Literature DB >> 29556034

Clinical and molecular consequences of exon 78 deletion in DMD gene.

Monica Traverso1, Stefania Assereto2, Serena Baratto2, Michele Iacomino1, Marina Pedemonte2, Maria Cristina Diana2, Marta Ferretti2, Paolo Broda2, Carlo Minetti1,2, Elisabetta Gazzerro2, Francesca Madia3, Claudio Bruno2, Federico Zara3, Chiara Fiorillo4,5.   

Abstract

We present a 13-year-old patient with persistent increase of serum Creatine Kinase (CK) and myalgia after exertion. Skeletal muscle biopsy showed marked reduction of dystrophin expression leading to genetic analysis of DMD gene by MLPA, which detected a single deletion of exon 78. To the best of our knowledge, DMD exon 78 deletion has never been described in literature and, according to prediction, it should lead to loss of reading frame in the dystrophin gene. To further assess the actual effect of exon 78 deletion, we analysed cDNA from muscle mRNA. This analysis confirmed the absence of 32 bp of exon 78. Exclusion of exon 78 changes the open reading frame of exon 79 and generate a downstream stop codon, producing a dystrophin protein of 3703 amino acids instead of 3685 amino acids. Albeit loss of reading frame usually leads to protein degradation and severe phenotype, in this case, we demonstrated that deletion of DMD exon 78 can be associated with a functional protein able to bind DGC complex and a very mild phenotype. This study adds a novel deletion in DMD gene in human and helps to define the compliance between maintaining/disrupting the reading frame and clinical form of the disease.

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Year:  2018        PMID: 29556034     DOI: 10.1038/s10038-018-0439-6

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  3 in total

1.  EMQN best practice guidelines for genetic testing in dystrophinopathies.

Authors:  Carl Fratter; Raymond Dalgleish; Stephanie K Allen; Rosário Santos; Stephen Abbs; Sylvie Tuffery-Giraud; Alessandra Ferlini
Journal:  Eur J Hum Genet       Date:  2020-05-18       Impact factor: 4.246

2.  First Identification of RNA-Binding Proteins That Regulate Alternative Exons in the Dystrophin Gene.

Authors:  Julie Miro; Anne-Laure Bougé; Eva Murauer; Emmanuelle Beyne; Dylan Da Cunha; Mireille Claustres; Michel Koenig; Sylvie Tuffery-Giraud
Journal:  Int J Mol Sci       Date:  2020-10-21       Impact factor: 5.923

Review 3.  An Overview of Alternative Splicing Defects Implicated in Myotonic Dystrophy Type I.

Authors:  Andrea López-Martínez; Patricia Soblechero-Martín; Laura de-la-Puente-Ovejero; Gisela Nogales-Gadea; Virginia Arechavala-Gomeza
Journal:  Genes (Basel)       Date:  2020-09-22       Impact factor: 4.096

  3 in total

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