نتایج جستجو برای: dyt1 dystonia

تعداد نتایج: 6648  

2012
Lauren M. Tanabe Caitlin Martin William T. Dauer

DYT1 dystonia is a debilitating neurological disease characterized by involuntary twisting movements. The disease is caused by an in-frame deletion (GAG, "ΔE") mutation in the TOR1A gene that encodes the torsinA protein. Intriguingly, only 30% of mutation carriers exhibit motor symptoms despite the fact that functional brain imaging studies show abnormal brain metabolism in all carriers. Becaus...

Journal: :The EMBO journal 2011
Alessandra Granata Seong Joo Koo Volker Haucke Giampietro Schiavo Thomas T Warner

DYT1 dystonia is caused by an autosomal dominant mutation that leads to a glutamic acid deletion in torsinA (TA), a member of the AAA+ ATPase superfamily. In this study, we identified a novel-binding partner of TA, the subunit 4 (CSN4) of CSN signalosome. TA binds CSN4 and the synaptic regulator snapin in neuroblastoma cells and in brain synaptosomes. CSN4 and TA are required for the stability ...

Journal: :Journal of neurology, neurosurgery, and psychiatry 2009
K Ritz M C F Gerrits E M J Foncke F van Ruissen C van der Linden M D I Vergouwen B R Bloem W Vandenberghe R Crols J D Speelman F Baas M A J Tijssen

BACKGROUND Myoclonus-dystonia (M-D) is an autosomal dominant inherited movement disorder. Various mutations within the epsilon-sarcoglycan (SGCE) gene have been associated with M-D, but mutations are detected in only about 30% of patients. The lack of stringent clinical inclusion criteria and limitations of mutation screens by direct sequencing might explain this observation. METHODS Eighty-s...

Journal: :Journal of cell science 2008
Flávia C Nery Juan Zeng Brian P Niland Jeffrey Hewett Jonathan Farley Daniel Irimia Yuqing Li Gerhard Wiche Arnoud Sonnenberg Xandra O Breakefield

A specific mutation (DeltaE) in torsinA underlies most cases of the dominantly inherited movement disorder, early-onset torsion dystonia (DYT1). TorsinA, a member of the AAA+ ATPase superfamily, is located within the lumen of the nuclear envelope (NE) and endoplasmic reticulum (ER). We investigated an association between torsinA and nesprin-3, which spans the outer nuclear membrane (ONM) of the...

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