ArticleEMBO molecular medicine2019
Clarin-2 is essential for hearing by maintaining stereocilia integrity and function.
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Article in EMBO molecular medicine, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.
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16citing papers in PubMed
2.6field-weighted citation impact, top 12% of its field
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Who cites it
16 citing papers in PubMed, 31 citations in OpenAlex.
- Molecular identity of the mechanotransduction machinery in inner ear hair cells and mechanotransduction-linked hearing loss.Fundamental research · 2026Review
- Polygenic Contribution to Sensorineural Hearing Loss Implicates Novel Risk Loci and Convergence with Congenital Hearing Loss Genes.Journal of the Association for Research in Otolaryngology : JARO · 2026Article
- Compensatory Interplay Between Clarin-1 and Clarin-2 Deafness-Associated Proteins Governs Phenotypic Variability in Hearing.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2026Article
- Myosin 7a is required for maintaining the transducing stereocilia and for force transmission to the MET channel during cochlear hair cell development.The Journal of physiology · 2026Article
- Accelerated Hydration Site Localization and Thermodynamic Profiling.Journal of chemical information and modeling · 2025Article
- Myosin-based nucleation of actin filaments contributes to stereocilia development critical for hearing.Nature communications · 2025Article
- Defects in hair cells disrupt the development of auditory peripheral circuitry.Nature communications · 2024Article
- Clarin-2 gene supplementation durably preserves hearing in a model of progressive hearing loss.Molecular therapy : the journal of the American Society of Gene Therapy · 2024Article
- Mechanoelectrical transduction-related genetic forms of hearing loss.Current opinion in physiology · 2023Article
- Mutations inProceedings of the National Academy of Sciences of the United States of America · 2022Article
- Usher syndrome IIIA: a review of the disorder and preclinical research advances in therapeutic approaches.Human genetics · 2022Review
- Neuroplastin genetically interacts with Cadherin 23 and the encoded isoform Np55 is sufficient for cochlear hair cell function and hearing.PLoS genetics · 2022Article
- A combined genome-wide association and molecular study of age-related hearing loss in H. sapiens.BMC medicine · 2021Article
- Identification of Novel Candidate Genes and Variants for Hearing Loss and Temporal Bone Anomalies.Genes · 2021Article
- Inner Ear Gene Therapies Take Off: Current Promises and Future Challenges.Journal of clinical medicine · 2020Review
- Clarin-2 is essential for hearing by maintaining stereocilia integrity and function.EMBO molecular medicine · 2019Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
32 authors at 8 institutions in 3 countries.
Lucy A DunbarMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Pranav PatniDéficits Sensoriels Progressifs, Institut Pasteur, INSERM UMR-S 1120, Sorbonne Universités, Paris, France.
Carlos AguilarMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Philomena MburuMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Laura CornsDepartment of Biomedical Science, University of Sheffield, Sheffield, UK.
Helena Rr WellsDepartment of Twin Research & Genetic Epidemiology, King's College London, London, UK.
Sedigheh DelmaghaniDéficits Sensoriels Progressifs, Institut Pasteur, INSERM UMR-S 1120, Sorbonne Universités, Paris, France.
Andrew ParkerMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Stuart JohnsonDepartment of Biomedical Science, University of Sheffield, Sheffield, UK.
Debbie WilliamsMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Christopher T EsapaMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Michelle M SimonMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Lauren ChessumMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Sherylanne NewtonMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Joanne DorningMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Prashanthini JeyarajanMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Susan MorseMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Andrea LelliGénétique et Physiologie de l'Audition, Institut Pasteur, INSERM UMR-S 1120, Collège de France, Sorbonne Universités, Paris, France.
Gemma F CodnerMary Lyon Centre, MRC Harwell Institute, Harwell, UK.
Thibault PeineauLaboratoire de Neurophysiologie de la Synapse Auditive, Université de Bordeaux, Bordeaux, France.
Suhasini R GopalDepartment of Otolaryngology - Head and Neck Surgery, University Hospitals Cleveland Medical Center, Case Western Reserve University, Cleveland, OH, USA.
Kumar N AlagramamDepartment of Otolaryngology - Head and Neck Surgery, University Hospitals Cleveland Medical Center, Case Western Reserve University, Cleveland, OH, USA.
Ronna HertzanoDepartment of Otorhinolaryngology Head and Neck Surgery, Anatomy and Neurobiology and Institute for Genome Sciences, University of Maryland School of Medicine, Baltimore, MD, USA.
Didier DulonLaboratoire de Neurophysiologie de la Synapse Auditive, Université de Bordeaux, Bordeaux, France.
Sara WellsMary Lyon Centre, MRC Harwell Institute, Harwell, UK.
Frances M WilliamsDepartment of Twin Research & Genetic Epidemiology, King's College London, London, UK.
Christine PetitGénétique et Physiologie de l'Audition, Institut Pasteur, INSERM UMR-S 1120, Collège de France, Sorbonne Universités, Paris, France.
Sally J DawsonUCL Ear Institute, University College London, London, UK.
Steve Dm BrownMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.
Walter MarcottiDepartment of Biomedical Science, University of Sheffield, Sheffield, UK.ORCID 0000-0002-8770-7628
Aziz El-AmraouiDéficits Sensoriels Progressifs, Institut Pasteur, INSERM UMR-S 1120, Sorbonne Universités, Paris, France.ORCID 0000-0003-2692-4984
Michael R BowlMammalian Genetics Unit, MRC Harwell Institute, Harwell, UK.ORCID 0000-0001-7971-445X
Mary Lyon Centre at MRC Harwell · GBInserm · FRUniversity of Sheffield · GBKing's College London · GBUniversité de Bordeaux · FRUniversity Hospitals of Cleveland · USUniversity College London · GBUniversity of Maryland, Baltimore · US
Funding
Cell Type Specific Transcriptional Cascades in Inner Ear DevelopmentR01DC013817 · NIDCD · UNIVERSITY OF MARYLAND BALTIMORE · PI HERTZANO, RONNA · 2015 to 2022
$3.6MIlluminating Neurodevelopment through Integrated Analysis and Vizualization of Multi-Omic DataR24MH114815 · NIMH · UNIVERSITY OF MARYLAND BALTIMORE · PI HERTZANO, RONNA, WHITE, OWEN R · 2018 to 2020
$2.3MMedical Research Council MC_PC_17228Medical Research Council MC_QA137853Medical Research Council MC_U142684175Medical Research Council MC_UP_1502/1Medical Research Council MC_UP_1503/2NIDCD NIH HHS R01 DC013817NIMH NIH HHS R24 MH114815Wellcome Trust 102892Wellcome Trust 102892/Z/13/Z
Abstract
Hearing relies on mechanically gated ion channels present in the actin-rich stereocilia bundles at the apical surface of cochlear hair cells. Our knowledge of the mechanisms underlying the formation and maintenance of the sound-receptive structure is limited. Utilizing a large-scale forward genetic screen in mice, genome mapping and gene complementation tests, we identified Clrn2 as a new deafness gene. The Clrn2
Indexed as
AdultAgedAnimalsCohort StudiesFemaleHair Cells, AuditoryHearingHearing LossHumansMaleMiceMice, Inbred C3HMice, Inbred C57BLMiddle AgedStereociliahair cellsmechanotransductionmouse modelsmutagenesisstereocilia
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Read underepoch 390
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.