Literature DB >> 9881948

Restoration of open reading frame resulting from skipping of an exon with an internal deletion in the COL7A1 gene.

P B Cserhalmi-Friedman1, J A McGrath, J E Mellerio, R Romero, J C Salas-Alanis, A S Paller, H C Dietz, A M Christiano.   

Abstract

Restoration of open reading frame resulting from exon skipping has been documented as a mechanism of rescuing an mRNA transcript containing mutations. In this study, we describe a mutation in a family with dystrophic epidermolysis bullosa consisting of a 16-bp deletion within exon 87 of the type VII collagen gene (COL7A1) and predicted to lead to a frameshift and downstream premature termination codon. Unexpectedly, mRNA analysis revealed instead that the intraexonic deletion led to skipping of exon 87 and subsequent restoration of the open reading frame. The phenotypes and patterns of inheritance observed in this family arise from three different mutations, all of which affect RNA processing. Restoration of open reading frame by exon skipping represents a previously undescribed mechanism of action of intraexonic deletion mutations.

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Year:  1998        PMID: 9881948

Source DB:  PubMed          Journal:  Lab Invest        ISSN: 0023-6837            Impact factor:   5.662


  4 in total

1.  Epidermolysis bullosa. II. Type VII collagen mutations and phenotype-genotype correlations in the dystrophic subtypes.

Authors:  Roslyn Varki; Sara Sadowski; Jouni Uitto; Ellen Pfendner
Journal:  J Med Genet       Date:  2006-09-13       Impact factor: 6.318

2.  A novel COL7A1 gene mutation in an Iranian individual suffering dystrophic epidermolysis bullosa.

Authors:  Hamid Galehdari; Gholamreza Mohammadian; Somayeh Azmoon; Bahaoddin Salehi; Mohammad Pedram
Journal:  J Mol Diagn       Date:  2010-02-26       Impact factor: 5.568

3.  Revertant mosaicism in junctional epidermolysis bullosa due to multiple correcting second-site mutations in LAMB3.

Authors:  Anna M G Pasmooij; Hendri H Pas; Maria C Bolling; Marcel F Jonkman
Journal:  J Clin Invest       Date:  2007-05       Impact factor: 14.808

4.  Two novel mutations on exon 8 and intron 65 of COL7A1 gene in two Chinese brothers result in recessive dystrophic epidermolysis bullosa.

Authors:  Ying Lin; Xue-Jun Chen; Wei Liu; Bo Gong; Jun Xie; Jun-Hao Xiong; Jing Cheng; Xi-Ling Duan; Zhao-Chun Lin; Lu-Lin Huang; Hui-Ying Wan; Xiao-Qi Liu; Lin-Hong Song; Zheng-Lin Yang
Journal:  PLoS One       Date:  2012-11-30       Impact factor: 3.240

  4 in total

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