ArticlePloS one2012
Role for a novel Usher protein complex in hair cell synaptic maturation.
Article in PloS one, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers.
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Who cites it
33 citing papers in PubMed, 50 citations in OpenAlex.
- Adhesion G-Protein Coupled Receptors in Neurological and Psychiatric Disorders.Journal of psychiatry and brain science · 2025Article
- Morphological phenotyping of the aging cochlea in inbred C57BL/6N and outbred CD1 mouse strains.Aging cell · 2025Article
- Outcomes of cochlear implants in patients withFrontiers in genetics · 2025Article
- Piplartine attenuates aminoglycoside-induced TRPV1 activity and protects from hearing loss in mice.Science translational medicine · 2024Article
- Expression of the human usherin c.2299delG mutation leads to early-onset auditory loss and stereocilia disorganization.Communications biology · 2023Article
- Adenylyl cyclase 6 plays a minor role in the mouse inner ear and retina.Scientific reports · 2023Article
- Protocadherin 15 suppresses oligodendrocyte progenitor cell proliferation and promotes motility through distinct signalling pathways.Communications biology · 2022Article
- Immunohistochemistry localises myosin-7a to cochlear efferent boutons.Wellcome open research · 2022Article
- G protein-coupled receptors in cochlea: Potential therapeutic targets for hearing loss.Frontiers in molecular neuroscience · 2022Review
- Usher syndrome type 1-associated gene, pcdh15b, is required for photoreceptor structural integrity in zebrafish.Disease models & mechanisms · 2021Article
- [The value of genetic diagnosis of deafness in evaluating the prognosis of cochlear implantation].Lin chuang er bi yan hou tou jing wai ke za zhi = Journal of clinical otorhinolaryngology head and neck surgery · 2021Article
- Clarin-1 expression in adult mouse and human retina highlights a role of Müller glia in Usher syndrome.The Journal of pathology · 2020Article
- Hair Cell Afferent Synapses: Function and Dysfunction.Cold Spring Harbor perspectives in medicine · 2019Review
- Clarin-2 is essential for hearing by maintaining stereocilia integrity and function.EMBO molecular medicine · 2019Article
- Integrin α8 and Pcdh15 act as a complex to regulate cilia biogenesis in sensory cells.Journal of cell science · 2017Article
- Modeling and Preventing Progressive Hearing Loss in Usher Syndrome III.Scientific reports · 2017Article
- Cadherin 23-C Regulates Microtubule Networks by Modifying CAMSAP3's Function.Scientific reports · 2016Article
- Plasma Membrane Targeting of Protocadherin 15 Is Regulated by the Golgi-Associated Chaperone Protein PIST.Neural plasticity · 2016Article
- AAV-Mediated Clarin-1 Expression in the Mouse Retina: Implications for USH3A Gene Therapy.PloS one · 2016Article
- Emerging Roles of BAI Adhesion-GPCRs in Synapse Development and Plasticity.Neural plasticity · 2016Review
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Authors and funding
7 authors at 3 institutions in 1 country.
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Abstract
The molecular mechanisms underlying hair cell synaptic maturation are not well understood. Cadherin-23 (CDH23), protocadherin-15 (PCDH15) and the very large G-protein coupled receptor 1 (VLGR1) have been implicated in the development of cochlear hair cell stereocilia, while clarin-1 has been suggested to also play a role in synaptogenesis. Mutations in CDH23, PCDH15, VLGR1 and clarin-1 cause Usher syndrome, characterized by congenital deafness, vestibular dysfunction and retinitis pigmentosa. Here we show developmental expression of these Usher proteins in afferent spiral ganglion neurons and hair cell synapses. We identify a novel synaptic Usher complex comprised of clarin-1 and specific isoforms of CDH23, PCDH15 and VLGR1. To establish the in vivo relevance of this complex, we performed morphological and quantitative analysis of the neuronal fibers and their synapses in the Clrn1-/- mouse, which was generated by incomplete deletion of the gene. These mice showed a delay in neuronal/synaptic maturation by both immunostaining and electron microscopy. Analysis of the ribbon synapses in Ames waltzer(av3J) mice also suggests a delay in hair cell synaptogenesis. Collectively, these results show that, in addition to the well documented role for Usher proteins in stereocilia development, Usher protein complexes comprised of specific protein isoforms likely function in synaptic maturation as well.
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