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CHD2-Related CNS Pathologies.
Wilson, Marc-Michel; Henshall, David C; Byrne, Susan M; Brennan, Gary P.
Afiliación
  • Wilson MM; Department of Physiology and Medical Physics, RCSI, University of Medicine and Health Sciences, Dublin 02, Ireland.
  • Henshall DC; FutureNeuro SFI Research Centre, RCSI, University of Medicine and Health Sciences, Dublin D02 YN77, Ireland.
  • Byrne SM; Department of Physiology and Medical Physics, RCSI, University of Medicine and Health Sciences, Dublin 02, Ireland.
  • Brennan GP; FutureNeuro SFI Research Centre, RCSI, University of Medicine and Health Sciences, Dublin D02 YN77, Ireland.
Int J Mol Sci ; 22(2)2021 Jan 08.
Article en En | MEDLINE | ID: mdl-33435571
Epileptic encephalopathies (EE) are severe epilepsy syndromes characterized by multiple seizure types, developmental delay and even regression. This class of disorders are increasingly being identified as resulting from de novo genetic mutations including many identified mutations in the family of chromodomain helicase DNA binding (CHD) proteins. In particular, several de novo pathogenic mutations have been identified in the gene encoding chromodomain helicase DNA binding protein 2 (CHD2), a member of the sucrose nonfermenting (SNF-2) protein family of epigenetic regulators. These mutations in the CHD2 gene are causative of early onset epileptic encephalopathy, abnormal brain function, and intellectual disability. Our understanding of the mechanisms by which modification or loss of CHD2 cause this condition remains poorly understood. Here, we review what is known and still to be elucidated as regards the structure and function of CHD2 and how its dysregulation leads to a highly variable range of phenotypic presentations.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Epilepsia Generalizada / Predisposición Genética a la Enfermedad / Proteínas de Unión al ADN / Discapacidad Intelectual / Mutación Tipo de estudio: Prognostic_studies Límite: Animals / Humans Idioma: En Revista: Int J Mol Sci Año: 2021 Tipo del documento: Article País de afiliación: Irlanda Pais de publicación: Suiza

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Epilepsia Generalizada / Predisposición Genética a la Enfermedad / Proteínas de Unión al ADN / Discapacidad Intelectual / Mutación Tipo de estudio: Prognostic_studies Límite: Animals / Humans Idioma: En Revista: Int J Mol Sci Año: 2021 Tipo del documento: Article País de afiliación: Irlanda Pais de publicación: Suiza