KIF13B establishes a CAV1-enriched microdomain at the ciliary transition zone to promote Sonic hedgehog signalling.
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KIF13B establishes a CAV1-enriched microdomain at the ciliary transition zone to promote Sonic hedgehog signalling. / Schou, Kenneth Bødtker; Mogensen, Johanne Bay; Morthorst, Stine Kjær; Nielsen, Brian Skriver; Aleliunaite, Aiste; Serra-Marques, Andrea; Fürstenberg, Nicoline; Saunier, Sophie; Bizet, Albane A.; Veland, Iben Rønn; Akhmanova, Anna; Christensen, Søren Tvorup; Pedersen, Lotte Bang.
I: Nature Communications, Bind 8, 14177, 30.01.2017.Publikation: Bidrag til tidsskrift › Tidsskriftartikel › Forskning › fagfællebedømt
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T1 - KIF13B establishes a CAV1-enriched microdomain at the ciliary transition zone to promote Sonic hedgehog signalling.
AU - Schou, Kenneth Bødtker
AU - Mogensen, Johanne Bay
AU - Morthorst, Stine Kjær
AU - Nielsen, Brian Skriver
AU - Aleliunaite, Aiste
AU - Serra-Marques, Andrea
AU - Fürstenberg, Nicoline
AU - Saunier, Sophie
AU - Bizet, Albane A.
AU - Veland, Iben Rønn
AU - Akhmanova, Anna
AU - Christensen, Søren Tvorup
AU - Pedersen, Lotte Bang
PY - 2017/1/30
Y1 - 2017/1/30
N2 - 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.
AB - 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.
U2 - 10.1038/ncomms14177
DO - 10.1038/ncomms14177
M3 - Journal article
C2 - 28134340
VL - 8
JO - Nature Communications
JF - Nature Communications
SN - 2041-1723
M1 - 14177
ER -
ID: 173088008