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Viral-mediated Ntf3 overexpression disrupts innervation and hearing in nondeafened guinea pig cochleae

Synaptopathy in the cochlea occurs when the connection between inner hair cells and the auditory nerve is disrupted, leading to impaired hearing and nerve degeneration. Experiments using transgenic mice have shown that overexpression of NT3 by supporting cells repairs synaptopathy caused by overstim...

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Autores principales: Lee, Min Young, Kurioka, Takaomi, Nelson, Megan M, Prieskorn, Diane M, Swiderski, Donald L, Takada, Yohei, Beyer, Lisa A, Raphael, Yehoash
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4972090/
https://www.ncbi.nlm.nih.gov/pubmed/27525291
http://dx.doi.org/10.1038/mtm.2016.52
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author Lee, Min Young
Kurioka, Takaomi
Nelson, Megan M
Prieskorn, Diane M
Swiderski, Donald L
Takada, Yohei
Beyer, Lisa A
Raphael, Yehoash
author_facet Lee, Min Young
Kurioka, Takaomi
Nelson, Megan M
Prieskorn, Diane M
Swiderski, Donald L
Takada, Yohei
Beyer, Lisa A
Raphael, Yehoash
author_sort Lee, Min Young
collection PubMed
description Synaptopathy in the cochlea occurs when the connection between inner hair cells and the auditory nerve is disrupted, leading to impaired hearing and nerve degeneration. Experiments using transgenic mice have shown that overexpression of NT3 by supporting cells repairs synaptopathy caused by overstimulation. To accomplish such therapy in the clinical setting, it would be necessary to activate the neurotrophin receptor on auditory neurons by other means. Here we test the outcome of NT3 overexpression using viral-mediated gene transfer into the perilymph versus the endolymph of the normal guinea pig cochlea. We inoculated two different Ntf3 viral vectors, adenovirus (Adv) or adeno-associated virus (AAV) into the perilymph, to facilitate transgene expression in the mesothelial cells and cochlear duct epithelium, respectively. We assessed outcomes by comparing Auditory brainstem response (ABR) thresholds prior to that at baseline to thresholds at 1 and 3 weeks after inoculation, and then performed histologic evaluation of hair cells, nerve endings, and synaptic ribbons. We observed hearing threshold shifts as well as disorganization of peripheral nerve endings and disruption of synaptic connections between inner hair cells and peripheral nerve endings with both vectors. The data suggest that elevation of NT3 levels in the cochlear fluids can disrupt innervation and degrade hearing.
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spelling pubmed-49720902016-08-12 Viral-mediated Ntf3 overexpression disrupts innervation and hearing in nondeafened guinea pig cochleae Lee, Min Young Kurioka, Takaomi Nelson, Megan M Prieskorn, Diane M Swiderski, Donald L Takada, Yohei Beyer, Lisa A Raphael, Yehoash Mol Ther Methods Clin Dev Article Synaptopathy in the cochlea occurs when the connection between inner hair cells and the auditory nerve is disrupted, leading to impaired hearing and nerve degeneration. Experiments using transgenic mice have shown that overexpression of NT3 by supporting cells repairs synaptopathy caused by overstimulation. To accomplish such therapy in the clinical setting, it would be necessary to activate the neurotrophin receptor on auditory neurons by other means. Here we test the outcome of NT3 overexpression using viral-mediated gene transfer into the perilymph versus the endolymph of the normal guinea pig cochlea. We inoculated two different Ntf3 viral vectors, adenovirus (Adv) or adeno-associated virus (AAV) into the perilymph, to facilitate transgene expression in the mesothelial cells and cochlear duct epithelium, respectively. We assessed outcomes by comparing Auditory brainstem response (ABR) thresholds prior to that at baseline to thresholds at 1 and 3 weeks after inoculation, and then performed histologic evaluation of hair cells, nerve endings, and synaptic ribbons. We observed hearing threshold shifts as well as disorganization of peripheral nerve endings and disruption of synaptic connections between inner hair cells and peripheral nerve endings with both vectors. The data suggest that elevation of NT3 levels in the cochlear fluids can disrupt innervation and degrade hearing. Nature Publishing Group 2016-08-03 /pmc/articles/PMC4972090/ /pubmed/27525291 http://dx.doi.org/10.1038/mtm.2016.52 Text en Copyright © 2016 Official journal of the American Society of Gene & Cell Therapy http://creativecommons.org/licenses/by-nc-sa/4.0/ This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in the credit line; if the material is not included under the Creative Commons license, users will need to obtain permission from the license holder to reproduce the material. To view a copy of this license, visit http://creativecommons.org/licenses/by-nc-sa/4.0/
spellingShingle Article
Lee, Min Young
Kurioka, Takaomi
Nelson, Megan M
Prieskorn, Diane M
Swiderski, Donald L
Takada, Yohei
Beyer, Lisa A
Raphael, Yehoash
Viral-mediated Ntf3 overexpression disrupts innervation and hearing in nondeafened guinea pig cochleae
title Viral-mediated Ntf3 overexpression disrupts innervation and hearing in nondeafened guinea pig cochleae
title_full Viral-mediated Ntf3 overexpression disrupts innervation and hearing in nondeafened guinea pig cochleae
title_fullStr Viral-mediated Ntf3 overexpression disrupts innervation and hearing in nondeafened guinea pig cochleae
title_full_unstemmed Viral-mediated Ntf3 overexpression disrupts innervation and hearing in nondeafened guinea pig cochleae
title_short Viral-mediated Ntf3 overexpression disrupts innervation and hearing in nondeafened guinea pig cochleae
title_sort viral-mediated ntf3 overexpression disrupts innervation and hearing in nondeafened guinea pig cochleae
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4972090/
https://www.ncbi.nlm.nih.gov/pubmed/27525291
http://dx.doi.org/10.1038/mtm.2016.52
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