ArticleNature communications2019
PKHD1L1 is a coat protein of hair-cell stereocilia and is required for normal hearing.
Article in Nature communications, 2019. 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, 46 citations in OpenAlex.
- Activated Platelets Express GPI-Anchored Fibrocystin-L (PKHD1L1), from Granules, Absent or Deficient in Paroxysmal Nocturnal Hemoglobinuria.International journal of molecular sciences · 2026Article
- Phylogenomic analysis of vocal fold length evolution reveals links between vocal and auditory systems in terrestrial mammals.Molecular biology and evolution · 2026Article
- Spatially Resolved Heterogeneity of Lymphatic Vasculature in the Adult Lung-Brief Report.Arteriosclerosis, thrombosis, and vascular biology · 2026Article
- GPR156 is required in sensory hair cells for proper auditory and vestibular function.Scientific reports · 2026Article
- VASCilia is an open-source, deep learning-based tool for 3D analysis of cochlear hair cell stereocilia bundles.PLoS biology · 2026Article
- Cell-specific delivery of GJB2 restores auditory function in mouse models of DFNB1 deafness and mediates appropriate expression in NHP cochlea.Nature communications · 2025Article
- TMEM145 is a key stereociliary component in the link structures of outer hair cells and mediates the secretion of stereocilin and tubby.Nature communications · 2025Article
- VASCilia (Vision Analysis StereoCilia): A Napari Plugin for Deep Learning-Based 3D Analysis of Cochlear Hair Cell Stereocilia Bundles.bioRxiv : the preprint server for biology · 2025Article
- Research Progress on MET, Tip Link, and Stereocilia Complex with Special Reference to Zebrafish.International journal of molecular sciences · 2025Review
- Analysis of thyroid carcinoma composition and spatial architecture in the progression of dedifferentiation, lymphatic metastasis, and gastric metastasis.Journal of translational medicine · 2025Article
- Integrative analysis of single-cell and transcriptome sequencing with experimental validation reveals PKHD1L1 as a novel biomarker in lung adenocarcinoma.Scientific reports · 2025Article
- PKHD1L1 is required for stereocilia bundle maintenance, durable hearing function and resilience to noise exposure.Communications biology · 2024Article
- LOXHD1 is indispensable for maintaining TMC1 auditory mechanosensitive channels at the site of force transmission.Nature communications · 2024Article
- SUB-immunogold-SEM reveals nanoscale distribution of submembranous epitopes.Nature communications · 2024Article
- Keratan sulfate, an electrosensory neurosentient bioresponsive cell instructive glycosaminoglycan.Glycobiology · 2024Review
- PKHD1L1 is required for stereocilia bundle maintenance, durable hearing function and resilience to noise exposure.bioRxiv : the preprint server for biology · 2024Article
- PKHD1L1, a gene involved in the stereocilia coat, causes autosomal recessive nonsyndromic hearing loss.Human genetics · 2024Article
- SUB-Immunogold-SEM reveals nanoscale distribution of submembranous epitopes.Research square · 2024Article
- LOXHD1 is indispensable for coupling auditory mechanosensitive channels to the site of force transmission.Research square · 2024Article
- Systematic Multiomic Analysis ofInternational journal of molecular sciences · 2023Article
Corrections and comments
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Authors and funding
6 authors at 3 institutions in 1 country.
Funding
Abstract
The bundle of stereocilia on inner ear hair cells responds to subnanometer deflections produced by sound or head movement. Stereocilia are interconnected by a variety of links and also carry an electron-dense surface coat. The coat may contribute to stereocilia adhesion or protect from stereocilia fusion, but its molecular identity remains unknown. From a database of hair-cell-enriched translated proteins, we identify Polycystic Kidney and Hepatic Disease 1-Like 1 (PKHD1L1), a large, mostly extracellular protein of 4249 amino acids with a single transmembrane domain. Using serial immunogold scanning electron microscopy, we show that PKHD1L1 is expressed at the tips of stereocilia, especially in the high-frequency regions of the cochlea. PKHD1L1-deficient mice lack the surface coat at the upper but not lower regions of stereocilia, and they develop progressive hearing loss. We conclude that PKHD1L1 is a component of the surface coat and is required for normal hearing in mice.
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