Department of Dermatology, Keio University School of Medicine, Tokyo, Japan.
J Invest Dermatol. 2012 Apr;132(4):1141-8. doi: 10.1038/jid.2011.449. Epub 2012 Jan 26.
Paraneoplastic pemphigus (PNP) is an autoimmune blistering disease associated with lymphoproliferative neoplasms and characterized by antibodies against plakins and desmoglein 3 (Dsg3). Anti-Dsg3 antibodies have a primary role in blister formation in PNP. In this study, we used phage display to clone monoclonal anti-Dsg3 antibodies from a PNP patient to further characterize their pathogenicity. We isolated 20 unique Dsg3-reactive mAbs, which we classified into four groups according to the heavy-chain complementarity-determining region 3 (CDR3) region. Genetic analyses demonstrated that three antibody groups used the VH1-46 gene (18 clones) and one group used the VH1-02 gene (2 clones). The results of an in vitro keratinocyte dissociation assay and a human skin organ culture injection assay showed that three antibodies displayed pathogenic activity in blister formation with different potencies. Epitope mapping using domain-swapped Dsg3/Dsg2 showed that these pathogenic mAbs bound Ca(2+)-dependent conformational epitopes in the middle portion of the extracellular region of Dsg3 (EC2 and EC3 domains), in contrast to most previously characterized pathogenic pemphigus vulgaris antibodies, which bound to the EC1 domain of Dsg3. These mAbs reflect the unique polyclonal nature of anti-Dsg3 antibodies in PNP and represent an important tool for detailing the pathophysiological mechanisms of blister formation in PNP.
副肿瘤天疱疮(PNP)是一种与淋巴增生性肿瘤相关的自身免疫性水疱病,其特征是针对桥粒蛋白和桥粒芯糖蛋白 3(Dsg3)的抗体。抗 Dsg3 抗体在 PNP 中水疱形成中起主要作用。在这项研究中,我们使用噬菌体展示技术从 PNP 患者中克隆单克隆抗 Dsg3 抗体,以进一步研究其致病性。我们分离出 20 种独特的 Dsg3 反应性 mAb,根据重链互补决定区 3(CDR3)区域将其分为四组。遗传分析表明,三个抗体组使用 VH1-46 基因(18 个克隆),一个组使用 VH1-02 基因(2 个克隆)。体外角质形成细胞解离试验和人皮肤器官培养注射试验的结果表明,三种抗体以不同的效力显示出在水疱形成中的致病性。使用域交换 Dsg3/Dsg2 的表位作图表明,这些致病性 mAb 结合 Dsg3 细胞外区中部(EC2 和 EC3 结构域)Ca(2+)依赖性构象表位,而大多数先前表征的致病性寻常性天疱疮抗体则结合 Dsg3 的 EC1 结构域。这些 mAb 反映了 PNP 中抗 Dsg3 抗体的独特多克隆性质,代表了详细研究 PNP 中水疱形成的病理生理机制的重要工具。