ArticleDevelopment (Cambridge, England)2021
Multiple PDZ domain protein maintains patterning of the apical cytoskeleton in sensory hair cells.
Article in Development (Cambridge, England), 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.
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Who cites it
10 citing papers in PubMed, 13 citations in OpenAlex.
- An ancient evolutionary origin for IL-1 cytokines.Nature ecology & evolution · 2026Article
- The Enigmatic Presence of Actin Rings, Actin Cables and Ectopic Hair Cells in the Vestibular Maculae of Mice and Humans.Journal of the Association for Research in Otolaryngology : JARO · 2026Article
- Article
- Large-scale annotated dataset for cochlear hair cell detection and classification.Scientific data · 2024Article
- Large-scale annotated dataset for cochlear hair cell detection and classification.bioRxiv : the preprint server for biology · 2023Article
- The hair cell analysis toolbox is a precise and fully automated pipeline for whole cochlea hair cell quantification.PLoS biology · 2023Article
- RGS12 polarizes the GPSM2-GNAI complex to organize and elongate stereocilia in sensory hair cells.Science advances · 2022Article
- Novel m.4268T>C mutation in the mitochondrial tRNAWorld journal of clinical cases · 2022Article
- Article
- Conserved and Divergent Principles of Planar Polarity Revealed by Hair Cell Development and Function.Frontiers in neuroscience · 2021Review
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Authors and funding
2 authors at 2 institutions in 1 country.
Funding
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.
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