238 results found
    1. Developmental Biology

    Primary cilia deficiency in neural crest cells models anterior segment dysgenesis in mouse

    Céline Portal et al.
    Primary cilia of neural crest-derived cells mediate Indian hedgehog-induced signal transduction in the periocular mesenchyme and are required for normal anterior segment development.
    1. Cell Biology

    A pathway for Parkinson’s Disease LRRK2 kinase to block primary cilia and Sonic hedgehog signaling in the brain

    Herschel S Dhekne et al.
    Pathogenic LRRK2kinase requires Rab10 and RILPL1 to block primary cilia formation, shortening cilia on cholinergic neurons needed for a hedgehog driven circuit that supports dopaminergic neurons in mouse brain.
    1. Cell Biology
    2. Neuroscience

    TTBK2 and primary cilia are essential for the connectivity and survival of cerebellar Purkinje neurons

    Emily Bowie, Sarah C Goetz
    Signaling at the primary cilium is important to sustain the morphology, connectivity, and survival of a key neural population within the brain.
    1. Cell Biology

    Dopamine receptors reveal an essential role of IFT-B, KIF17, and Rab23 in delivering specific receptors to primary cilia

    Alison Leaf, Mark Von Zastrow
    The mechanism of signaling receptor delivery to primary cilia involves a specific cellular role of a Rab protein that is critical for vertebrate development.
    1. Cell Biology

    Dynein-2 intermediate chains play crucial but distinct roles in primary cilia formation and function

    Laura Vuolo et al.
    Genetic knock-outs of the dynein-2 intermediate chains reveals that both are essential for correct cilia function and transition zone organization, but play different functions in the assembly of dynein-2 motor and in primary cilia formation.
    1. Structural Biology and Molecular Biophysics

    Cryo-EM structure of the polycystin 2-l1 ion channel

    Raymond E Hulse et al.
    The selectivity characteristics of PC-2L1, which make it a principal source of calcium in primary cilia function, is presented and advances understanding of the similarity and differences of TRPP ion channels.
    1. Structural Biology and Molecular Biophysics
    2. Cell Biology

    Polycystin-2 is an essential ion channel subunit in the primary cilium of the renal collecting duct epithelium

    Xiaowen Liu et al.
    The primary cilia polycystin proteins, polycystin-1 and polycystin-2, affect cilia length in the kidney collecting duct epithelia, but only polycystin-2 is required for the functional ion channel in this organelle.
    1. Cell Biology
    2. Developmental Biology

    LUZP1, a novel regulator of primary cilia and the actin cytoskeleton, is a contributing factor in Townes-Brocks Syndrome

    Laura Bozal-Basterra et al.
    LUZP1 is required for proper cilia and cytoskeleton formation.
    1. Cell Biology
    2. Developmental Biology

    ARL13B regulates Sonic hedgehog signaling from outside primary cilia

    Eduardo D Gigante et al.
    ARL13B regulates cell ciliation and cilia length from within cilia and Sonic hedgehog response from outside of cilia indicating the two processes can be spatially uncoupled.
    1. Developmental Biology

    Notch signalling maintains Hedgehog responsiveness via a Gli-dependent mechanism during spinal cord patterning in zebrafish

    Craig T Jacobs, Peng Huang
    Genetic analyses combining photoconvertible cell signalling reporters with gain- and loss-of function manipulations reveal a novel role for Notch signalling in controlling Hedgehog response in neural progenitor cells.

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