Literature DB >> 15807692

Identification of novel type VII collagen gene mutations resulting in severe recessive dystrophic epidermolysis bullosa.

M Massé1, P B Cserhalmi-Friedman, V Falanga, J T Celebi, A Martinez-Mir, A M Christiano.   

Abstract

In this work, we studied the proband in a small nuclear family of Chinese and Dutch/German descent and identified two novel mutations in the type VII collagen gene leading to recessive dystrophic epidermolysis bullosa, Hallopeau-Siemens variant (HS-RDEB). The maternal mutation is a single base pair deletion of a cytosine nucleotide in exon 26, designated 3472delC, resulting in a frameshift and a premature termination codon (PTC) within the same exon, 7 bp downstream of the site of the mutation. The paternal mutation is a G-->A transition located at the 5' donor splice site within intron 51, designated IVS51 + 1G-->A. This mutation leads to the activation of a cryptic splice site, 32 bp downstream of the mutation site and to subsequent aberrant out-of-frame splicing, resulting in two alternative mRNA transcripts and a downstream PTC. To our knowledge, these two mutations have not been previously reported. These findings extend the body of evidence for compound heterozygous mutations leading to HS-RDEB and provide the basis for prenatal diagnosis in this family.

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Year:  2005        PMID: 15807692     DOI: 10.1111/j.1365-2230.2005.01763.x

Source DB:  PubMed          Journal:  Clin Exp Dermatol        ISSN: 0307-6938            Impact factor:   3.470


  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.  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

3.  Autoimmunity and Cytokine Imbalance in Inherited Epidermolysis Bullosa.

Authors:  Susanna Esposito; Sophie Guez; Annalisa Orenti; Gianluca Tadini; Giulietta Scuvera; Laura Corti; Alessia Scala; Elia Biganzoli; Emilio Berti; Nicola Principi
Journal:  Int J Mol Sci       Date:  2016-09-24       Impact factor: 5.923

4.  Transposon clusters as substrates for aberrant splice-site activation.

Authors:  Maria Elena Vilar Alvarez; Martin Chivers; Ivana Borovska; Steven Monger; Eleni Giannoulatou; Jana Kralovicova; Igor Vorechovsky
Journal:  RNA Biol       Date:  2020-09-23       Impact factor: 4.652

  4 in total

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