نتایج جستجو برای: dyt6 dystonia

تعداد نتایج: 6616  

Journal: :American journal of human genetics 2015
Gavin Charlesworth Plamena R Angelova Fernando Bartolomé-Robledo Mina Ryten Daniah Trabzuni Maria Stamelou Andrey Y Abramov Kailash P Bhatia Nicholas W Wood

Reports of primary isolated dystonia inherited in an autosomal-recessive (AR) manner, often lumped together as "DYT2 dystonia," have appeared in the scientific literature for several decades, but no genetic cause has been identified to date. Using a combination of homozygosity mapping and whole-exome sequencing in a consanguineous kindred affected by AR isolated dystonia, we identified homozygo...

Journal: :Neuron 2016
Joseph E. Rittiner Zachary F. Caffall Ricardo Hernández-Martinez Sydney M. Sanderson James L. Pearson Kaylin K. Tsukayama Anna Y. Liu Changrui Xiao Samantha Tracy Miranda K. Shipman Patrick Hickey Julia Johnson Burton Scott Mark Stacy Rachel Saunders-Pullman Susan Bressman Kristina Simonyan Nutan Sharma Laurie J. Ozelius Elizabeth T. Cirulli Nicole Calakos

Dystonia is a brain disorder causing involuntary, often painful movements. Apart from a role for dopamine deficiency in some forms, the cellular mechanisms underlying most dystonias are currently unknown. Here, we discover a role for deficient eIF2α signaling in DYT1 dystonia, a rare inherited generalized form, through a genome-wide RNAi screen. Subsequent experiments including patient-derived ...

Journal: :Journal of neurology, neurosurgery, and psychiatry 1997
P R Jarman O Bandmann C D Marsden N W Wood

OBJECTIVES To investigate the hypothesis that GTP cyclohydrolase I (GCH1) mutations are responsible for the phenotype of highly anticholinergic responsive dystonia in patients with apparent primary torsion dystonia. METHODS From 107 British patients with clinically diagnosed primary torsion dystonia, seven patients were identified with an excellent response to anticholinergic drugs. All six e...

Journal: :Journal of neurology, neurosurgery, and psychiatry 2016
Anna Sadnicka Panagiotis Kassavetis Isabel Pareés Anne Marthe Meppelink Katherine Butler Mark Edwards

Task-specific dystonia is a form of isolated focal dystonia with the peculiarity of being displayed only during performance of a specific skilled motor task. This distinctive feature makes task-specific dystonia a particularly mysterious and fascinating neurological condition. In this review, we cover phenomenology and its increasingly broad-spectrum risk factors for the disease, critically rev...

2006
Katherine Butler Karin Rosenkranz

In 1911, Oppenheim coined the term ‘dystonia’ to describe disordered motor control, characterised by an association of hypotonia and tonic muscle spasm. Focal hand dystonia is one form of this disorder, in which symptoms are often task-specific and occur during skilled movements such as writing (writer’s cramp) or playing a musical instrument (musician’s cramp). Much research has been conducted...

Journal: :The Journal of neuropsychiatry and clinical neurosciences 2004
Edward C Lauterbach Alan Freeman Robert L Vogel

The authors investigated the prevalence of DIS-ascertained DSM-III psychiatric disorders occurring in 28 patients with dystonia and 28 patients with Parkinson's Disease (PD). In patients with dystonia, lifetime prevalences of major depression (25.0%), bipolar disorder (7.1%), atypical bipolar disorder (7.1%), social phobia (17.9%), and generalized anxiety disorder (25.0%) were significantly mor...

2011
Mark J Edwards Araceli Alonso-Canovas Arnette Schrag Bastiaan R Bloem Philip D Thompson Kailash Bhatia

Fixed dystonia is a disabling disorder mainly affecting young women who develop fixed abnormal limb postures and pain after apparently minor peripheral injury. There is continued debate regarding its pathophysiology and management. We report 5 cases of fixed dystonia in patients who sought amputation of the affected limb. We place these cases in the context of previous reports of patients with ...

2010
L. Timmermann K. A. M. Pauls K. Wieland R. Jech G. Kurlemann N. Sharma S. S. Gill C. A. Haenggeli S. J. Hayflick P. Hogarth K. L. Leenders P. Limousin C. J. Malanga E. Moro J. L. Ostrem F. J. Revilla P. Santens A. Schnitzler S. Tisch F. Valldeoriola J. Vesper J. Volkmann D. Woitalla, S. Peker

Neurodegeneration with brain iron accumulation encompasses a heterogeneous group of rare neurodegenerative disorders that are characterized by iron accumulation in the brain. Severe generalized dystonia is frequently a prominent symptom and can be very disabling, causing gait impairment, difficulty with speech and swallowing, pain and respiratory distress. Several case reports and one case seri...

Journal: :Journal of neurophysiology 1999
F A Lenz N N Byl

A wide range of observations suggest that sensory inputs play a significant role in dystonia. For example, the map of the hand representation in the primary sensory cortex (area 3b) is altered in monkeys with dystonia-like movements resulting from overtraining in a gripping task. We investigated whether similar reorganization occurs in the somatic sensory thalamus of patients with dystonia (dys...

2015
Daniel Tibussek Ertan Mayatepek Dirk Klee Anne Koy

BACKGROUND Childhood arterial ischemic stroke (CAIS) is increasingly recognized as an important cause of significant long-term morbidity in the pediatric population. Post stroke movement disorders, above all hemi-dystonias, are much more common in children after stroke compared to adults. However, research in this field is largely lacking. By highlighting some important knowledge gaps, we aim t...

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