Dopa-responsive dystonia (DRD): systematic search in Latin America

Dopa-responsive dystonia (DRD) encompasses a heterogenous group of primary dystonias, caused by enzymatic deficiencies across the amines pathway and, by definition, show as their main characteristic a favorable and sustained response to levodopa. There are up to 6 genes associated with DRD, includin...

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Detalles Bibliográficos
Autores: Zelada-Ríos, Laura, Sarapura-Castro, Elison, Solórzano-Palacios, Karol, La Serna-Infantes, Jorge, Aguirre-Quispe, Wilfor, Cosentino-Esquerre, Carlos, Urbina-Ramírez, Luis, Torres-Ramírez, Luis, Mazzetti, Pilar, Cornejo-Olivas, Mario
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2022
País:Perú
Institución:Universidad Peruana Cayetano Heredia
Repositorio:Revistas - Universidad Peruana Cayetano Heredia
Idioma:español
OAI Identifier:oai:revistas.upch.edu.pe:article/4154
Acceso en línea:https://revistas.upch.edu.pe/index.php/RNP/article/view/4154
Access Level:acceso abierto
Palabra clave:Dopa-responsive dystonia
Segawa disease
GTP-cyclohydrolase 1 deficiency
tyrosine hydroxylase deficiency
tetrahydrobiopterin
Distonías respondedoras a levodopa
Enfermedad de Segawa
deficiencia de GTP-ciclohidrolasa 1
deficiencia de tirosina hidroxilasa
tetrahidrobiopterina
Descripción
Sumario:Dopa-responsive dystonia (DRD) encompasses a heterogenous group of primary dystonias, caused by enzymatic deficiencies across the amines pathway and, by definition, show as their main characteristic a favorable and sustained response to levodopa. There are up to 6 genes associated with DRD, including pathogenic variants of the GCH1 gene as the most frequently involved. The typical presentation of DRD is characterized by start in childhood, lower limb-onset dystonia with daytime fluctuation, mild parkinsonism, and a sustained response to low doses of levodopa. A systematic literature search on DRD reported cases in Latin America is presented.