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Ann Neurol ; 68(4): 549-53, 2010 Oct.
Article in English | MEDLINE | ID: mdl-20865765

ABSTRACT

Primary dystonia is a movement disorder characterized by sustained muscle contractions and in which dystonia is the only or predominant clinical feature. TOR1A(DYT1) and the transcription factor THAP1(DYT6) are the only genes identified thus far for primary dystonia. Using electromobility shift assays and chromatin immunoprecipitation (ChIP) quantitative polymerase chain reaction (qPCR), we demonstrate a physical interaction between THAP1 and the TOR1A promoter that is abolished by pathophysiologic mutations. Our findings provide the first evidence that causative genes for primary dystonia intersect in a common pathway and raise the possibility of developing novel therapies targeting this pathway.


Subject(s)
Apoptosis Regulatory Proteins/genetics , Apoptosis Regulatory Proteins/metabolism , DNA-Binding Proteins/genetics , DNA-Binding Proteins/metabolism , Dystonic Disorders/genetics , Gene Expression Regulation/genetics , Molecular Chaperones/genetics , Molecular Chaperones/metabolism , Nuclear Proteins/genetics , Nuclear Proteins/metabolism , Adult , Aged , Animals , Brain/metabolism , Cell Nucleus/metabolism , Cells, Cultured , Chromatin Immunoprecipitation/methods , Dystonic Disorders/pathology , Electrophoretic Mobility Shift Assay/methods , Endothelial Cells/drug effects , Endothelial Cells/ultrastructure , Female , Fibroblasts/metabolism , Fibroblasts/ultrastructure , Gene Expression Regulation/drug effects , Glioblastoma/pathology , Humans , Male , Mice , Middle Aged , Mutation/genetics , Promoter Regions, Genetic/physiology , Protein Binding , RNA, Messenger/metabolism , RNA, Small Interfering/metabolism , RNA, Small Interfering/pharmacology , Transfection/methods , Umbilical Veins
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