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    1. Developmental Biology

    Dysregulation of sonic hedgehog signaling causes hearing loss in ciliopathy mouse models

    Kyeong-Hye Moon et al.
    Developmental defects of the cochlea caused by dysregulation of sonic hedgehog signaling are the potential etiology for hearing loss in a group of ciliopathies with defective ciliogenesis.
    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.
    1. Cell Biology
    2. Structural Biology and Molecular Biophysics

    The heteromeric PC-1/PC-2 polycystin complex is activated by the PC-1 N-terminus

    Kotdaji Ha et al.
    Soluble fragments cleaved from the N-terminus of PC-1 activate the PC-1/PC-2 heteromeric polycystin channel, describing for the first time that the N-terminus itself is an endogenous ligand.
    1. Biochemistry and Chemical Biology
    2. Cell Biology

    ARL3 activation requires the co-GEF BART and effector-mediated turnover

    Yasmin ElMaghloob et al.
    BART is a co-GEF for ARL3 and maintains the active ARL3-GTP until it is recycled by ARL3 effectors.
    1. Cell Biology

    Functional partitioning of a liquid-like organelle during assembly of axonemal dyneins

    Chanjae Lee et al.
    DynAPs are liquid-like organelles that are partitioned into functional sub-domains.
    1. Cell Biology

    A liquid-like organelle at the root of motile ciliopathy

    Ryan L Huizar et al.
    DynAPs reveal that biological phase separation provides the organizing principle for the complex process of dynein motor assembly in cells with motile cilia.
    1. Developmental Biology

    T-box3 is a ciliary protein and regulates stability of the Gli3 transcription factor to control digit number

    Uchenna Emechebe et al.
    The transcription and splicing factor T-box3 is present in primary cilia, regulates multiple aspects of limb development, and interacts with members of the protein complex required for the stability and processing of the Gli3 transcription factor.
    1. Cell Biology

    Nanobody-directed targeting of optogenetic tools to study signaling in the primary cilium

    Jan N Hansen et al.
    Trafficking of optogenetic tools to primary cilia using nanobodies allows to study cAMP signaling with spatial and temporal resolution independent of the cell body.
    1. Cell Biology

    Single molecule imaging reveals a major role for diffusion in the exploration of ciliary space by signaling receptors

    Fan Ye et al.
    Membrane receptor proteins move through primary cilia largely by passive diffusion rather than by active intraflagellar transport.
    1. Developmental Biology

    A transient role of the ciliary gene Inpp5e in controlling direct versus indirect neurogenesis in cortical development

    Kerstin Hasenpusch-Theil et al.
    Primary-cilia-mediated processing of the Gli3 transcription factor enables the formation of subtypes of projection neurons in appropriate numbers during the development of the cerebral cortex.