren, University of Edinburgh
  • ,
  • Joseph A Marsh, Molecular Medicine Centre, MRC Institute of Genetics and Molecular Medicine, Western General Hospital, Edinburgh, United Kingdom.
  • ,
  • Esben Lorentzen
  • Gaia Pigino, Human Technopole
  • ,
  • Pleasantine Mill, University of Edinburgh

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 which 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.

OriginalsprogEngelsk
Artikelnummere69786
TidsskrifteLife
Vol/bind10
Antal sider39
ISSN2050-084X
DOI
StatusUdgivet - nov. 2021

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