ArticlePloS one2017
Spatiotemporal changes in the distribution of LHFPL5 in mice cochlear hair bundles during development and in the absence of PCDH15.
Article in PloS one, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 21 papers.
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Who cites it
21 citing papers in PubMed, 29 citations in OpenAlex.
- Morphological phenotyping of the aging cochlea in inbred C57BL/6N and outbred CD1 mouse strains.Aging cell · 2025Article
- LOXHD1 is indispensable for maintaining TMC1 auditory mechanosensitive channels at the site of force transmission.Nature communications · 2024Article
- LHFPL5 is a key element in force transmission from the tip link to the hair cell mechanotransducer channel.Proceedings of the National Academy of Sciences of the United States of America · 2024Article
- LOXHD1 is indispensable for coupling auditory mechanosensitive channels to the site of force transmission.Research square · 2024Article
- The tetraspan LHFPL5 is critical to establish maximal force sensitivity of the mechanotransduction channel of cochlear hair cells.Cell reports · 2023Article
- Multiple plasma membrane reporters discern LHFPL5 region that blocks trafficking to the plasma membrane.Scientific reports · 2023Article
- Alternative splicing in shaping the molecular landscape of the cochlea.Frontiers in cell and developmental biology · 2023Review
- Review
- The conductance and organization of the TMC1-containing mechanotransducer channel complex in auditory hair cells.Proceedings of the National Academy of Sciences of the United States of America · 2022Article
- How Transmembrane Inner Ear (TMIE) plays role in the auditory system: A mystery to us.Journal of cellular and molecular medicine · 2021Review
- The Mechanosensory Transduction Machinery in Inner Ear Hair Cells.Annual review of biophysics · 2021Review
- Electron Microscopy Techniques for Investigating Structure and Composition of Hair-Cell Stereociliary Bundles.Frontiers in cell and developmental biology · 2021Article
- Deafness mutation D572N of TMC1 destabilizes TMC1 expression by disrupting LHFPL5 binding.Proceedings of the National Academy of Sciences of the United States of America · 2020Article
- TMIE Defines Pore and Gating Properties of the Mechanotransduction Channel of Mammalian Cochlear Hair Cells.Neuron · 2020Article
- Disruption ofThe Journal of neuroscience : the official journal of the Society for Neuroscience · 2020Article
- Function and Dysfunction of TMC Channels in Inner Ear Hair Cells.Cold Spring Harbor perspectives in medicine · 2019Review
- Molecular Structure of the Hair Cell Mechanoelectrical Transduction Complex.Cold Spring Harbor perspectives in medicine · 2019Review
- Ultrastructural localization of the likely mechanoelectrical transduction channel protein, transmembrane-like channel 1 (TMC1) during development of cochlear hair cells.Scientific reports · 2019Article
- Subunits of the mechano-electrical transduction channel, Tmc1/2b, require Tmie to localize in zebrafish sensory hair cells.PLoS genetics · 2019Article
- TheFrontiers in molecular neuroscience · 2019Article
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Authors and funding
5 authors at 3 institutions in 2 countries.
Funding
Abstract
Mechanosensory transduction by vertebrate hair cells depends on a protein complex at the tips of shorter stereocilia associated with mechanoelectrical transduction channels activated by tip links in the hair bundle. In mammalian hair cells, this complex includes transmembrane channel-like protein subunit 1 (TMC1), lipoma HMGIC fusion partner-like 5 protein (LHFPL5) and protocadherin 15 (PCDH15), a lower-end component of the tip link. TMC1 interacts with LHFPL5 and PCDH15 but how the complex develops to maturity, and the relationships between these proteins, remains uncertain. Here we evaluate the spatiotemporal development of LHFPL5 distributions in mouse cochlear hair bundles by immunofluorescence and immunogold transmission electron microscopy, from postnatal day 0 (P0) through P21 in wild type and PCDH15-deficient mice. At P0, hair bundles contain many short microvilli-like processes which we term unranked stereocilia, and a subset of lengthening rows, adjacent to a kinocilium. LHFPL5 is distributed throughout the bundle, including on stereocilia tips and the kinocilium. At P3, 4-to-6 rows of ranked stereocilia are evident, total LHFPL5 expression peaks, and LHFPL5 is localised to ranked stereocilia tips of all rows and to lower shaft/ankle links. By P12, the bundle has a mature pattern with 3 ranked rows but virtually no unranked stereocilia or kinocilium; LHFPL5 expression has declined and become restricted to the tips of shorter stereocilia. Throughout development from P0, expression of LHFPL5 is greater overall on apical than basal bundles, but there is, on average, an equal amount of labelling per labelled tip. In P3 mice lacking PCDH15, LHFPL5 labelling is not at the tips but is primarily on unranked stereocilia and lower lateral links. These data show that LHFPL5 is already present in the MET apparatus at P0 but requires PCDH15 at P3 to remain there. Shaft/ankle link localisation suggests it interacts with link proteins other than PCDH15.
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