Literature DB >> 21856558

Extensive acantholysis as the major histological feature of a severe case of Dowling Meara-epidermolysis bullosa simplex: a reappraisal of acantholysis in the newborn.

Esteve Darwich1, Asunción Vicente, Maria C Bolling, Maria A González-Enseñat, Victoria Cusi, Claudia Fortuny, José A Bombí, Marcel F Jonkman, José M Mascaró.   

Abstract

Epidermolysis bullosa (EB) is a heterogeneous group of inherited skin disorders characterized by blistering and skin fragility secondary to mechanical trauma. Epidermolysis bullosa simplex (EBS) is the most frequent form of EB, with Dowling-Meara (DM-EBS) subtype being the most severe form in this group. Conventional histopathological evaluation is usually of low value in the diagnosis of EB, and significant histological features have rarely been reported in this group of diseases. We describe a case of severe DM-EBS in which acantholysis was observed in the histological examination. This finding led us to consider other diagnoses, such as neonatal pemphigus vulgaris or lethal acantholytic EB. Histological, immunological, ultrastructural and genetic tests were performed, leading to a final diagnosis of DM-EBS. Therefore, we believe that DM-EBS should be considered in the differential diagnosis of a newborn with blisters, where acantholysis is the main histological feature.

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Year:  2011        PMID: 21856558     DOI: 10.1684/ejd.2011.1497

Source DB:  PubMed          Journal:  Eur J Dermatol        ISSN: 1167-1122            Impact factor:   3.328


  2 in total

1.  Distinct Impact of Two Keratin Mutations Causing Epidermolysis Bullosa Simplex on Keratinocyte Adhesion and Stiffness.

Authors:  Melanie Homberg; Lena Ramms; Nicole Schwarz; Georg Dreissen; Rudolf E Leube; Rudolf Merkel; Bernd Hoffmann; Thomas M Magin
Journal:  J Invest Dermatol       Date:  2015-05-11       Impact factor: 8.551

2.  MMP-9 and CXCL8/IL-8 are potential therapeutic targets in epidermolysis bullosa simplex.

Authors:  Thomas Lettner; Roland Lang; Alfred Klausegger; Stefan Hainzl; Johann W Bauer; Verena Wally
Journal:  PLoS One       Date:  2013-07-19       Impact factor: 3.240

  2 in total

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