Sensory transduction is required for normal development and maturation of cochlear inner hair cell synapses

Acoustic overexposure and aging can damage auditory synapses in the inner ear by a process known as synaptopathy. These insults may also damage hair bundles and the sensory transduction apparatus in auditory hair cells. However, a connection between sensory transduction and synaptopathy has not been...

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Autores principales:	John Lee, Kosuke Kawai, Jeffrey R Holt, Gwenaëlle SG Géléoc
Formato:	article
Lenguaje:	EN
Publicado:	eLife Sciences Publications Ltd 2021
Materias:	hair cell sensory transduction synaptopathy synaptogenesis spiral ganglion neuron TMC1 Medicine R Science Q Biology (General) QH301-705.5
Acceso en línea:	https://doaj.org/article/ae6d9e09635b4bedb9676c00615f3f00
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id	oai:doaj.org-article:ae6d9e09635b4bedb9676c00615f3f00
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spelling	oai:doaj.org-article:ae6d9e09635b4bedb9676c00615f3f002021-11-17T13:23:28ZSensory transduction is required for normal development and maturation of cochlear inner hair cell synapses10.7554/eLife.694332050-084Xe69433https://doaj.org/article/ae6d9e09635b4bedb9676c00615f3f002021-11-01T00:00:00Zhttps://elifesciences.org/articles/69433https://doaj.org/toc/2050-084XAcoustic overexposure and aging can damage auditory synapses in the inner ear by a process known as synaptopathy. These insults may also damage hair bundles and the sensory transduction apparatus in auditory hair cells. However, a connection between sensory transduction and synaptopathy has not been established. To evaluate potential contributions of sensory transduction to synapse formation and development, we assessed inner hair cell synapses in several genetic models of dysfunctional sensory transduction, including mice lacking transmembrane channel-like (Tmc) 1, Tmc2, or both, in Beethoven mice which carry a dominant Tmc1 mutation and in Spinner mice which carry a recessive mutation in transmembrane inner ear (Tmie). Our analyses reveal loss of synapses in the absence of sensory transduction and preservation of synapses in Tmc1-null mice following restoration of sensory transduction via Tmc1 gene therapy. These results provide insight into the requirement of sensory transduction for hair cell synapse development and maturation.John LeeKosuke KawaiJeffrey R HoltGwenaëlle SG GéléoceLife Sciences Publications Ltdarticlehair cellsensory transductionsynaptopathysynaptogenesisspiral ganglion neuronTMC1MedicineRScienceQBiology (General)QH301-705.5ENeLife, Vol 10 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	hair cell sensory transduction synaptopathy synaptogenesis spiral ganglion neuron TMC1 Medicine R Science Q Biology (General) QH301-705.5
spellingShingle	hair cell sensory transduction synaptopathy synaptogenesis spiral ganglion neuron TMC1 Medicine R Science Q Biology (General) QH301-705.5 John Lee Kosuke Kawai Jeffrey R Holt Gwenaëlle SG Géléoc Sensory transduction is required for normal development and maturation of cochlear inner hair cell synapses
description	Acoustic overexposure and aging can damage auditory synapses in the inner ear by a process known as synaptopathy. These insults may also damage hair bundles and the sensory transduction apparatus in auditory hair cells. However, a connection between sensory transduction and synaptopathy has not been established. To evaluate potential contributions of sensory transduction to synapse formation and development, we assessed inner hair cell synapses in several genetic models of dysfunctional sensory transduction, including mice lacking transmembrane channel-like (Tmc) 1, Tmc2, or both, in Beethoven mice which carry a dominant Tmc1 mutation and in Spinner mice which carry a recessive mutation in transmembrane inner ear (Tmie). Our analyses reveal loss of synapses in the absence of sensory transduction and preservation of synapses in Tmc1-null mice following restoration of sensory transduction via Tmc1 gene therapy. These results provide insight into the requirement of sensory transduction for hair cell synapse development and maturation.
format	article
author	John Lee Kosuke Kawai Jeffrey R Holt Gwenaëlle SG Géléoc
author_facet	John Lee Kosuke Kawai Jeffrey R Holt Gwenaëlle SG Géléoc
author_sort	John Lee
title	Sensory transduction is required for normal development and maturation of cochlear inner hair cell synapses
title_short	Sensory transduction is required for normal development and maturation of cochlear inner hair cell synapses
title_full	Sensory transduction is required for normal development and maturation of cochlear inner hair cell synapses
title_fullStr	Sensory transduction is required for normal development and maturation of cochlear inner hair cell synapses
title_full_unstemmed	Sensory transduction is required for normal development and maturation of cochlear inner hair cell synapses
title_sort	sensory transduction is required for normal development and maturation of cochlear inner hair cell synapses
publisher	eLife Sciences Publications Ltd
publishDate	2021
url	https://doaj.org/article/ae6d9e09635b4bedb9676c00615f3f00
work_keys_str_mv	AT johnlee sensorytransductionisrequiredfornormaldevelopmentandmaturationofcochlearinnerhaircellsynapses AT kosukekawai sensorytransductionisrequiredfornormaldevelopmentandmaturationofcochlearinnerhaircellsynapses AT jeffreyrholt sensorytransductionisrequiredfornormaldevelopmentandmaturationofcochlearinnerhaircellsynapses AT gwenaellesggeleoc sensorytransductionisrequiredfornormaldevelopmentandmaturationofcochlearinnerhaircellsynapses
_version_	1718425565542744064

Sensory transduction is required for normal development and maturation of cochlear inner hair cell synapses

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