| Literature DB >> 15807692 |
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.Entities:
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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