Literature DB >> 18263761

Lafora progressive myoclonus epilepsy: disease course homogeneity in a genetic isolate.

Julie Turnbull1, Santosh Kumar, Zhi-Ping Ren, Shanmugakonar Muralitharan, Taline Naranian, Cameron A Ackerley, Berge A Minassian.   

Abstract

Lafora epilepsy is characterized by starch formation in brain and skin and is diagnosed by skin biopsy or mutation detection. It has variable ages of onset (6-19 years) and death (18-32 years) even with the same mutation, likely due to extramutational factors. The authors identified 14 Lafora epilepsy patients in the genetic isolate of tribal Oman. The authors show that in this homogeneous environment and gene pool, the same mutation, EPM2B-c.468-469delAG, results in highly uniform ages of onset (14 years) and death (21 years). Biopsy, on the other hand, was not homogeneous (positive in 4/5 patients) and is, therefore, less sensitive than mutation testing.

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Year:  2008        PMID: 18263761     DOI: 10.1177/0883073807309245

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  5 in total

Review 1.  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

2.  Natural history of Lafora disease: a prognostic systematic review and individual participant data meta-analysis.

Authors:  Luca Vignatelli; Francesca Bisulli; Federica Pondrelli; Lorenzo Muccioli; Laura Licchetta; Barbara Mostacci; Corrado Zenesini; Paolo Tinuper
Journal:  Orphanet J Rare Dis       Date:  2021-08-16       Impact factor: 4.123

3.  An empirical pipeline for personalized diagnosis of Lafora disease mutations.

Authors:  M Kathryn Brewer; Maria Machio-Castello; Rosa Viana; Jeremiah L Wayne; Andrea Kuchtová; Zoe R Simmons; Sarah Sternbach; Sheng Li; Maria Adelaida García-Gimeno; Jose M Serratosa; Pascual Sanz; Craig W Vander Kooi; Matthew S Gentry
Journal:  iScience       Date:  2021-10-13

4.  Novel mutation in the NHLRC1 gene in a Malian family with a severe phenotype of Lafora disease.

Authors:  M Traoré; G Landouré; W Motley; M Sangaré; K Meilleur; S Coulibaly; S Traoré; B Niaré; F Mochel; A La Pean; A Vortmeyer; H Mani; K H Fischbeck
Journal:  Neurogenetics       Date:  2009-03-26       Impact factor: 2.660

5.  Lafora Disease: Report of a Rare Entity.

Authors:  Younis Al Mufargi; Asim Qureshi; Abdullah Al Asmi
Journal:  Cureus       Date:  2020-01-28
  5 in total

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