Hoffman Coryn L, Bharathan Navaneetha Krishnan, Shibata Yoshitaka, Giang William, Gudjonsson Johann E, Seykora John T, Prouty Stephen M, Stahley Sara N, Payne Aimee S, Kowalczyk Andrew P
Department of Dermatology, Penn State College of Medicine, Hershey, Pennsylvania, USA.
Department of Dermatology, University of Michigan Medical School, Ann Arbor, Michigan, USA.
J Invest Dermatol. 2025 Feb 3. doi: 10.1016/j.jid.2024.12.028.
Desmosomes are intercellular junctions that mediate cell-cell adhesion and are essential for maintaining tissue integrity. Pemphigus vulgaris (PV) is an autoimmune epidermal blistering disease caused by autoantibodies (IgG) targeting desmoglein 3, a desmosomal cadherin. PV autoantibodies cause desmosome disassembly and loss of cell-cell adhesion; however, the molecular signaling pathways that regulate these processes are not fully understood. Using high-resolution time-lapse imaging of live keratinocytes, we found that endoplasmic reticulum (ER) tubules make frequent and persistent contacts with internalizing desmoglein 3 puncta in keratinocytes treated with IgG of patients with PV. Biochemical experiments demonstrated that PV IgG activated ER stress signaling pathways, including both IRE1⍺ and PERK pathways, in cultured keratinocytes. Furthermore, ER stress transcripts were upregulated in the skin of patients with PV. Pharmacological inhibition of ER stress protects against PV IgG-induced desmosome disruption and loss of keratinocyte cell-cell adhesion, suggesting that ER stress may be an important pathomechanism and a therapeutically targetable pathway for PV treatment. These data support a model in which desmosome adhesion is integrated with ER function to serve as a cell adhesion stress sensor that is activated in blistering skin diseases.
桥粒是介导细胞间黏附的细胞间连接,对于维持组织完整性至关重要。寻常型天疱疮(PV)是一种自身免疫性表皮水疱病,由靶向桥粒芯糖蛋白3(一种桥粒钙黏蛋白)的自身抗体(IgG)引起。PV自身抗体导致桥粒解体和细胞间黏附丧失;然而,调节这些过程的分子信号通路尚未完全了解。通过对活角质形成细胞进行高分辨率延时成像,我们发现内质网(ER)小管与用PV患者IgG处理的角质形成细胞中内化的桥粒芯糖蛋白3斑点频繁且持续接触。生化实验表明,PV IgG在培养的角质形成细胞中激活了ER应激信号通路,包括IRE1α和PERK通路。此外,PV患者皮肤中的ER应激转录物上调。ER应激的药理学抑制可防止PV IgG诱导的桥粒破坏和角质形成细胞间黏附丧失,这表明ER应激可能是PV治疗的重要发病机制和可治疗的靶点通路。这些数据支持一种模型,即桥粒黏附与ER功能整合,作为一种在水疱性皮肤病中被激活的细胞黏附应激传感器。