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Article

Harmonin mutations cause mechanotransduction defects in cochlear hair cells.

Neuron - 14 May 2009

Grillet Nicolas, Xiong Wei, Reynolds Anna, Kazmierczak Piotr, Sato Takashi, Lillo Concepcion, Dumont Rachel A, Hintermann Edith, Sczaniecka Anna, Schwander Martin, Williams David, Kachar Bechara, Gillespie Peter G, Müller Ulrich

Abstract excerpt

In hair cells, mechanotransduction channels are gated by tip links, the extracellular filaments that consist of cadherin 23 (CDH23) and protocadherin 15 (PCDH15) and connect the stereocilia of each hair cell. However, which molecules mediate cadherin function at tip links is not known. Here we show that the PDZ-domain protein harmonin is a component of the upper tip-link density (UTLD), where CDH23 inserts into...

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Methodology Data sources Editorial policy Privacy About Contact Helena Bioinformatics

Share to Hair Cells, Auditory, Inner

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Source

Open in PubMed Open full text

Identifiers

PMID: 19447093
DOI: 10.1016/j.neuron.2009.04.006
PMCID: PMC2691393

Genes

CDH23
PCDH15

Provenance

PubMed metadata and Literature Corpus relationships. Corpus snapshot updated 7 Sept 2026. Manifest publication-graph-cohort-2026-09-07.1.

Source receipt: pubmed, retrieved 7 Sept 2026.

Related research

Semantic proximity does not establish scientific evidence.

Research trail→

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Harmonin mutations cause mechanotransduction defects in cochlear hair cells.PMID 19447093Read ↗

Cadherin 23 and protocadherin 15 interact to form tip-link filaments in sensory hair cells.Semantic proximity Read ↗

Tip links in hair cells: molecular composition and role in hearing lossSemantic proximity Read ↗

Mutations in cadherin 23 affect tip links in zebrafish sensory hair cells.Semantic proximity Read ↗

MAGI-1, A Candidate Stereociliary Scaffolding Protein, Associates with the Tip-Link Component Cadherin 23Semantic proximity Read ↗

Cadherins and mechanotransduction by hair cells.Semantic proximity Read ↗

Loxhd1 Mutations Cause Mechanotransduction Defects in Cochlear Hair Cells.Semantic proximity Read ↗

Mutations in protocadherin 15 and cadherin 23 affect tip links and mechanotransduction in mammalian sensory hair cells.Semantic proximity Read ↗

Presence of interstereocilial links in waltzer mutants suggests Cdh23 is not essential for tip link formation.Semantic proximity Read ↗

Noddy, a mouse harboring a missense mutation in protocadherin-15, reveals the impact of disrupting a critical interaction site between tip-link cadherins in inner ear hair cells.Semantic proximity Read ↗

Compound heterozygosity of the functionally null Cdh23(v-ngt) and hypomorphic Cdh23(ahl) alleles leads to early-onset progressive hearing loss in mice.Semantic proximity Read ↗

The CD2 isoform of protocadherin-15 is an essential component of the tip-link complex in mature auditory hair cells.Semantic proximity Read ↗

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