Literature DB >> 12391355

Clinical findings of a myoclonus-dystonia family with two distinct mutations.

D Doheny1, F Danisi, C Smith, C Morrison, M Velickovic, D De Leon, S B Bressman, J Leung, L Ozelius, C Klein, X O Breakefield, M F Brin, J M Silverman.   

Abstract

Myoclonus-dystonia has recently been associated with mutations in the epsilon-sarcoglycan gene (SCGE) on 7q21. Previously, the authors reported a patient with myoclonus-dystonia and an 18-bp deletion in the DYT1 gene on 9q34. The authors have now re-evaluated the patient harboring this deletion for mutations in the SGCE gene and identified a missense change. In the current study, the authors describe the clinical details of this family carrying mutations in two different dystonia genes. Further analysis of these mutations separately and together in cell culture and in animal models should clarify their functional consequences.

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Year:  2002        PMID: 12391355     DOI: 10.1212/wnl.59.8.1244

Source DB:  PubMed          Journal:  Neurology        ISSN: 0028-3878            Impact factor:   9.910


  18 in total

1.  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

2.  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

3.  Improved motor performance in Dyt1 ΔGAG heterozygous knock-in mice by cerebellar Purkinje-cell specific Dyt1 conditional knocking-out.

Authors:  Fumiaki Yokoi; Mai Tu Dang; Yuqing Li
Journal:  Behav Brain Res       Date:  2012-02-25       Impact factor: 3.332

4.  Improved survival and overt "dystonic" symptoms in a torsinA hypofunction mouse model.

Authors:  Fumiaki Yokoi; Fangfang Jiang; Kelly Dexter; Bryan Salvato; Yuqing Li
Journal:  Behav Brain Res       Date:  2019-12-28       Impact factor: 3.332

5.  Epsilon sarcoglycan mutations and phenotype in French patients with myoclonic syndromes.

Authors:  S Tezenas du Montcel; F Clot; M Vidailhet; E Roze; P Damier; C P Jedynak; A Camuzat; A Lagueny; L Vercueil; D Doummar; L Guyant-Maréchal; J-L Houeto; G Ponsot; S Thobois; M-A Cournelle; A Durr; F Durif; B Echenne; D Hannequin; C Tranchant; A Brice
Journal:  J Med Genet       Date:  2005-10-14       Impact factor: 6.318

6.  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

Review 7.  Anxiety and affective disorder comorbidity related to serotonin and other neurotransmitter systems: obsessive-compulsive disorder as an example of overlapping clinical and genetic heterogeneity.

Authors:  Dennis L Murphy; Pablo R Moya; Meredith A Fox; Liza M Rubenstein; Jens R Wendland; Kiara R Timpano
Journal:  Philos Trans R Soc Lond B Biol Sci       Date:  2013-02-25       Impact factor: 6.237

8.  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

9.  Exome sequencing for gene discovery: time does not stand still.

Authors:  Mark S LeDoux
Journal:  Ann Neurol       Date:  2012-10       Impact factor: 10.422

Review 10.  Update on pediatric dystonias: etiology, epidemiology, and management.

Authors:  Emilio Fernández-Alvarez; Nardo Nardocci
Journal:  Degener Neurol Neuromuscul Dis       Date:  2012-04-11
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