Autoimmune Bullous Diseases Research Center, Tehran University of Medical Sciences, Tehran, Iran.
Dana-Farber Cancer Institute, Harvard Medical School, Boston, Massachusetts, USA.
Dermatol Ther. 2022 Jun;35(6):e15475. doi: 10.1111/dth.15475. Epub 2022 Apr 9.
Pemphigus is a group of autoimmune diseases characterized by flaccid lesions on the skin and mucous membranes. In pemphigus vulgaris, the most common subtype of pemphigus, lesions might be appeared anywhere on the oral mucosa, mostly in the buccal mucosa. However, the gingiva is a less frequently affected site. Here, we performed a retrospective study at Tehran University of Medical Sciences, covering a two-year period to identify pemphigus patients with active lesions confined to the gingiva. Considering 1787 initially evaluated pemphigus cases, 512 (28.6%) were found to have a history of gingival involvement. Among them, 31 patients had only gingival involvement during their last visit, including 29 (93.5%) women and only two (6.5%) men. The mean of disease duration in this group was 5.29 ± 3.46 years, and they had gingival involvement for a mean of 23.9 ± 19.3 months. Of 28 patients, nine were negative for anti-Dsg3 and 24 were negative for anti-Dsg1. In 24 patients, who received rituximab, the mean pemphigus disease area index specifically for gingiva was 4.76 ± 0.74 at baseline, which had changed to 4.13 ± 0.75 and 3.26 ± 0.63 three and 6 months after rituximab administration, respectively. After 3 months, gingival lesions were either entirely resolved (n = 3), partially resolved (n = 11), remained unchanged (n = 2), or progressed (n = 7). Gingiva-confined presentation of lesions in pemphigus could be non-anti-Dsg1/3 dependent in some patients. Such patients do not respond well to conventional treatments and rituximab therapy. More studies on the pathogenesis of gingiva-confined presentation of pemphigus are required.
天疱疮是一组自身免疫性疾病,其特征为皮肤和黏膜松弛性病变。在寻常型天疱疮中,最常见的天疱疮亚型,病变可能出现在口腔黏膜的任何部位,主要在颊黏膜。然而,牙龈是较少受影响的部位。在这里,我们在德黑兰医科大学进行了一项回顾性研究,涵盖了两年的时间,以确定局限于牙龈的活动性病变的天疱疮患者。在最初评估的 1787 例天疱疮病例中,发现有 512 例(28.6%)有牙龈受累史。其中,31 例患者在最后一次就诊时仅出现牙龈受累,包括 29 例(93.5%)女性和仅 2 例(6.5%)男性。该组的疾病平均病程为 5.29±3.46 年,牙龈受累的平均时间为 23.9±19.3 个月。28 例患者中,9 例抗 Dsg3 阴性,24 例抗 Dsg1 阴性。在接受利妥昔单抗治疗的 24 例患者中,基线时专门针对牙龈的天疱疮疾病面积指数平均为 4.76±0.74,分别在利妥昔单抗治疗后 3 个月和 6 个月时变为 4.13±0.75 和 3.26±0.63。3 个月后,牙龈病变要么完全缓解(n=3),要么部分缓解(n=11),要么保持不变(n=2),要么进展(n=7)。在某些患者中,天疱疮病变的牙龈局限表现可能不依赖于抗 Dsg1/3。这些患者对常规治疗和利妥昔单抗治疗反应不佳。需要更多研究天疱疮牙龈局限表现的发病机制。