Diociaiuti Andrea, Castiglia Daniele, Giancristoforo Simona, Guerra Liliana, Proto Vittoria, Dotta Andrea, Boldrini Renata, Zambruno Giovanna, El Hachem Maya
Dermatology Unit, Bambino Gesù Children's Hospital-IRCCS, P.zza Sant'Onofrio, 4, IT-00165 Rome, Italy.
Acta Derm Venereol. 2016 Aug 23;96(6):784-7. doi: 10.2340/00015555-2364.
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.
新生儿大疱性皮肤松解症(BDN)是营养不良性大疱性表皮松解症的一种亚型,其特征为在生命的最初几个月内皮肤脆性迅速改善,并伴有典型的免疫荧光和超微结构特征。遗传方式可为常染色体显性或隐性。我们在此报告4例BDN,其中2例伴有下肢先天性皮肤发育不全。所有患者病情迅速改善,3例在出生后第三个月水泡形成停止。在这些患者中,到1岁时仅可见先天性皮肤发育不全部位残留的粟丘疹、指甲营养不良和萎缩性瘢痕。家族史显示2例为显性遗传,通过鉴定COL7A1突变得以证实。分子分析还显示2例散发病例为隐性遗传。文献检索发现数例BDN患者出生时存在局限于下肢的皮肤缺陷。总之,这些发现表明先天性皮肤发育不全是BDN的常见表现。