Kanwar Amrinder J, Vinay Keshavamurthy, Saikia Uma N, Koga Hiroshi, Teye Kwesi, Tsuruta Daisuke, Hashimoto Takashi
Department of Dermatology, Venereology and Leprology, Postgraduate Institute of Medical Education and Research, Chandigarh, India.
Indian J Dermatol Venereol Leprol. 2014 Jan-Feb;80(1):46-50. doi: 10.4103/0378-6323.125499.
IgG/IgA pemphigus is an extremely rare subset of pemphigus, showing anti-keratinocyte cell surface antibodies of both IgG and IgA classes. Herein, we describe a unique case of IgG/IgA pemphigus with clinical features of edematous erythema and peripheral vesiculopustules. Histopathology showed the presence of subcorneal pustules and acantholytic blisters in the mid-epidermis with neutrophilic infiltration and eosinophilic spongiosis. Direct immunofluorescence of perilesional skin showed both IgG and IgA deposits to keratinocyte cell surfaces and unusual granular deposits of IgG, IgM, and C3 along basement membrane zone. On enzyme linked immunosorbent assay , the auto-antibodies were found to be reactive to desmoglein 1 antigen. Various clinical, histopathological, and immunological findings in our case overlapped with the features of IgA pemphigus, pemphigus herpetiformis, and pemphigus foliaceus. These findings indicate that IgG/IgA pemphigus may be a transitional form between IgA pemphigus and pemphigus herpetiformis, and thus provides insight into the pathogenicity of this rare disorder.
IgG/IgA 类天疱疮是天疱疮中极为罕见的一个亚型,表现为同时具有 IgG 和 IgA 两类抗角质形成细胞表面抗体。在此,我们描述了一例独特的 IgG/IgA 类天疱疮病例,其临床特征为水肿性红斑和外周水疱脓疱。组织病理学显示角质层下脓疱及表皮中部棘层松解性水疱,伴有中性粒细胞浸润和嗜酸性海绵形成。皮损周围皮肤的直接免疫荧光显示 IgG 和 IgA 均沉积于角质形成细胞表面,且 IgG、IgM 和 C3 沿基底膜带呈异常颗粒状沉积。酶联免疫吸附测定发现自身抗体与桥粒芯糖蛋白 1 抗原反应。我们病例中的各种临床、组织病理学和免疫学表现与 IgA 类天疱疮、疱疹样天疱疮和落叶型天疱疮的特征重叠。这些发现表明 IgG/IgA 类天疱疮可能是 IgA 类天疱疮和疱疹样天疱疮之间的一种过渡形式,从而为这种罕见疾病的发病机制提供了见解。