نتایج جستجو برای: dyt6 dystonia

تعداد نتایج: 6616  

2012
Georgia Xiromerisiou Henry Houlden Nikolaos Scarmeas Maria Stamelou Eleanna Kara John Hardy Andrew J Lees Prasad Korlipara Patricia Limousin Reema Paudel Georgios M Hadjigeorgiou Kailash P Bhatia

THAP1 mutations have been shown to be the cause of DYT6. A number of different mutation types and locations in the THAP1 gene have been associated with a range of severity and dystonia phenotypes, but, as yet, it has been difficult to identify clear genotype phenotype patterns. Here, we screened the THAP1 gene in a further series of dystonia cases and evaluated the mutation pathogenicity in thi...

Journal: :Otolaryngology--head and neck surgery : official journal of American Academy of Otolaryngology-Head and Neck Surgery 2016
Claudio M de Gusmão Tania Fuchs Andrew Moses Trisha Multhaupt-Buell Phillip C Song Laurie J Ozelius Ramon A Franco Nutan Sharma

OBJECTIVE Spasmodic dysphonia is a focal dystonia of the larynx with heterogeneous manifestations and association with familial risk factors. There are scarce data to allow precise understanding of etiology and pathophysiology. Screening for dystonia-causing genetic mutations has the potential to allow accurate diagnosis, inform about genotype-phenotype correlations, and allow a better understa...

2014
Maitane Ortiz-Virumbrales Marta Ruiz Eugene Hone Georgia Dolios Rong Wang Andrika Morant Jessica Kottwitz Laurie J Ozelius Sam Gandy Michelle E Ehrlich

Mutations in THAP1 result in dystonia type 6, with partial penetrance and variable phenotype. The goal of this study was to examine the nature and expression pattern of the protein product(s) of the Thap1 transcription factor (DYT6 gene) in mouse neurons, and to study the regional and developmental distribution, and subcellular localization of Thap1 protein. The goal was accomplished via overex...

2012
Sébastien Campagne Isabelle Muller Alain Milon Virginie Gervais

The transcription factor THAP1 (THanatos Associated Protein 1) has emerged recently as the cause of DYT6 primary dystonia, a type of rare, familial and mostly early-onset syndrome that leads to involuntary muscle contractions. Many of the mutations described in the DYT6 patients fall within the sequence-specific DNA-binding domain (THAP domain) of THAP1 and are believed to negatively affect DNA...

Journal: :Brain : a journal of neurology 2009
Ulrich Müller

Presently, 17 distinct monogenic primary dystonias referred to as dystonias 1- 4, 5a,b, 6-8, 10-13 and 15-18 (loci DYT 1-4, 5a,b, 6-8, 10-13, 15-18) have been recognized. Twelve forms are inherited as autosomal dominant, four as autosomal recessive and one as an X-linked recessive trait. Three additional autosomal dominant forms (DYT9, DYT19 and DYT20) might exist based on linkage mapping to re...

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