Utsunomiya Natsuko, Chino Takenao, Oyama Noritaka, Utsunomiya Akira, Yamaguchi Yuka, Takashima Wataru, Tokuriki Atsushi, Hasegawa Minoru
Department of Dermatology, Division of Medical Sciences, University of Fukui, Fukui, Japan.
Dermatol Online J. 2017 Nov 15;23(11):13030/qt7gj3j797.
Linear IgA bullous dermatosis (LABD) is a rare autoimmune bullous disorder characterized by linear deposits of IgA at the basement membrane zone(BMZ) and/or by circulating IgA anti-BMZ antibodies. Comparing with other immuno-bullous diseases, LABD represents a heterogeneous disease entitywith diversity of pathogenic IgA autoantibodies to different hemidesmosomal antigens and an association with malignancies and occasional drug use. We herein present an 82-year-old Japanese man with LABD, whose indirect immunofluorescence using 1M NaCl-split skin showed positive staining for IgA at the dermal side alone. Fluorescence overlay antigen mapping using laser scanning confocal microscopy (FOAM-LSCM) was employed to examine the in vivo bound patient's IgA, which was specific for type VII collagen (COL7), a prominent antigen of the sublamina densa. One year later, he developed malignant lymphoma, suggesting the diagnosis of paraneoplastic LABD. We reviewed 32 cases of sublamina-densa type LABD with anti-COL7 IgA antibodies thus far reported in the literature to compare the clinicopathological characteristics of this rare disease variant and emphasize that COL7 is the main autoantigen in sublamina densa disease.
线状IgA大疱性皮肤病(LABD)是一种罕见的自身免疫性大疱性疾病,其特征为IgA在基底膜带(BMZ)呈线状沉积和/或存在循环IgA抗BMZ抗体。与其他免疫性大疱性疾病相比,LABD是一种异质性疾病实体,其针对不同半桥粒抗原的致病性IgA自身抗体具有多样性,且与恶性肿瘤及偶尔的药物使用有关。我们在此报告一名82岁的日本男性LABD患者,其使用1M NaCl分离皮肤进行的间接免疫荧光显示仅在真皮侧IgA染色呈阳性。采用激光扫描共聚焦显微镜荧光叠加抗原定位法(FOAM-LSCM)检测患者体内结合的IgA,其对VII型胶原蛋白(COL7)具有特异性,COL7是致密板下的一种主要抗原。一年后,他患上了恶性淋巴瘤,提示副肿瘤性LABD的诊断。我们回顾了迄今为止文献中报道的32例具有抗COL7 IgA抗体的致密板下型LABD病例,以比较这种罕见疾病变体的临床病理特征,并强调COL7是致密板下疾病的主要自身抗原。