ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2016
Absence of Neuroplastin-65 Affects Synaptogenesis in Mouse Inner Hair Cells and Causes Profound Hearing Loss.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.
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Who cites it
23 citing papers in PubMed, 33 citations in OpenAlex.
- CaProceedings of the National Academy of Sciences of the United States of America · 2026Article
- De novo variants in NPTN cause a neurodevelopmental disorder with autism and neuroplastin-PMCA hypofunction.Genome medicine · 2026Article
- Neuroplastin 65 deficiency leads to the impairment of visual function through affecting ribbon synapse in retina of mice.Frontiers in cellular neuroscience · 2025Article
- Deafness causing neuroplastin missense variants fail to promote plasma membrane CaThe Journal of biological chemistry · 2024Article
- Presynaptic Nrxn3 is essential for ribbon-synapse assembly in hair cells.bioRxiv : the preprint server for biology · 2024Article
- Age-related alterations in efferent medial olivocochlear-outer hair cell and primary auditory ribbon synapses in CBA/J mice.Frontiers in cellular neuroscience · 2024Article
- Neuroplastin Expression in Male Mice Is Essential for Fertility, Mating, and Adult Testosterone Levels.International journal of molecular sciences · 2023Article
- Absence of Embigin accelerates hearing loss and causes sub-viability, brain and heart defects in C57BL/6N mice due to interaction withiScience · 2023Article
- A dominant variant in apoptosis-related gene XKR8 is relevant to hereditary auditory neuropathy.Journal of translational medicine · 2023Article
- Neuroplastin genetically interacts with Cadherin 23 and the encoded isoform Np55 is sufficient for cochlear hair cell function and hearing.PLoS genetics · 2022Article
- Neuroplastin in Neuropsychiatric Diseases.Genes · 2021Review
- Neuroplastin expression is essential for hearing and hair cell PMCA expression.Brain structure & function · 2021Article
- The Immunoglobulin Superfamily Member Basigin Is Required for Complex Dendrite Formation inFrontiers in cellular neuroscience · 2021Article
- Retrograde Amnesia - A Question of Disturbed Calcium Levels?Frontiers in cellular neuroscience · 2021Review
- The Interaction of TRAF6 With Neuroplastin Promotes Spinogenesis During Early Neuronal Development.Frontiers in cell and developmental biology · 2020Article
- Current concepts in cochlear ribbon synapse formation.Synapse (New York, N.Y.) · 2019Review
- Microarray Analysis of Gene Expression Changes in Neuroplastin 65-Knockout Mice: Implications for Abnormal Cognition and Emotional Disorders.Neuroscience bulletin · 2018Article
- CRISPR/Cas9-mediated knockout of Lim-domain only four retards organ of Corti cell growth.Journal of cellular biochemistry · 2018Article
- A large scale hearing loss screen reveals an extensive unexplored genetic landscape for auditory dysfunction.Nature communications · 2017Article
- A complex of Neuroplastin and Plasma Membrane CaScientific reports · 2017Article
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Authors and funding
17 authors at 3 institutions in 1 country.
Funding
Abstract
The Neuroplastin gene encodes two synapse-enriched protein isoforms, Np55 and Np65, which are transmembrane glycoproteins that regulate several cellular processes, including the genesis, maintenance, and plasticity of synapses. We found that an absence of Np65 causes early-onset sensorineural hearing loss and prevented the normal synaptogenesis in inner hair cells (IHCs) in the newly identified mouse mutant pitch. In wild-type mice, Np65 is strongly upregulated in the cochlea from around postnatal day 12 (P12), which corresponds to the onset of hearing. Np65 was specifically localized at the presynaptic region of IHCs. We found that the colocalization of presynaptic IHC ribbons and postsynaptic afferent terminals is greatly reduced in pitch mutants. Moreover, IHC exocytosis is also reduced with mutant mice showing lower rates of vesicle release. Np65 appears to have a nonessential role in vision. We propose that Np65, by regulating IHC synaptogenesis, is critical for auditory function in mammals. SIGNIFICANCE STATEMENT: In the mammalian cochlea, the sensory inner hair cells (IHCs) encode auditory information. They do this by converting sound wave-induced mechanical motion of their hair bundles into an electrical current. This current generates a receptor potential that controls release of glutamate neurotransmitter from their ribbon synapses onto the auditory afferent fiber. We show that the synapse-enriched protein Np65, encoded by the Neuroplastin gene, is localized at the IHC presynaptic region. In mutant mice, absence of Np65 causes early-onset sensorineural hearing loss and prevents normal neurotransmitter release in IHCs and colocalization of presynaptic ribbons with postsynaptic afferents. We identified Neuroplastin as a novel deafness gene required for ribbon synapse formation and function, which is critical for sound perception in mammals.
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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.