Literature DB >> 26864810

Frequent Occurrence of Aplasia Cutis Congenita in Bullous Dermolysis of the Newborn.

Andrea Diociaiuti1, Daniele Castiglia, Simona Giancristoforo, Liliana Guerra, Vittoria Proto, Andrea Dotta, Renata Boldrini, Giovanna Zambruno, Maya El Hachem.   

Abstract

Bullous dermolysis of the newborn (BDN) is a subtype of dystrophic epidermolysis bullosa characterized by rapid improvement in skin fragility within the first months of life, associated with typical immunofluorescence and ultrastructural features. Inheritance can be autosomal dominant or recessive. We report here 4 cases of BDN, 2 of which presented with aplasia cutis congenita of the lower extremities. All patients improved rapidly and blister formation ceased by the third month of life in 3 cases. In these patients only residual milia, nail dystrophies and atrophic scarring at sites of aplasia cutis were visible by one year. Family history indicated dominant inheritance in 2 cases, confirmed by identification of COL7A1 mutation. Molecular analysis also revealed recessive inheritance in the 2 sporadic cases. A literature search identified several patients with BDN born with skin defects localized to the lower extremities. In conclusion, these findings indicate that aplasia cutis congenita is not an infrequent manifestation of BDN.

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Year:  2016        PMID: 26864810     DOI: 10.2340/00015555-2364

Source DB:  PubMed          Journal:  Acta Derm Venereol        ISSN: 0001-5555            Impact factor:   4.437


  3 in total

1.  Clinical practice guidelines for laboratory diagnosis of epidermolysis bullosa.

Authors:  C Has; L Liu; M C Bolling; A V Charlesworth; M El Hachem; M J Escámez; I Fuentes; S Büchel; R Hiremagalore; G Pohla-Gubo; P C van den Akker; K Wertheim-Tysarowska; G Zambruno
Journal:  Br J Dermatol       Date:  2019-08-09       Impact factor: 9.302

2.  Self-improving dominant dystrophic epidermolysis bullosa: phenotypic variability associated with COL7A1 mutation p.Gly2037Glu.

Authors:  Andrea Diociaiuti; Cristina Guerriero; Claudia Cesario; Sabrina Rossi; Roberta Rotunno; Giovanna Zambruno; May El Hachem
Journal:  Eur J Dermatol       Date:  2020-12-01       Impact factor: 3.328

3.  Case Report: Uncommon Association of ITGB4 and KRT10 Gene Mutation in a Case of Epidermolysis Bullosa With Pyloric Atresia and Aplasia Cutis Congenita.

Authors:  Melinda Matyas; Diana Miclea; Gabriela Zaharie
Journal:  Front Genet       Date:  2021-07-08       Impact factor: 4.599

  3 in total

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