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J Cell Biol. July 9, 2012; 198 (1): 37-45.

Control of vertebrate intraflagellar transport by the planar cell polarity effector Fuz.

Cilia play key roles in development and homeostasis, and defects in cilia structure or function lead to an array of human diseases. Ciliogenesis is accomplished by the intraflagellar transport (IFT) system, a set of proteins governing bidirectional transport of cargoes within ciliary axonemes. In this paper, we present a novel platform for in vivo analysis of vertebrate IFT dynamics. Using this platform, we show that the planar cell polarity (PCP) effector Fuz was required for normal IFT dynamics in vertebrate cilia, the first evidence directly linking PCP to the core machinery of ciliogenesis. Further, we show that Fuz played a specific role in trafficking of retrograde, but not anterograde, IFT proteins. These data place Fuz in the small group of known IFT effectors outside the core machinery and, additionally, identify Fuz as a novel cytoplasmic effector that differentiates between the retrograde and anterograde IFT complexes.

PubMed ID: 22778277
PMC ID: PMC3392940
Article link: J Cell Biol.
Grant support: R01 GM074104 NIGMS NIH HHSHoward Hughes Medical Institute

Genes referenced: fuz ift20 ift43 map7 plin1 tbx2

Disease Ontology references: ciliopathy

External Resources: GO Terms referenced: intraciliary anterograde transport [+]

Article Images: [+] show captions

Arts, 2011, Pubmed [+]

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