Literature DB >> 20930056

Multiexon deletions account for 15% of congenital myasthenic syndromes with RAPSN mutations after negative DNA sequencing.

Karen Gaudon1, Isabelle Pénisson-Besnier, Brigitte Chabrol, Françoise Bouhour, Laurence Demay, Asma Ben Ammar, Stéphanie Bauché, Christophe Vial, Guillaume Nicolas, Bruno Eymard, Daniel Hantaï, Pascale Richard.   

Abstract

Congenital myasthenic syndromes (CMS) are a heterogeneous group of genetic disorders that give rise to a defect in neuromuscular transmission. We described here three patients with a characteristic phenotype of recessive CMS and presenting mutation in the gene encoding rapsyn (RAPSN). Familial analysis showed that one allelic mutation failed to be detected by direct sequencing. An allelic quantification on patient's DNA identified three novel multi-exon deletions of RAPSN. These three genomic rearrangements in RAPSN represent 15% of our CMS patients with RAPSN mutations and we emphasize that single-nucleotide polymorphism markers and a gene dosage method should be performed in addition to DNA direct sequencing analysis particularly when there is a genetic counselling issue.

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Year:  2010        PMID: 20930056     DOI: 10.1136/jmg.2010.081034

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  4 in total

1.  Congenital familial myasthenic syndromes: disease and course in an affected dizygotic twin pair.

Authors:  Piero Pavone; Andrea Domenico Praticò; Vito Pavone; Raffaele Falsaperla
Journal:  BMJ Case Rep       Date:  2013-01-29

2.  Copy number analysis reveals a novel multiexon deletion of the COLQ gene in congenital myasthenia.

Authors:  Wei Wang; Yanhong Wu; Chen Wang; Jinsong Jiao; Christopher J Klein
Journal:  Neurol Genet       Date:  2016-10-31

3.  Intragenic DOK7 deletion detected by whole-genome sequencing in congenital myasthenic syndromes.

Authors:  Yoshiteru Azuma; Ana Töpf; Teresinha Evangelista; Paulo José Lorenzoni; Andreas Roos; Pedro Viana; Hidehito Inagaki; Hiroki Kurahashi; Hanns Lochmüller
Journal:  Neurol Genet       Date:  2017-05-03

4.  Congenital myasthenic syndromes: Clinical and molecular report on 7 Sicilian patients.

Authors:  Piero Pavone; Agata Polizzi; Maria Roberta Longo; Katia Romano; Michele Vecchio; Andrea D Praticò; Raffaele Falsaperla
Journal:  J Pediatr Neurosci       Date:  2013-01
  4 in total

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