Mayuzumi M, Akiyama M, Nishie W, Ukae S, Abe M, Sawamura D, Hashimoto T, Shimizu H
Department of Dermatology, Hokkaido University Graduate School of Medicine, N15 W7, Sapporo 060-8638, Japan.
Br J Dermatol. 2006 Nov;155(5):1048-52. doi: 10.1111/j.1365-2133.2006.07443.x.
Epidermolysis bullosa acquisita (EBA) is an acquired subepidermal bullous disease characterized by IgG autoantibodies to type VII collagen, a major component of anchoring fibrils. Most patients with EBA are adult and develop autoantibodies to the noncollagenous (NC) 1 domain of type VII collagen. We describe a 4-year-old Japanese boy presenting pruritic vesicles and tense blisters over his whole body. Immunofluorescence studies revealed linear IgG/C3 deposits along the dermal-epidermal junction of the patient's skin, and circulating IgG autoantibodies mapping to the dermal side of 1 M NaCl-split skin. By immunoblotting analysis using dermal extracts as a substrate, the patient's IgG antibodies labelled a 290-kDa protein corresponding to type VII collagen. Immunoblotting studies using recombinant proteins demonstrated that the patient's circulating autoantibodies recognized not only the NC1 but also the NC2 domain of type VII procollagen. Review of the previously reported cases and the present case suggested that patients with EBA with autoantibodies to regions other than the NC1 domain are all children younger than 10 years of age with clinical features of an inflammatory phenotype.
获得性大疱性表皮松解症(EBA)是一种获得性表皮下大疱性疾病,其特征是针对VII型胶原蛋白(锚定原纤维的主要成分)产生IgG自身抗体。大多数EBA患者为成年人,且会产生针对VII型胶原蛋白非胶原(NC)1结构域的自身抗体。我们描述了一名4岁日本男孩,其全身出现瘙痒性水疱和紧张性大疱。免疫荧光研究显示,患者皮肤的真皮 - 表皮交界处有线性IgG/C3沉积,循环IgG自身抗体定位于1 M NaCl分离皮肤的真皮侧。以真皮提取物为底物进行免疫印迹分析,患者的IgG抗体标记了一条对应于VII型胶原蛋白的290 kDa蛋白。使用重组蛋白进行的免疫印迹研究表明,患者的循环自身抗体不仅识别前VII型胶原蛋白的NC1结构域,还识别NC2结构域。对先前报道的病例和本病例的回顾表明,具有针对NC1结构域以外区域的自身抗体的EBA患者均为10岁以下儿童,具有炎症表型的临床特征。