Literature DB >> 24297365

Myoclonus-dystonia and epilepsy in a family with a novel epsilon-sarcoglycan mutation.

Kristoffer Haugarvoll1, Charalampos Tzoulis, Gia T Tran, Bjørn Karlsen, Bernt A Engelsen, Per M Knappskog, Laurence A Bindoff.   

Abstract

Seizures have been reported in two families with myoclonus-dystonia due to epsilon-sarcoglycan (SGCE) mutations. We report a Norwegian family with myoclonus-dystonia and epilepsy associated with a novel SGCE mutation. All six manifesting SGCE mutation carriers had myoclonus, and dystonia was present in two patients. Sequencing of the SGCE gene in the proband identified a novel frameshift c.372delG mutation that predicts the amino acid change [p.Lys125SerfsX7] and the formation of a premature stop codon. The mutation segregated with myoclonus-dystonia in the family. The typical motor symptoms were accompanied by generalized seizures in four of six affected mutation carriers. The seizure type included febrile, absence and generalized tonic-clonic seizures. One deceased patient with severe epilepsy and myoclonus could not be tested for the SGCE mutation. Seizures are rarely observed in myoclonus-dystonia patients with SGCE mutations, and may not be a part of the phenotype. The co-occurrence of seizures and myoclonus-dystonia suggests that they are both due to the same underlying SGCE mutation. However, with epilepsy being a relatively common disorder and lack of complete co-segregation in our and previous families, it is possible that some patients suffer from two different genetic disorders. The presence of seizures and EEG abnormalities should not be considered exclusion criteria for the diagnosis of myoclonus-dystonia.

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Year:  2013        PMID: 24297365     DOI: 10.1007/s00415-013-7203-9

Source DB:  PubMed          Journal:  J Neurol        ISSN: 0340-5354            Impact factor:   4.849


  28 in total

1.  Bilateral deep brain stimulation of the pallidum for myoclonus-dystonia due to ε-sarcoglycan mutations: a pilot study.

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Journal:  Arch Neurol       Date:  2011-01

Review 2.  Management of patients with myoclonus: available therapies and the need for an evidence-based approach.

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Journal:  Lancet Neurol       Date:  2010-10       Impact factor: 44.182

3.  SCN1A testing for epilepsy: application in clinical practice.

Authors:  Shinichi Hirose; Ingrid E Scheffer; Carla Marini; Peter De Jonghe; Eva Andermann; Alica M Goldman; Marcelo Kauffman; Nigel C K Tan; Daniel H Lowenstein; Sanjay M Sisodiya; Ruth Ottman; Samuel F Berkovic
Journal:  Epilepsia       Date:  2013-04-15       Impact factor: 5.864

4.  Inherited myoclonus-dystonia and epilepsy: further evidence of an association?

Authors:  Sean O'Riordan; Laurie J Ozelius; Patricia de Carvalho Aguiar; Michael Hutchinson; Mary King; Tim Lynch
Journal:  Mov Disord       Date:  2004-12       Impact factor: 10.338

5.  Phenotype-genotype correlation in Dutch patients with myoclonus-dystonia.

Authors:  M C F Gerrits; E M J Foncke; R de Haan; K Hedrich; Y L C van de Leemput; F Baas; L J Ozelius; J D Speelman; C Klein; M A J Tijssen
Journal:  Neurology       Date:  2006-03-14       Impact factor: 9.910

6.  Metabolic changes in DYT11 myoclonus-dystonia.

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Journal:  Neurology       Date:  2013-01-02       Impact factor: 9.910

7.  Evidence that paternal expression of the epsilon-sarcoglycan gene accounts for reduced penetrance in myoclonus-dystonia.

Authors:  Birgitt Müller; Katja Hedrich; Norman Kock; Natasa Dragasevic; Marina Svetel; Jennifer Garrels; Olfert Landt; Matthias Nitschke; Peter P Pramstaller; Wolf Reik; Eberhard Schwinger; Jürgen Sperner; Laurie Ozelius; Vladimir Kostic; Christine Klein
Journal:  Am J Hum Genet       Date:  2002-11-20       Impact factor: 11.025

8.  Myoclonic dystonia.

Authors:  J A Obeso; J C Rothwell; A E Lang; C D Marsden
Journal:  Neurology       Date:  1983-07       Impact factor: 9.910

9.  The epsilon-sarcoglycan gene (SGCE), mutated in myoclonus-dystonia syndrome, is maternally imprinted.

Authors:  Monika Grabowski; Alexander Zimprich; Bettina Lorenz-Depiereux; Vera Kalscheuer; Friedrich Asmus; Thomas Gasser; Thomas Meitinger; Tim M Strom
Journal:  Eur J Hum Genet       Date:  2003-02       Impact factor: 4.246

10.  Myoclonus-dystonia: clinical and genetic evaluation of a large cohort.

Authors:  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
Journal:  J Neurol Neurosurg Psychiatry       Date:  2008-12-09       Impact factor: 10.154

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  2 in total

Review 1.  Genetic Aspects of Myoclonus-Dystonia Syndrome (MDS).

Authors:  Laila Rachad; Nadia El Kadmiri; Ilham Slassi; Hicham El Otmani; Sellama Nadifi
Journal:  Mol Neurobiol       Date:  2016-01-20       Impact factor: 5.590

2.  Exploring the pathogenesis of canine epilepsy using a systems genetics method and implications for anti-epilepsy drug discovery.

Authors:  Ze-Jia Cui; Ye-Mao Liu; Qiang Zhu; Jingbo Xia; Hong-Yu Zhang
Journal:  Oncotarget       Date:  2017-12-27
  2 in total

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