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KIF13B establishes a CAV1-enriched microdomain at the ciliary transition zone to promote Sonic hedgehog signalling

Author

Listed:
  • Kenneth B. Schou

    (Section of Cell Biology and Physiology)

  • Johanne B. Mogensen

    (Section of Cell Biology and Physiology)

  • Stine K. Morthorst

    (Section of Cell Biology and Physiology)

  • Brian S. Nielsen

    (Section of Cell Biology and Physiology
    Present address: Department of Neuroscience and Pharmacology, University of Copenhagen, Blegdamsvej 3, Copenhagen N DK-2200, Denmark)

  • Aiste Aleliunaite

    (Section of Cell Biology and Physiology
    Present address: Department of Cellular and Molecular Medicine, University of Copenhagen, Blegdamsvej 3, Copenhagen N DK-2200, Denmark)

  • Andrea Serra-Marques

    (Cell Biology, Faculty of Science, Utrecht University
    Present address: Department of Cell & Tissue Biology, University of California San Francisco, San Francisco, 513 Parnassus Avenue, San Francisco, California 94143, USA)

  • Nicoline Fürstenberg

    (Section of Cell Biology and Physiology)

  • Sophie Saunier

    (Inserm UMR-1163, Laboratory of Hereditary Kidney diseases
    Paris Descartes Sorbonne Paris Cité University, Imagine Institut)

  • Albane A. Bizet

    (Inserm UMR-1163, Laboratory of Hereditary Kidney diseases
    Paris Descartes Sorbonne Paris Cité University, Imagine Institut)

  • Iben R. Veland

    (Section of Cell Biology and Physiology
    Present address: Biotech Research and Innovation Centre, Department of Health and Medical Sciences, University of Copenhagen, Ole Maaløes Vej 5, Copenhagen N DK-2200, Denmark)

  • Anna Akhmanova

    (Cell Biology, Faculty of Science, Utrecht University)

  • Søren T. Christensen

    (Section of Cell Biology and Physiology)

  • Lotte B. Pedersen

    (Section of Cell Biology and Physiology)

Abstract

Ciliary membrane composition is controlled by transition zone (TZ) proteins such as RPGRIP1, RPGRIPL and NPHP4, which are vital for balanced coordination of diverse signalling systems like the Sonic hedgehog (Shh) pathway. Activation of this pathway involves Shh-induced ciliary accumulation of Smoothened (SMO), which is disrupted by disease-causing mutations in TZ components. Here we identify kinesin-3 motor protein KIF13B as a novel member of the RPGRIP1N-C2 domain-containing protein family and show that KIF13B regulates TZ membrane composition and ciliary SMO accumulation. KIF13B is upregulated during ciliogenesis and is recruited to the ciliary base by NPHP4, which binds to two distinct sites in the KIF13B tail region, including an RPGRIP1N-C2 domain. KIF13B and NPHP4 are both essential for establishment of a CAV1 membrane microdomain at the TZ, which in turn is required for Shh-induced ciliary SMO accumulation. Thus KIF13B is a novel regulator of ciliary TZ configuration, membrane composition and Shh signalling.

Suggested Citation

  • Kenneth B. Schou & Johanne B. Mogensen & Stine K. Morthorst & Brian S. Nielsen & Aiste Aleliunaite & Andrea Serra-Marques & Nicoline Fürstenberg & Sophie Saunier & Albane A. Bizet & Iben R. Veland & A, 2017. "KIF13B establishes a CAV1-enriched microdomain at the ciliary transition zone to promote Sonic hedgehog signalling," Nature Communications, Nature, vol. 8(1), pages 1-15, April.
    • Handle: RePEc:nat:natcom:v:8:y:2017:i:1:d:10.1038_ncomms14177
    DOI: 10.1038/ncomms14177
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