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Absence of Neuroplastin-65 Affects Synaptogenesis in Mouse Inner Hair Cells and Causes Profound Hearing Loss.

Leanne CarrottMichael R BowlCarlos AguilarStuart L JohnsonLauren ChessumMelissa WestSusan MorseJoanne DorningElizabeth SmartRachel Hardisty-HughesGreg BallAndrew ParkerAlun R BarnardRobert E MacLarenSara WellsWalter MarcottiSteve D M Brown
Published in: The Journal of neuroscience : the official journal of the Society for Neuroscience (2016)
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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