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• W3178158594 abstract "Sound transduction occurs in the hair bundle, the apical compartment of sensory hair cells in the inner ear. The hair bundle is formed of actin-based stereocilia aligned in rows of graded heights. It was previously shown that the GNAI-GPSM2 complex is part of a developmental blueprint that defines the polarized organization of the apical cytoskeleton in hair cells, including stereocilia distribution and elongation. Here, we report a role for multiple PDZ domain (MPDZ) protein during apical hair cell morphogenesis in mouse. We show that MPDZ is enriched at the hair cell apical membrane along with MAGUK p55 subfamily member 5 (MPP5/PALS1) and the Crumbs protein CRB3. MPDZ is required there to maintain the proper segregation of apical blueprint proteins, including GNAI-GPSM2. Loss of the blueprint coincides with misaligned stereocilia placement in Mpdz mutant hair cells, and results in permanently misshapen hair bundles. Graded molecular and structural defects along the cochlea can explain the profile of hearing loss in Mpdz mutants, where deficits are most severe at high frequencies." @default.
• W3178158594 created "2021-07-19" @default.
• W3178158594 creator A5052214589 @default.
• W3178158594 creator A5070051988 @default.
• W3178158594 date "2021-07-15" @default.
• W3178158594 modified "2023-10-06" @default.
• W3178158594 title "Multiple PDZ domain protein maintains patterning of the apical cytoskeleton in sensory hair cells" @default.
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• W3178158594 doi "https://doi.org/10.1242/dev.199549" @default.
• W3178158594 hasPubMedCentralId "https://www.ncbi.nlm.nih.gov/pmc/articles/8326923" @default.
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• W3178158594 hasPublicationYear "2021" @default.
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