Literature DB >> 11260188

Dominant dystrophic epidermolysis bullosa presenting as familial nail dystrophy.

B Dharma1, C Moss, J A McGrath, J E Mellerio, A Ilchyshyn.   

Abstract

Nail dystrophy, a well-recognized feature of dystrophic epidermolysis bullosa (EB), is usually accompanied by skin fragility. We present a three-generation family with an autosomal dominant history of dystrophic nails, but without skin fragility or trauma-induced blisters. No specific diagnosis had been made. However, in the fourth generation, an infant presented with nail dystrophy, acral blistering and milia, raising the possibility of dominant dystrophic EB. This was confirmed by mutational analysis of the type VII collagen gene, COL7A1. We identified a glycine substitution mutation, G1776A, in exon 61 of COL7A1, characteristic of dominant dystrophic EB, which segregated with nail dystrophy in this family. A diagnosis of dominant dystrophic EB should be considered in families with autosomal dominant nail dystrophy even when there is no history of blistering.

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Year:  2001        PMID: 11260188

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


  5 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.  Dominant-negative effects of COL7A1 mutations can be rescued by controlled overexpression of normal collagen VII.

Authors:  Anja Fritsch; Sashko Spassov; Susanne Elfert; Andreas Schlosser; Yannick Gache; Guerrino Meneguzzi; Leena Bruckner-Tuderman
Journal:  J Biol Chem       Date:  2009-09-02       Impact factor: 5.157

3.  Diagnostic pitfalls in newborns and babies with blisters and erosions.

Authors:  Elke Nischler; Alfred Klausegger; Clemens Hüttner; Gabriele Pohla-Gubo; Anja Diem; Johann W Bauer; Helmut Hintner
Journal:  Dermatol Res Pract       Date:  2010-01-20

Review 4.  Dystrophic epidermolysis bullosa: a review.

Authors:  Satoru Shinkuma
Journal:  Clin Cosmet Investig Dermatol       Date:  2015-05-26

5.  Foot care in epidermolysis bullosa: evidence-based guideline.

Authors:  M T Khan; M O'Sullivan; B Faitli; J E Mellerio; R Fawkes; M Wood; L D Hubbard; A G Harris; L Iacobaccio; T Vlahovic; L James; L Brains; M Fitzpatrick; K Mayre-Chilton
Journal:  Br J Dermatol       Date:  2019-10-23       Impact factor: 9.302

  5 in total

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