A patient presenting a 22q13 deletion associated with an apparently balanced translocation t(16;22) : an ilustrative case in the investigation of patients with low ARSA activity

A 10-year-old speechless, mentally deficient male, with low arylsulfatase A (ARSA) activity, and presumably, methachromatic leukodystrophy, underwent genetic evaluation. As the clinical picture was not compatible with this diagnosisan ARSA gene and chromosome analysis were performed, showing the pre...

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Detalles Bibliográficos
Autores: Artigalas, Osvaldo Alfonso Pinto, Paskulin, Giorgio Adriano, Riegel, Mariluce, Burin, Maira Graeff, Pereira, Maria Luiza Saraiva, Maluf, Sharbel Weidner, Kiss, Andréia, Schwartz, Ida Vanessa Doederlein
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2012
País:Brasil
Institución:Universidade Federal do Rio Grande do Sul (UFRGS)
Repositorio:Repositório Institucional da UFRGS
Idioma:inglés
OAI Identifier:oai:www.lume.ufrgs.br:10183/87730
Acceso en línea:http://hdl.handle.net/10183/87730
Access Level:acceso abierto
Palabra clave:Leucodistrofia metacromática
22q13 deletion
apparently balanced translocation
ARSA gene
arylsulfatase A pseudodeficiency
metachromatic leukodystrophy
Descripción
Sumario:A 10-year-old speechless, mentally deficient male, with low arylsulfatase A (ARSA) activity, and presumably, methachromatic leukodystrophy, underwent genetic evaluation. As the clinical picture was not compatible with this diagnosisan ARSA gene and chromosome analysis were performed, showing the presence of a pseudodeficiency ARSA allele and a de novo apparently balanced t(16;22)(p11.2;q13) translocation. A deletion on the long arm of chromosome 22 encompassing the ARSA gene, as shown by FISH and array-CGH, indicated a 22q13 deletion syndrome. This case illustrates the importance of detailed cytogenetic investigation in patients presenting low arylsulfatase A activity and atypical/unspecific clinical features.