The pathophysiology of rett syndrome with a focus on breathing dysfunctions

Rett syndrome (RTT), an X-chromosome-linked neurological disorder, is characterized by serious pathophysiology, including breathing and feeding dysfunctions, and alteration of cardiorespiratory coupling, a consequence of multiple interrelated disturbances in the genetic and homeostatic regulation of...

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Detalhes bibliográficos
Autores: Ramirez, Jan-Marino, Karlen-Amarante, Marlusa [UNESP], Wang, Jia-Der Ju, Bush, Nicholas E., Carroll, Michael S., Weese-Mayer, Debra E., Huff, Alyssa
Formato: artículo
Estado:Versión publicada
Fecha de publicación:2020
País:Brasil
Recursos:Universidade Estadual Paulista (UNESP)
Repositorio:Repositório Institucional da UNESP
Idioma:inglés
OAI Identifier:oai:repositorio.unesp.br:11449/206700
Acesso em linha:http://dx.doi.org/10.1152/physiol.00008.2020
http://hdl.handle.net/11449/206700
Access Level:acceso abierto
Palavra-chave:Autonomic dysregulation
Breathing
Dysphagia
Oxidative stress
Descrição
Resumo:Rett syndrome (RTT), an X-chromosome-linked neurological disorder, is characterized by serious pathophysiology, including breathing and feeding dysfunctions, and alteration of cardiorespiratory coupling, a consequence of multiple interrelated disturbances in the genetic and homeostatic regulation of central and peripheral neuronal networks, redox state, and control of inflammation. Characteristic breath-holds, obstructive sleep apnea, and aerophagia result in intermittent hypoxia, which, combined with mitochondrial dysfunction, causes oxidative stress-an important driver of the clinical presentation of RTT.