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Genetic analysis of polymorphisms in the kalirin gene for association with age-at-onset in European Huntington disease patients.
Tsai, Yu-Chun; Metzger, Silke; Riess, Olaf; Soehn, Anne S; Nguyen, Huu Phuc.
Afiliación
  • Tsai YC; Department of Medical Genetics, University of Tuebingen, Calwerstr 7, Tuebingen, 72076, Germany.
BMC Med Genet ; 13: 48, 2012 Jun 21.
Article en En | MEDLINE | ID: mdl-22720673
BACKGROUND: Huntington disease (HD) is caused by an expanded CAG repeat in the HD gene. Although the length of the CAG repeat strongly correlates with the age-at-onset (AAO), AAO in HD individuals may differ dramatically in spite of similar expanded CAG repeat lengths. Additional genetic or environmental factors are thought to influence the disease onset. Several modifier genes have been discovered so far but they do not fully explain the variability of AAO in HD. To potentially identify a novel genetic modifier, we analyzed single nucleotide polymorphisms (SNPs) in the kalirin (KALRN) gene. Kalirin is a protein crucially involved in spine plasticity and its interaction with huntingtin-associated protein-1 (HAP-1) and a potential protein dysfunction might contribute to spine pathogenesis in HD. METHODS: The selected SNPs were genotyped by polymerase chain reaction-restriction fragment length polymorphism (PCR-RFLP) and association of SNPs with AAO was investigated with the framework of linear models in an analysis of variance and covariance. RESULTS: Eleven SNPs in the kalirin gene were examined in an association study in European HD patients. The ten coding SNPs under investigation were monomorphic, whereas SNP rs10934657 in the promoter region showed a minor allele frequency >1%. An analysis of covariance together with the influence of the expanded HD allele was applied in 680 HD patients. SNP rs10934657 did not affect the AAO of the examined HD population. CONCLUSIONS: The results did not reveal an association between the analyzed kalirin polymorphisms and the AAO in HD. However, it does not exclude other SNPs of the kalirin gene as susceptible genetic modifiers.
Asunto(s)

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Proteínas Serina-Treonina Quinasas / Enfermedad de Huntington / Factores de Intercambio de Guanina Nucleótido / Polimorfismo de Nucleótido Simple / Población Blanca / Genes Modificadores Tipo de estudio: Prognostic_studies / Risk_factors_studies Límite: Adolescent / Adult / Aged / Aged80 / Child / Child, preschool / Female / Humans / Male / Middle aged Idioma: En Revista: BMC Med Genet Asunto de la revista: GENETICA MEDICA Año: 2012 Tipo del documento: Article País de afiliación: Alemania Pais de publicación: Reino Unido

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Proteínas Serina-Treonina Quinasas / Enfermedad de Huntington / Factores de Intercambio de Guanina Nucleótido / Polimorfismo de Nucleótido Simple / Población Blanca / Genes Modificadores Tipo de estudio: Prognostic_studies / Risk_factors_studies Límite: Adolescent / Adult / Aged / Aged80 / Child / Child, preschool / Female / Humans / Male / Middle aged Idioma: En Revista: BMC Med Genet Asunto de la revista: GENETICA MEDICA Año: 2012 Tipo del documento: Article País de afiliación: Alemania Pais de publicación: Reino Unido