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

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, Anna Akhmanova, Søren T. Christensen and Lotte B. Pedersen ()
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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
Aiste Aleliunaite: Section of Cell Biology and Physiology
Andrea Serra-Marques: Cell Biology, Faculty of Science, Utrecht University
Nicoline Fürstenberg: Section of Cell Biology and Physiology
Sophie Saunier: Inserm UMR-1163, Laboratory of Hereditary Kidney diseases
Albane A. Bizet: Inserm UMR-1163, Laboratory of Hereditary Kidney diseases
Iben R. Veland: Section of Cell Biology and Physiology
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

Nature Communications, 2017, vol. 8, issue 1, 1-15

Abstract: 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.

Date: 2017
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DOI: 10.1038/ncomms14177

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