Literature DB >> 14643920

Bioptically demonstrated Lafora disease without EPM2A mutation: a clinical and neurophysiological study of two sisters.

P Boccella1, P Striano, F Zara, F Barbieri, C Sarappa, G Vacca, F A de Falco, S Striano.   

Abstract

Lafora disease (LD) is an autosomal recessive inherited form of progressive myoclonic epilepsy with dementia and ataxia, usually presenting in the second decade of life and inexorably progressing until death. Neuropathological hallmarks are Lafora bodies, intracytoplasmic inclusions that can be found in neurons and in other tissues. LD gene (EPM2A), mapping on chromosome 6, encodes for a tyrosine phosphatase protein called laforin. However, up to 20% cases of LD are not genetically linked to chromosome 6. We report two sisters affected from bioptically diagnosed LD but without evidence of EPM2A mutation. Although familial cases of LD are already reported in literature, our observation leads to some considerations on clinical-electrophysiological evolution as well as to remark the genetic heterogeneity of this condition. In addition, we report the good effect of the Levetiracetam for the treatment of myoclonus in these patients, also demonstrated by the electrophysiological findings.

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Year:  2003        PMID: 14643920     DOI: 10.1016/j.clineuro.2003.07.003

Source DB:  PubMed          Journal:  Clin Neurol Neurosurg        ISSN: 0303-8467            Impact factor:   1.876


  4 in total

Review 1.  Lafora disease: epidemiology, pathophysiology and management.

Authors:  Thomas S Monaghan; Norman Delanty
Journal:  CNS Drugs       Date:  2010-07       Impact factor: 5.749

Review 2.  Lafora disease.

Authors:  Julie Turnbull; Erica Tiberia; Pasquale Striano; Pierre Genton; Stirling Carpenter; Cameron A Ackerley; Berge A Minassian
Journal:  Epileptic Disord       Date:  2016-09-01       Impact factor: 1.819

3.  Seizure control and improvement of neurological dysfunction in Lafora disease with perampanel.

Authors:  Maya Dirani; Wassim Nasreddine; Fatima Abdulla; Ahmad Beydoun
Journal:  Epilepsy Behav Case Rep       Date:  2014-09-29

4.  Levetiracetam in the treatment of epilepsy.

Authors:  Bassel Abou-Khalil
Journal:  Neuropsychiatr Dis Treat       Date:  2008-06       Impact factor: 2.570

  4 in total

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