ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2011
Polycystin-1 is required for stereocilia structure but not for mechanotransduction in inner ear hair cells.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 25 papers.
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Who cites it
25 citing papers in PubMed, 37 citations in OpenAlex.
- Distribution of ciliary adaptor proteins tubby and TULP3 in the organ of Corti.Frontiers in neuroscience · 2023Article
- Article
- Hearing impairment due toDisease models & mechanisms · 2020Article
- Diversity of cilia-based mechanosensory systems and their functions in marine animal behaviour.Philosophical transactions of the Royal Society of London. Series B, Biological sciences · 2020Review
- Elmod3 knockout leads to progressive hearing loss and abnormalities in cochlear hair cell stereocilia.Human molecular genetics · 2019Article
- Tprn is essential for the integrity of stereociliary rootlet in cochlear hair cells in mice.Frontiers of medicine · 2019Article
- Molecular Structure of the Hair Cell Mechanoelectrical Transduction Complex.Cold Spring Harbor perspectives in medicine · 2019Review
- Neural circuitry of a polycystin-mediated hydrodynamic startle response for predator avoidance.eLife · 2018Article
- Knock-In Mice with Myo3a Y137C Mutation Displayed Progressive Hearing Loss and Hair Cell Degeneration in the Inner Ear.Neural plasticity · 2018Article
- TRPV6, TRPM6 and TRPM7 Do Not Contribute to Hair-Cell Mechanotransduction.Frontiers in cellular neuroscience · 2018Article
- Characterization of the Transcriptomes of Lgr5+ Hair Cell Progenitors and Lgr5- Supporting Cells in the Mouse Cochlea.Frontiers in molecular neuroscience · 2017Article
- Are TMCs the Mechanotransduction Channels of Vertebrate Hair Cells?The Journal of neuroscience : the official journal of the Society for Neuroscience · 2016Article
- The polycystin complex mediates Wnt/Ca(2+) signalling.Nature cell biology · 2016Article
- Article
- Whole Mount Dissection and Immunofluorescence of the Adult Mouse Cochlea.Journal of visualized experiments : JoVE · 2016Article
- Review
- Mechanosensory Genes Pkd1 and Pkd2 Contribute to the Planar Polarization of Brain Ventricular Epithelium.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2015Article
- LKB1 Is Required for the Development and Maintenance of Stereocilia in Inner Ear Hair Cells in Mice.PloS one · 2015Article
- Article
- Spontaneous hair cell regeneration in the neonatal mouse cochlea in vivo.Development (Cambridge, England) · 2014Article
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Authors and funding
14 authors at 6 institutions in 2 countries.
Funding
Abstract
The polycystic kidney disease-1 (Pkd1) gene encodes a large transmembrane protein (polycystin-1, or PC-1) that is reported to function as a fluid flow sensor in the kidney. As a member of the transient receptor potential family, PC-1 has also been hypothesized to play a role in the elusive mechanoelectrical transduction (MET) channel in inner ear hair cells. Here, we analyze two independent mouse models of PC-1, a knock-in (KI) mutant line and a hair cell-specific inducible Cre-mediated knock-out line. Both models exhibit normal MET channel function at neonatal ages despite hearing loss and ultrastructural abnormalities of sterecilia that remain properly polarized at adult ages. These findings demonstrate that PC-1 plays an essential role in stereocilia structure and maintenance but not directly in MET channel function or planar cell polarity. We also demonstrate that PC-1 is colocalized with F-actin in hair cell stereocilia in vivo, using a hemagglutinin-tagged PC-1 KI mouse model, and in renal epithelial cell microvilli in vitro. These results not only demonstrate a novel role for PC-1 in the cochlea, but also suggest insight into the development of polycystic kidney disease.
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Registered trials
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.