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TALPID3 and ANKRD26 selectively orchestrate FBF1 localization and cilia gating

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  • Hao Yan

    (CAS Key Laboratory of Insect Developmental and Evolutionary Biology, CAS Center for Excellence in Molecular Plant Sciences, Institute of Plant Physiology and Ecology, Chinese Academy of Sciences
    University of Chinese Academy of Sciences
    Center for Reproduction and Health Development, Institute of Biomedicine and Biotechnology, Shenzhen Institutes of Advanced Technology, Chinese Academy of Sciences (CAS))

  • Chuan Chen

    (Department of Biochemistry and Molecular Biology, Mayo Clinic)

  • Huicheng Chen

    (CAS Key Laboratory of Insect Developmental and Evolutionary Biology, CAS Center for Excellence in Molecular Plant Sciences, Institute of Plant Physiology and Ecology, Chinese Academy of Sciences
    University of Chinese Academy of Sciences
    Center for Reproduction and Health Development, Institute of Biomedicine and Biotechnology, Shenzhen Institutes of Advanced Technology, Chinese Academy of Sciences (CAS))

  • Hui Hong

    (CAS Key Laboratory of Insect Developmental and Evolutionary Biology, CAS Center for Excellence in Molecular Plant Sciences, Institute of Plant Physiology and Ecology, Chinese Academy of Sciences)

  • Yan Huang

    (Department of Biochemistry and Molecular Biology, Mayo Clinic
    Mayo Translational PKD Center, Mayo Clinic)

  • Kun Ling

    (Department of Biochemistry and Molecular Biology, Mayo Clinic
    Mayo Translational PKD Center, Mayo Clinic)

  • Jinghua Hu

    (Department of Biochemistry and Molecular Biology, Mayo Clinic
    Mayo Translational PKD Center, Mayo Clinic
    Division of Nephrology and Hypertension, Mayo Clinic)

  • Qing Wei

    (Center for Reproduction and Health Development, Institute of Biomedicine and Biotechnology, Shenzhen Institutes of Advanced Technology, Chinese Academy of Sciences (CAS))

Abstract

Transition fibers (TFs) regulate cilia gating and make the primary cilium a distinct functional entity. However, molecular insights into the biogenesis of a functional cilia gate remain elusive. In a forward genetic screen in Caenorhabditis elegans, we uncover that TALP-3, a homolog of the Joubert syndrome protein TALPID3, is a TF-associated component. Genetic analysis reveals that TALP-3 coordinates with ANKR-26, the homolog of ANKRD26, to orchestrate proper cilia gating. Mechanistically, TALP-3 and ANKR-26 form a complex with key gating component DYF-19, the homolog of FBF1. Co-depletion of TALP-3 and ANKR-26 specifically impairs the recruitment of DYF-19 to TFs. Interestingly, in mammalian cells, TALPID3 and ANKRD26 also play a conserved role in coordinating the recruitment of FBF1 to TFs. We thus report a conserved protein module that specifically regulates the functional component of the ciliary gate and suggest a correlation between defective gating and ciliopathy pathogenesis.

Suggested Citation

  • Hao Yan & Chuan Chen & Huicheng Chen & Hui Hong & Yan Huang & Kun Ling & Jinghua Hu & Qing Wei, 2020. "TALPID3 and ANKRD26 selectively orchestrate FBF1 localization and cilia gating," Nature Communications, Nature, vol. 11(1), pages 1-14, December.
    • Handle: RePEc:nat:natcom:v:11:y:2020:i:1:d:10.1038_s41467-020-16042-w
    DOI: 10.1038/s41467-020-16042-w
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