6 results found
    1. Cell Biology
    2. Developmental Biology

    Inactivation of Invs/Nphp2 in renal epithelial cells drives infantile nephronophthisis like phenotypes in mouse

    Yuanyuan Li, Wenyan Xu ... Zhaoxia Sun
    Genetic and chemical genetic analysis pinpoint defective epithelial cells as the driver for NPHP like phenotypes in Invs mutants, demonstrate genetic interaction between Invs and Ift88 and identify the HDAC inhibitor valproic acid as a suppressor for Invs mutant phenotypes.
    1. Stem Cells and Regenerative Medicine

    Stem cells and fluid flow drive cyst formation in an invertebrate excretory organ

    Hanh Thi-Kim Vu, Jochen C Rink ... Alejandro Sánchez Alvarado
    Planarians provide evidence for a common evolutionary origin of vertebrate and invertebrate excretory systems and provide a novel experimental model to study human kidney diseases.
    1. Cell Biology

    Organization, functions, and mechanisms of the BBSome in development, ciliopathies, and beyond

    Xiaoyu Tian, Huijie Zhao, Jun Zhou
    Theoretical analysis of the current literature reveals the structural assembly, transport regulation, and molecular functions of the BBSome, emphasizing its roles in cilium-related processes.
    1. Cell Biology
    2. Developmental Biology

    Dzip1 and Fam92 form a ciliary transition zone complex with cell type specific roles in Drosophila

    Jean-André Lapart, Marco Gottardo ... Bénédicte Durand
    Genetic analyses in Drosophila establish the functional hierarchy of the ciliary transition zone module, Dzip1/Fam92/Cby, and reveal tissue specific variations in basal body anchoring pathways in fly ciliated tissues.
    1. Cell Biology

    A bioactive peptide amidating enzyme is required for ciliogenesis

    Dhivya Kumar, Daniela Strenkert ... Betty A Eipper
    Analysis of Chlamydomonas, planaria and mice reveals a novel and unanticipated role for a peptide amidating enzyme in primary and motile ciliary assembly through effects on post-Golgi trafficking.
    1. Biochemistry and Chemical Biology

    Reserpine maintains photoreceptor survival in retinal ciliopathy by resolving proteostasis imbalance and ciliogenesis defects

    Holly Y Chen, Manju Swaroop ... Anand Swaroop
    High-throughput screening of over 6000 drugs using cells and retina tissue with a CEP290 ciliopathy mutation identified a small molecule, reserpine, which enhanced photoreceptor survival in retinal organoids and in a mouse disease model by partially restoring balance in proteostasis.

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