Clinical features of acute generalized exanthematous pustulosis caused by hydroxychloroquine in rheumatology patients and exploration of CARD14 gene mutations

Frontiers in Medicine

Medicine

Chang-ming Chen¹  Feng Luo²  Hong Xiong²  Xue-mei Yuan²  Yu-zheng Yang²  Xue-ming Yao³  Wu-kai Ma³ 

[1] Department of Rheumatology and Immunology, Second Affiliated Hospital of Guizhou University of Chinese Medicine, Guiyang, China;Graduate School, Guizhou University of Traditional Chinese Medicine, Guiyang, China;Graduate School, Guizhou University of Traditional Chinese Medicine, Guiyang, China;Department of Rheumatology and Immunology, Second Affiliated Hospital of Guizhou University of Chinese Medicine, Guiyang, China;

关键词: acute generalized exanthematous pustulosis;    hydroxychloroquine;    CARD14;    gene mutation;    gene sequencing;   

DOI  :  10.3389/fmed.2023.1161837

 received in 2023-02-08, accepted in 2023-03-20,  发布年份 2023

来源: Frontiers

【 摘 要 】

IntroductionAcute generalized exanthematous pustulosis (AGEP) is a rare condition characterized by superficial pustules following drug ingestion or infection. Currently, there is no clear link between rheumatism and AGEP. It has been described that hydroxychloroquine (HCQ) is a rare cause of acute generalized epidermal necrolysis (AGEP). Presently, there are limited studies on HCQ-induced AGEP. We aimed to explore the clinical features and associated gene expression of AGEP induced after HCQ treatment exposure in rheumatology patients.MethodsWe assessed patients with HCQ-induced AGEP diagnosed at the Second Affiliated Hospital of Guizhou University of Chinese Medicine between January 1, 2017, and May 1, 2022. We also reviewed similar cases reported in specific databases.ResultsThe study included five females (mean age, 40.2 years), and the mean time from initiation of HCQ treatment to symptom onset was 12.2 d. All patients received steroids and allergy medications after HCQ discontinuation, and the rash completely resolved within an average of 25.2 d. We performed whole exome sequencing and Sanger validation in our patient sample. CARD14 gene mutations were detected in three patients. Additionally, seven mutation sites were detected.DiscussionHCQ-induced AGEP may have a longer latency period and regression time than AGEP induced by other drugs. Our patients all experienced CARD14 gene mutations. AGEP often resolves with topical therapy and drug discontinuation, although some cases require systemic steroid therapy. In the future, patients with rheumatism should pay attention to the effectiveness of HCQ during treatment and be aware of the associated skin toxicity.

【 授权许可】

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Copyright © 2023 Luo, Yuan, Xiong, Yang, Chen, Ma and Yao.