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Originally published as MBC in Press, 10.1091/mbc.E06-06-0571 on October 4, 2006

Vol. 17, Issue 12, 5053-5062, December 2006

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The WD Repeat-containing Protein IFTA-1 Is Required for Retrograde Intraflagellar TransportFormula Formula

Oliver E. Blacque*, Chunmei Li*, Peter N. Inglis*, Muneer A. Esmail*, Guangshuo Ou{dagger}, Allan K. Mah*, David L. Baillie*, Jonathan M. Scholey{dagger}, and Michel R. Leroux*

*Department of Molecular Biology and Biochemistry, Simon Fraser University, Burnaby, British Columbia V5A 1S6, Canada; and {dagger}Center for Genetics and Development, Section of Molecular and Cellular Biology, University of California, Davis, Davis, CA 95616

Submitted July 3, 2006; Revised August 14, 2006; Accepted September 22, 2006
Monitoring Editor: Trisha Davis

The assembly and maintenance of cilia require intraflagellar transport (IFT), a microtubule-dependent bidirectional motility of multisubunit protein complexes along ciliary axonemes. Defects in IFT and the functions of motile or sensory cilia are associated with numerous human ailments, including polycystic kidney disease and Bardet–Biedl syndrome. Here, we identify a novel Caenorhabditis elegans IFT gene, IFT-associated gene 1 (ifta-1), which encodes a WD repeat-containing protein with strong homology to a mammalian protein of unknown function. Both the C. elegans and human IFTA-1 proteins localize to the base of cilia, and in C. elegans, IFTA-1 can be observed to undergo IFT. IFTA-1 is required for the function and assembly of cilia, because a C. elegans ifta-1 mutant displays chemosensory abnormalities and shortened cilia with prominent ciliary accumulations of core IFT machinery components that are indicative of retrograde transport defects. Analyses of C. elegans IFTA-1 localization/motility along bbs mutant cilia, where anterograde IFT assemblies are destabilized, and in a che-11 IFT gene mutant, demonstrate that IFTA-1 is closely associated with the IFT particle A subcomplex, which is implicated in retrograde IFT. Together, our data indicate that IFTA-1 is a novel IFT protein that is required for retrograde transport along ciliary axonemes.


Formula Formula The online version of this contains supplemental material at MBC Online (http://www.molbiolcell.org).

This article was published online ahead of print in MBC in Press (http://www.molbiolcell.org/cgi/doi/10.1091/mbc.E06-06-0571 on October 4, 2006.

Address correspondence to: Michel R. Leroux (leroux{at}sfu.ca)

Abbreviations used: BBS, Bardet–Biedl syndrome; IFT, intraflagellar transport; IFT-A, intraflagellar transport particle A; IFT-B, intraflagellar transport particle B.




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