Literature DB >> 20055845

Epidermolysis bullosa pruriginosa in association with lichen planopilaris.

N Almaani1, L Liu, A Perez, A Robson, J E Mellerio, J A McGrath.   

Abstract

Epidermolysis bullosa pruriginosa (EBP) is a clinical variant of dominant or occasionally recessive, dystrophic epidermolysis bullosa (EB). Clinically, intense pruritus on a background of inherited skin fragility often leads to skin signs that resemble acquired inflammatory disorders such as hypertrophic lichen planus (LP) or nodular prurigo. Moreover, symptoms and signs may not appear until adult life, further compounding difficulties in distinguishing between inherited or acquired skin pathology. We describe a 61-year-old white British woman who developed EBP during her 40s, with lichenified plaques on the legs that resembled hypertrophic LP. Molecular screening of the COL7A1 gene showed a novel heterozygous glycine substitution in type VII collagen, designated p.G2290A, in keeping with dominant dystrophic EB. During her 50s, however, the patient developed new abnormalities with patchy scarring alopecia and perifollicular inflammation. Histological examination of a skin biopsy found features of lichen planopilaris. To our knowledge, this is the first example of a patient with EBP in whom the genetic disease does not merely resemble LP but is actually associated with coexisting acquired lichenoid skin pathology. Intriguingly, treatment with topical tacrolimus 0.03% led to marked improvement in the inflammation on the legs but had little effect on the scalp.

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Year:  2009        PMID: 20055845     DOI: 10.1111/j.1365-2230.2009.03568.x

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


  3 in total

1.  Epidermolysis Bullosa Pruriginosa Associated with Folliculitis Decalvans: Case Report and Review of the Literature.

Authors:  Maria Cecília Rivitti-Machado; Juliana T Toma; Vivianne M A Pompeu; Neusa Y S Valente; Isabella Doche
Journal:  Skin Appendage Disord       Date:  2017-12-22

2.  An incompletely penetrant novel mutation in COL7A1 causes epidermolysis bullosa pruriginosa and dominant dystrophic epidermolysis bullosa phenotypes in an extended kindred.

Authors:  Catherine S Yang; Yin Lu; Anita Farhi; Carol Nelson-Williams; Michael Kashgarian; Earl J Glusac; Richard P Lifton; Richard J Antaya; Keith A Choate
Journal:  Pediatr Dermatol       Date:  2012-04-20       Impact factor: 1.588

Review 3.  Therapeutic management of classic lichen planopilaris: a systematic review.

Authors:  Enzo Errichetti; Matteo Figini; Margherita Croatto; Giuseppe Stinco
Journal:  Clin Cosmet Investig Dermatol       Date:  2018-02-27
  3 in total

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