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    Ciliary proteins Bbs8 and Ift20 promote planar cell polarity in the cochlea

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    Authors
    May-Simera, Helen L.
    Petralia, Ronald S.
    Montcouquiol, Mireille
    Wang, Ya-Xian
    Szarama, Katherine B.
    Liu, Yun
    Lin, Weichun
    Deans, Michael R.
    Pazour, Gregory J.
    Kelley, Matthew W.
    UMass Chan Affiliations
    Program in Molecular Medicine
    Document Type
    Journal Article
    Publication Date
    2015-02-01
    Keywords
    Animals
    Carrier Proteins
    Cell Polarity
    Cilia
    Cochlea
    Hair Cells, Auditory
    Immunohistochemistry
    Immunoprecipitation
    Mice
    Mice, Knockout
    Microscopy, Electron, Scanning
    Microscopy, Electron, Transmission
    Microtubule-Associated Proteins
    Nerve Tissue Proteins
    Actin
    Cilia
    Cochlea
    Microtubules
    Mouse
    Polarity
    Biochemistry
    Cell Biology
    Developmental Biology
    Molecular Biology
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    Link to Full Text
    http://dx.doi.org/10.1242/dev.113696
    Abstract
    Primary cilia have been implicated in the generation of planar cell polarity (PCP). However, variations in the severity of polarity defects in different cilia mutants, coupled with recent demonstrations of non-cilia-related actions of some cilia genes, make it difficult to determine the basis of these polarity defects. To address this issue, we evaluated PCP defects in cochlea from a selection of mice with mutations in cilia-related genes. Results indicated notable PCP defects, including mis-oriented hair cell stereociliary bundles, in Bbs8 and Ift20 single mutants that are more severe than in other cilia gene knockouts. In addition, deletion of either Bbs8 or Ift20 results in disruptions in asymmetric accumulation of the core PCP molecule Vangl2 in cochlear cells, suggesting a role for Bbs8 and/or Ift20, possibly upstream of core PCP asymmetry. Consistent with this, co-immunoprecipitation experiments indicate direct interactions of Bbs8 and Ift20 with Vangl2. We observed localization of Bbs and Ift proteins to filamentous actin as well as microtubules. This could implicate these molecules in selective trafficking of membrane proteins upstream of cytoskeletal reorganization, and identifies new roles for cilia-related proteins in cochlear PCP.
    Source
    Development. 2015 Feb 1;142(3):555-66. doi: 10.1242/dev.113696. Link to article on publisher's site
    DOI
    10.1242/dev.113696
    Permanent Link to this Item
    http://hdl.handle.net/20.500.14038/44471
    PubMed ID
    25605782
    Related Resources
    Link to Article in PubMed
    Rights

    Publisher PDF posted after 12 months as allowed by the publisher's author rights policy at http://dev.biologists.org/content/rights-permissions.

    ae974a485f413a2113503eed53cd6c53
    10.1242/dev.113696
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