Literature DB >> 11523564

Novel mutation in the TOR1A (DYT1) gene in atypical early onset dystonia and polymorphisms in dystonia and early onset parkinsonism.

J C Leung1, C Klein, J Friedman, P Vieregge, H Jacobs, D Doheny, C Kamm, D DeLeon, P P Pramstaller, J B Penney, M Eisengart, J Jankovic, T Gasser, S B Bressman, D P Corey, P Kramer, M F Brin, L J Ozelius, X O Breakefield.   

Abstract

Dystonia is a movement disorder involving sustained muscle contractions and abnormal posturing with a strong hereditary predisposition and without a distinct neuropathology. In this study the TOR1A (DYT1) gene was screened for mutations in cases of early onset dystonia and early onset parkinsonism (EOP), which frequently presents with dystonic symptoms. In a screen of 40 patients, we identified three variations, none of which occurred in EOP patients. Two infrequent intronic single base pair (bp) changes of unknown consequences were found in a dystonia patient and the mother of an EOP patient. An 18-bp deletion (Phe323_Tyr328del) in the TOR1A gene was found in a patient with early onset dystonia and myoclonic features. This deletion would remove 6 amino acids close to the carboxy terminus, including a putative phosphorylation site of torsinA. This 18-bp deletion is the first additional mutation, beyond the GAG-deletion (Glu302/303del), to be found in the TOR1A gene, and is associated with a distinct type of early onset dystonia.

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Year:  2001        PMID: 11523564     DOI: 10.1007/s100480100111

Source DB:  PubMed          Journal:  Neurogenetics        ISSN: 1364-6745            Impact factor:   2.660


  39 in total

1.  RNA interference-mediated inhibition of wild-type Torsin A expression increases apoptosis caused by oxidative stress in cultured cells.

Authors:  Xue-Ping Chen; Xiao-Hui Hu; Shu-Hui Wu; Yang-Wei Zhang; Bo Xiao; Hui-Fang Shang
Journal:  Neurochem Res       Date:  2010-05-09       Impact factor: 3.996

Review 2.  Genetic and clinical features of primary torsion dystonia.

Authors:  Laurie J Ozelius; Susan B Bressman
Journal:  Neurobiol Dis       Date:  2010-12-17       Impact factor: 5.996

Review 3.  Torsins: not your typical AAA+ ATPases.

Authors:  April E Rose; Rebecca S H Brown; Christian Schlieker
Journal:  Crit Rev Biochem Mol Biol       Date:  2015-10-13       Impact factor: 8.250

Review 4.  Engineering animal models of dystonia.

Authors:  Janneth Oleas; Fumiaki Yokoi; Mark P DeAndrade; Antonio Pisani; Yuqing Li
Journal:  Mov Disord       Date:  2013-06-15       Impact factor: 10.338

5.  Decreased number of striatal cholinergic interneurons and motor deficits in dopamine receptor 2-expressing-cell-specific Dyt1 conditional knockout mice.

Authors:  Fumiaki Yokoi; Janneth Oleas; Hong Xing; Yuning Liu; Kelly M Dexter; Carly Misztal; Melinda Gerard; Iakov Efimenko; Patrick Lynch; Matthew Villanueva; Raul Alsina; Shiv Krishnaswamy; David E Vaillancourt; Yuqing Li
Journal:  Neurobiol Dis       Date:  2019-10-13       Impact factor: 5.996

6.  Earlier onset of motor deficits in mice with double mutations in Dyt1 and Sgce.

Authors:  Fumiaki Yokoi; Guang Yang; Jindong Li; Mark P DeAndrade; Tong Zhou; Yuqing Li
Journal:  J Biochem       Date:  2010-07-13       Impact factor: 3.387

7.  A knowledge-based approach for predicting gene-disease associations.

Authors:  Hongyi Zhou; Jeffrey Skolnick
Journal:  Bioinformatics       Date:  2016-06-09       Impact factor: 6.937

8.  Obsessive-compulsive disorder is not a clinical manifestation of the DYT1 dystonia gene.

Authors:  Gary A Heiman; Ruth Ottman; Rachel J Saunders-Pullman; Laurie J Ozelius; Neil J Risch; Susan B Bressman
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2007-04-05       Impact factor: 3.568

9.  Biochemical and cellular analysis of human variants of the DYT1 dystonia protein, TorsinA/TOR1A.

Authors:  Jasmin Hettich; Scott D Ryan; Osmar Norberto de Souza; Luís Fernando Saraiva Macedo Timmers; Shelun Tsai; Nadia A Atai; Cintia C da Hora; Xuan Zhang; Rashmi Kothary; Erik Snapp; Maria Ericsson; Kathrin Grundmann; Xandra O Breakefield; Flávia C Nery
Journal:  Hum Mutat       Date:  2014-07-17       Impact factor: 4.878

10.  Increased c-fos expression in the central nucleus of the amygdala and enhancement of cued fear memory in Dyt1 DeltaGAG knock-in mice.

Authors:  Fumiaki Yokoi; Mai T Dang; Courtney A Miller; Andrea G Marshall; Susan L Campbell; J David Sweatt; Yuqing Li
Journal:  Neurosci Res       Date:  2009-07-18       Impact factor: 3.304

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