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病例报告:一例罕见的咪喹莫特诱发的非典型寻常型天疱疮。

Case report: A rare case of imiquimod-induced atypical pemphigus vulgaris.

作者信息

Moro Francesco, Ciccone Davide, Fania Luca, Mariotti Feliciana, Salemme Adele, Rahimi Siavash, Pallotta Sabatino, Di Zenzo Giovanni

机构信息

Dermatology Clinic, Istituto Dermopatico dell'Immacolata (IDI)-IRCCS, Rome, Italy.

Molecular and Cell Biology Laboratory, Istituto Dermopatico dell'Immacolata (IDI)-IRCCS, Rome, Italy.

出版信息

Front Med (Lausanne). 2022 Nov 24;9:1054544. doi: 10.3389/fmed.2022.1054544. eCollection 2022.

Abstract

BACKGROUND

Pemphigus vulgaris is an autoimmune intraepithelial bullous disease involving the skin and the mucous membranes. Imiquimod, a topical therapy for skin basal cell carcinoma, is an amine that induces the production of tumor necrosis factor alfa, interleukin-1 and other cytokines. Pemphigus induced by drugs has been frequently reported, mostly after systemic therapy.

CASE PRESENTATION

We present the case of a 50-year-old man who developed skin, intraoral, and genital mucosae lesions 3 days after a treatment with Imiquimod for multiple superficial basal cell carcinoma of the trunk. Direct and indirect immunofluorescence results were compatible with the diagnosis of pemphigus vulgaris. Enzyme-linked immunosorbent assay was negative for desmoglein 1 and 3, but interestingly, by immunoblotting on keratinocyte extracts a band of 170 kDa was obtained by IgG. The patient, after interrupting Imiquimod application, started a treatment with prednisolone and in 4 weeks showed a complete remission.

CONCLUSION

Topical Imiquimod therapy might induce atypical pemphigus vulgaris in some patients.

摘要

背景

寻常型天疱疮是一种累及皮肤和黏膜的自身免疫性上皮内大疱性疾病。咪喹莫特是一种用于治疗皮肤基底细胞癌的局部用药,是一种能诱导肿瘤坏死因子α、白细胞介素-1和其他细胞因子产生的胺类物质。药物诱发的天疱疮已有频繁报道,大多发生在全身治疗之后。

病例报告

我们报告一例50岁男性患者,在用咪喹莫特治疗躯干多发浅表基底细胞癌3天后,出现皮肤、口腔内及生殖器黏膜损害。直接和间接免疫荧光结果符合寻常型天疱疮的诊断。酶联免疫吸附测定法检测桥粒芯糖蛋白1和3均为阴性,但有趣的是,通过对角质形成细胞提取物进行免疫印迹,IgG检测到一条170 kDa的条带。患者停用咪喹莫特后,开始使用泼尼松龙治疗,4周后完全缓解。

结论

局部应用咪喹莫特治疗可能会在一些患者中诱发非典型寻常型天疱疮。

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