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The Adult Ts65Dn Mouse Model of Down Syndrome Shows Altered Swallow Function

There are increased risks for deglutition disorders in people with Down syndrome (DS). Although mouse models have been used to study the biological underpinnings of DS in other areas, relatively little is known about swallowing phenotypes in these models. We hypothesized that swallowing performance...

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Autores principales: Glass, Tiffany J., Valmadrid, Luke Carmichael V., Connor, Nadine P.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6727863/
https://www.ncbi.nlm.nih.gov/pubmed/31555077
http://dx.doi.org/10.3389/fnins.2019.00906
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author Glass, Tiffany J.
Valmadrid, Luke Carmichael V.
Connor, Nadine P.
author_facet Glass, Tiffany J.
Valmadrid, Luke Carmichael V.
Connor, Nadine P.
author_sort Glass, Tiffany J.
collection PubMed
description There are increased risks for deglutition disorders in people with Down syndrome (DS). Although mouse models have been used to study the biological underpinnings of DS in other areas, relatively little is known about swallowing phenotypes in these models. We hypothesized that swallowing performance would be affected in adult mouse models of DS, relative to typical control mice. Videofluoroscopic swallow studies (VFSS) were conducted on adults of two mouse models of DS: Ts65Dn and Dp(16)1Yey, and evaluated in comparison with age-matched controls. Relative to other groups, adult Ts65Dn showed significantly slower swallow rates, longer inter-swallow intervals (ISI), and greater numbers of jaw excursion cycles preceding each swallow. In contrast, adult Dp(16)1Yey mice showed swallowing performance similar to control mice. Exploratory quantitative analyses of the intrinsic tongue (transverse muscle), and extrinsic tongue muscles [genioglossus (GG), styloglossus (SG), and hyoglossus (HG)] showed no significant differences between genotype groups in myosin heavy chain isoform profiles. Collectively, these findings suggest that while swallowing is typical in adult Dp(16)1Yey, swallowing in adult Ts65Dn is atypical due to unknown causes. The finding that adult Ts65Dn may have utility as a model of dysphagia provides new opportunities to elucidate biological underpinnings of dysphagia associated with DS.
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spelling pubmed-67278632019-09-25 The Adult Ts65Dn Mouse Model of Down Syndrome Shows Altered Swallow Function Glass, Tiffany J. Valmadrid, Luke Carmichael V. Connor, Nadine P. Front Neurosci Neuroscience There are increased risks for deglutition disorders in people with Down syndrome (DS). Although mouse models have been used to study the biological underpinnings of DS in other areas, relatively little is known about swallowing phenotypes in these models. We hypothesized that swallowing performance would be affected in adult mouse models of DS, relative to typical control mice. Videofluoroscopic swallow studies (VFSS) were conducted on adults of two mouse models of DS: Ts65Dn and Dp(16)1Yey, and evaluated in comparison with age-matched controls. Relative to other groups, adult Ts65Dn showed significantly slower swallow rates, longer inter-swallow intervals (ISI), and greater numbers of jaw excursion cycles preceding each swallow. In contrast, adult Dp(16)1Yey mice showed swallowing performance similar to control mice. Exploratory quantitative analyses of the intrinsic tongue (transverse muscle), and extrinsic tongue muscles [genioglossus (GG), styloglossus (SG), and hyoglossus (HG)] showed no significant differences between genotype groups in myosin heavy chain isoform profiles. Collectively, these findings suggest that while swallowing is typical in adult Dp(16)1Yey, swallowing in adult Ts65Dn is atypical due to unknown causes. The finding that adult Ts65Dn may have utility as a model of dysphagia provides new opportunities to elucidate biological underpinnings of dysphagia associated with DS. Frontiers Media S.A. 2019-08-29 /pmc/articles/PMC6727863/ /pubmed/31555077 http://dx.doi.org/10.3389/fnins.2019.00906 Text en Copyright © 2019 Glass, Valmadrid and Connor. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Neuroscience
Glass, Tiffany J.
Valmadrid, Luke Carmichael V.
Connor, Nadine P.
The Adult Ts65Dn Mouse Model of Down Syndrome Shows Altered Swallow Function
title The Adult Ts65Dn Mouse Model of Down Syndrome Shows Altered Swallow Function
title_full The Adult Ts65Dn Mouse Model of Down Syndrome Shows Altered Swallow Function
title_fullStr The Adult Ts65Dn Mouse Model of Down Syndrome Shows Altered Swallow Function
title_full_unstemmed The Adult Ts65Dn Mouse Model of Down Syndrome Shows Altered Swallow Function
title_short The Adult Ts65Dn Mouse Model of Down Syndrome Shows Altered Swallow Function
title_sort adult ts65dn mouse model of down syndrome shows altered swallow function
topic Neuroscience
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6727863/
https://www.ncbi.nlm.nih.gov/pubmed/31555077
http://dx.doi.org/10.3389/fnins.2019.00906
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