Literature DB >> 25683376

Clinical and genetic data on Lafora disease patients of Serbian/Montenegrin origin.

M Kecmanović1, N Jović2, M Keckarević-Marković1, D Keckarević1, G Stevanović2, P Ignjatović2, S Romac1.   

Abstract

Lafora disease (LD) is an autosomal recessive, progressive disorder characterized by myoclonus and seizures, inexorable neurologic deterioration, cognitive decline and poor prognosis. LD is caused by mutations either in the EPM2A or in NHLRC1 genes. Here we report clinical and genetic findings on 14 LD patients from 10 families of Serbian/Montenegrin origin. Molecular diagnostics was performed by sequencing the coding regions of the EPM2A and NHLRC1 genes. In addition, haplotype analysis of the chromosomes carrying the two most frequent mutations (c.1048-1049delGA and deletion of the whole NHLRC1 gene) using eight different markers flanking the NHLRC1 gene was conducted. We identified one new mutation (c.1028T>C) along with the 3 previously reported mutations (c.1048-1049delGA, c.990delG, deletion of the whole NHLRC1 gene), all of which were located on the NHLRC1 gene. The two predominant mutations (c.1048-1049delGA and complete NHLRC1 gene deletion) appear to be founder mutations. In addition to documenting the genetic heterogeneity observed for LD, our study suggests that mutations in the NHLRC1 gene may be a common cause of LD in the Serbian/Montenegrin population, primarily because of a founder effect.
© 2015 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  Lafora disease; NHLRC1 gene; Serbian/Montenegrin population; founder effect

Mesh:

Substances:

Year:  2015        PMID: 25683376     DOI: 10.1111/cge.12570

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  4 in total

1.  Italian cohort of Lafora disease: Clinical features, disease evolution, and genotype-phenotype correlations.

Authors:  Antonella Riva; Alessandro Orsini; Marcello Scala; Vittoria Taramasso; Laura Canafoglia; Giuseppe d'Orsi; Maria Teresa Di Claudio; Carlo Avolio; Alfredo D'Aniello; Maurizio Elia; Silvana Franceschetti; Giancarlo Di Gennaro; Francesca Bisulli; Paolo Tinuper; Maria Tappatà; Antonino Romeo; Elena Freri; Carla Marini; Cinzia Costa; Vito Sofia; Edoardo Ferlazzo; Adriana Magaudda; Pierangelo Veggiotti; Elena Gennaro; Angela Pistorio; Carlo Minetti; Amedeo Bianchi; Salvatore Striano; Roberto Michelucci; Federico Zara; Berge Arakel Minassian; Pasquale Striano
Journal:  J Neurol Sci       Date:  2021-03-20       Impact factor: 3.181

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

Review 3.  Genetics of Lafora progressive myoclonic epilepsy: current perspectives.

Authors:  Miljana Kecmanović; Milica Keckarević-Marković; Dušan Keckarević; Galina Stevanović; Nebojša Jović; Stanka Romac
Journal:  Appl Clin Genet       Date:  2016-05-02

4.  A recurrent homozygous NHLRC1 variant in siblings with Lafora disease.

Authors:  Nami Araya; Yukitoshi Takahashi; Masayuki Shimono; Tomofumi Fukuda; Mitsuhiro Kato; Mitsuko Nakashima; Naomichi Matsumoto; Hirotomo Saitsu
Journal:  Hum Genome Var       Date:  2018-07-12
  4 in total

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