Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome

Abstract A high prevalence of obstructive sleep apnea (OSA) has been reported in Down syndrome (DS) owing to the coexistence of multiple predisposing factors related to its genetic abnormality, posing a challenge for the management of OSA. We hypothesized that DS mice recapitulate craniofacial abnor...

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Autores principales:	Tatsunori Takahashi, Noriaki Sakai, Tomonori Iwasaki, Timothy C. Doyle, William C. Mobley, Seiji Nishino
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2020
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/30dc0ed05f7f43e0baf31e10c88765f7
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spelling	oai:doaj.org-article:30dc0ed05f7f43e0baf31e10c88765f72021-12-02T12:33:45ZDetailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome10.1038/s41598-020-78278-22045-2322https://doaj.org/article/30dc0ed05f7f43e0baf31e10c88765f72020-12-01T00:00:00Zhttps://doi.org/10.1038/s41598-020-78278-2https://doaj.org/toc/2045-2322Abstract A high prevalence of obstructive sleep apnea (OSA) has been reported in Down syndrome (DS) owing to the coexistence of multiple predisposing factors related to its genetic abnormality, posing a challenge for the management of OSA. We hypothesized that DS mice recapitulate craniofacial abnormalities and upper airway obstruction of human DS and can serve as an experimental platform for OSA research. This study, thus, aimed to quantitatively characterize the upper airway as well as craniofacial abnormalities in Dp(16)1Yey (Dp16) mice. Dp16 mice demonstrated craniofacial hypoplasia, especially in the ventral part of the skull and the mandible, and rostrally positioned hyoid. These changes were accompanied with a shorter length and smaller cross-sectional area of the upper airway, resulting in a significantly reduced upper airway volume in Dp16 mice. Our non-invasive approach, a combination of computational fluid dynamics and high-resolution micro-CT imaging, revealed a higher negative pressure inside the airway of Dp16 mice compared to wild-type littermates, showing the potential risk of upper airway collapse. Our study indicated that Dp16 mice can be a useful model to examine the pathophysiology of increased upper airway collapsibility of DS and to evaluate the efficacy of therapeutic interventions for breathing and sleep anomalies.Tatsunori TakahashiNoriaki SakaiTomonori IwasakiTimothy C. DoyleWilliam C. MobleySeiji NishinoNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 10, Iss 1, Pp 1-11 (2020)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Tatsunori Takahashi Noriaki Sakai Tomonori Iwasaki Timothy C. Doyle William C. Mobley Seiji Nishino Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome
description	Abstract A high prevalence of obstructive sleep apnea (OSA) has been reported in Down syndrome (DS) owing to the coexistence of multiple predisposing factors related to its genetic abnormality, posing a challenge for the management of OSA. We hypothesized that DS mice recapitulate craniofacial abnormalities and upper airway obstruction of human DS and can serve as an experimental platform for OSA research. This study, thus, aimed to quantitatively characterize the upper airway as well as craniofacial abnormalities in Dp(16)1Yey (Dp16) mice. Dp16 mice demonstrated craniofacial hypoplasia, especially in the ventral part of the skull and the mandible, and rostrally positioned hyoid. These changes were accompanied with a shorter length and smaller cross-sectional area of the upper airway, resulting in a significantly reduced upper airway volume in Dp16 mice. Our non-invasive approach, a combination of computational fluid dynamics and high-resolution micro-CT imaging, revealed a higher negative pressure inside the airway of Dp16 mice compared to wild-type littermates, showing the potential risk of upper airway collapse. Our study indicated that Dp16 mice can be a useful model to examine the pathophysiology of increased upper airway collapsibility of DS and to evaluate the efficacy of therapeutic interventions for breathing and sleep anomalies.
format	article
author	Tatsunori Takahashi Noriaki Sakai Tomonori Iwasaki Timothy C. Doyle William C. Mobley Seiji Nishino
author_facet	Tatsunori Takahashi Noriaki Sakai Tomonori Iwasaki Timothy C. Doyle William C. Mobley Seiji Nishino
author_sort	Tatsunori Takahashi
title	Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome
title_short	Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome
title_full	Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome
title_fullStr	Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome
title_full_unstemmed	Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome
title_sort	detailed evaluation of the upper airway in the dp(16)1yey mouse model of down syndrome
publisher	Nature Portfolio
publishDate	2020
url	https://doaj.org/article/30dc0ed05f7f43e0baf31e10c88765f7
work_keys_str_mv	AT tatsunoritakahashi detailedevaluationoftheupperairwayinthedp161yeymousemodelofdownsyndrome AT noriakisakai detailedevaluationoftheupperairwayinthedp161yeymousemodelofdownsyndrome AT tomonoriiwasaki detailedevaluationoftheupperairwayinthedp161yeymousemodelofdownsyndrome AT timothycdoyle detailedevaluationoftheupperairwayinthedp161yeymousemodelofdownsyndrome AT williamcmobley detailedevaluationoftheupperairwayinthedp161yeymousemodelofdownsyndrome AT seijinishino detailedevaluationoftheupperairwayinthedp161yeymousemodelofdownsyndrome
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Detailed evaluation of the upper airway in the Dp(16)1Yey mouse model of Down syndrome

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