Zhu Bizhen, Wu Jinzhun, Chen Guobing, Yang Yungang, Yi Cuili
Department of Pediatrics, The First Affiliated Hospital of Xiamen University, Xiamen, China.
Front Endocrinol (Lausanne). 2019 Jul 16;10:474. doi: 10.3389/fendo.2019.00474. eCollection 2019.
Drug reaction with eosinophilia and systemic symptoms (DRESS), also known as drug-induced hypersensitivity syndrome (DIHS) is a rare, severe cutaneous adverse drug reaction characterized by fever, skin rashes, lymphadenopathy, leukocytosis with eosinophilia, and/or atypical lymphocytosis, and multiple visceral organ involvement. Moreover, patients with DRESS are at risk of developing autoimmune diseases including thyroiditis, diabetes mellitus (DM), and systemic lupus erythematosus (SLE), etc. several weeks or months after the initial resolution. We described a 9-month boy who was admitted to our hospital because of severe pneumonia and developed DRESS 3 weeks later. After the withdrawal of suspicious drug and administration of systemic corticosteroids, the patient's condition improved gradually. Nevertheless, hyperglycemia was detected 20 days after the initial onset of DRESS, and subsequent fulminant type 1 diabetes mellitus (F1DM) was diagnosed requiring continuous intravenous insulin infusion. After 13 months of follow-up, the blood glucose levels are now well-controlled. Literature research in PubMed for diabetes mellitus associated with DRESS showed 16 articles and 27 related case reports. Of 27 patients with DM related to DRESS, 11 were male, 16 were female. The mean age was 46 years. The duration from the onset of DRESS to the development of DM was 21 days on average. F1DM was diagnosed in 21 patients, T1DM was confirmed in 5 patients, and T2DM was only defined in 1 patient. Glutamic acid decarboxylase antibodies (GAD) were detected in 4 cases. Of 22 cases in which virus examination was carried out, evidence of virus reactivation was established in 16 cases (72.7%). Of patients with F1DM, 16 (88.9%) cases were evidenced by reactivation of herpes virus. A high frequency of HLA genotype and haplotype were found in 11 cases. DM was concomitant with acute pancreatitis in 3 patients and thyroiditis in 2 patients. No patients died from the disease. This work aims to raise awareness of long-term autoimmune sequelae in patients with DRESS.
药物超敏反应伴嗜酸性粒细胞增多和全身症状(DRESS),也称为药物性超敏反应综合征(DIHS),是一种罕见的严重皮肤药物不良反应,其特征为发热、皮疹、淋巴结病、白细胞增多伴嗜酸性粒细胞增多和/或非典型淋巴细胞增多,以及多内脏器官受累。此外,DRESS患者在初始症状缓解数周或数月后有发生自身免疫性疾病的风险,包括甲状腺炎、糖尿病(DM)和系统性红斑狼疮(SLE)等。我们描述了一名9个月大的男孩,因重症肺炎入院,3周后发生了DRESS。停用可疑药物并给予全身糖皮质激素治疗后,患者病情逐渐好转。然而,在DRESS初始发病20天后检测到高血糖,随后诊断为暴发性1型糖尿病(F1DM),需要持续静脉输注胰岛素。经过13个月的随访,目前血糖水平得到良好控制。在PubMed上对与DRESS相关的糖尿病进行文献研究,共找到16篇文章和27例相关病例报告。在27例与DRESS相关的糖尿病患者中,男性11例,女性16例。平均年龄为46岁。从DRESS发病到发生糖尿病的平均时间为21天。21例诊断为F1DM,5例确诊为T1DM,仅1例确诊为T2DM。4例检测到谷氨酸脱羧酶抗体(GAD)。在进行病毒检测的22例病例中,16例(72.7%)有病毒再激活的证据。在F1DM患者中,16例(88.9%)有疱疹病毒再激活的证据。11例患者中发现了高频的HLA基因型和单倍型。3例患者的糖尿病与急性胰腺炎同时发生,2例患者与甲状腺炎同时发生。无患者死于该疾病。这项工作旨在提高对DRESS患者长期自身免疫后遗症的认识。