نتایج جستجو برای: dyt1 dystonia

تعداد نتایج: 6648  

Journal: :Human molecular genetics 2010
Pan Chen Alexander J Burdette J Christopher Porter John C Ricketts Stacey A Fox Flavia C Nery Jeffrey W Hewett Laura A Berkowitz Xandra O Breakefield Kim A Caldwell Guy A Caldwell

Early-onset torsion dystonia is the most severe heritable form of dystonia, a human movement disorder that typically starts during a developmental window in early adolescence. Deletion in the DYT1 gene, encoding the torsinA protein, is responsible for this dominantly inherited disorder, which is non-degenerative and exhibits reduced penetrance among carriers. Here, we explore the hypothesis tha...

Journal: :Human molecular genetics 2006
Norman Kock Teresa V Naismith Heather E Boston Laurie J Ozelius David P Corey Xandra O Breakefield Phyllis I Hanson

Four naturally occurring sequence variations have been found in the coding region of the DYT1 gene encoding torsinA. One of these, a 3 bp (DeltaGAG) deletion, underlies dominantly inherited cases of early-onset torsion dystonia. Others, including a single nucleotide polymorphism that replaces aspartic acid (D) at residue 216 with histidine (H) in 12% of normal alleles and two other rare deletio...

2012
Jonathan J. Sager Gonzalo E. Torres Edward A. Burton

DYT1 dystonia is caused by mutation of the TOR1A gene, resulting in the loss of a single glutamic acid residue near the carboxyl terminal of TorsinA. The neuronal functions perturbed by TorsinA[ΔE] are a major unresolved issue in understanding the pathophysiology of dystonia, presenting a critical roadblock to developing effective treatments. We identified and characterized the zebrafish homolo...

2013
Sadahiro Iwabuchi Jin-Young Koh Kai Wang K. W. David Ho N. Charles Harata

DYT1 dystonia is the most common hereditary form of primary torsion dystonia. This autosomal-dominant disorder is characterized by involuntary muscle contractions that cause sustained twisting and repetitive movements. It is caused by an in-frame deletion in the TOR1A gene, leading to the deletion of a glutamic acid residue in the torsinA protein. Heterozygous knock-in mice, which reproduce the...

2018
Zuchra Zakirova Tomas Fanutza Justine Bonet Ben Readhead Weijia Zhang Zhengzi Yi Genevieve Beauvais Thomas P Zwaka Laurie J Ozelius Robert D Blitzer Pedro Gonzalez-Alegre Michelle E Ehrlich

Dystonia is characterized by involuntary muscle contractions. Its many forms are genetically, phenotypically and etiologically diverse and it is unknown whether their pathogenesis converges on shared pathways. Mutations in THAP1 [THAP (Thanatos-associated protein) domain containing, apoptosis associated protein 1], a ubiquitously expressed transcription factor with DNA binding and protein-inter...

2012
Anne J. Blood John K. Kuster Sandra C. Woodman Namik Kirlic Miriam L. Makhlouf Trisha J. Multhaupt-Buell Nikos Makris Martin Parent Lewis R. Sudarsky Greta Sjalander Henry Breiter Hans C. Breiter Nutan Sharma

BACKGROUND There has been increasing interest in the interaction of the basal ganglia with the cerebellum and the brainstem in motor control and movement disorders. In addition, it has been suggested that these subcortical connections with the basal ganglia may help to coordinate a network of regions involved in mediating posture and stabilization. While studies in animal models support a role ...

Journal: :Movement disorders : official journal of the Movement Disorder Society 2007
Leonardo Bonilha Paulien M de Vries Diana J Vincent Chris Rorden Paul S Morgan Mark W Hurd Nada Besenski Kenneth J Bergmann Vanessa K Hinson

We investigated whether structural white matter abnormalities, in the form of disruption of axonal coherence and integrity as measured with diffusion tensor imaging (DTI), constitute an underlying pathological mechanism of idiopathic dystonia (ID), independent of genotype status. We studied seven subjects with ID: all had cervical dystonia as their main symptom (one patient also had spasmodic d...

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