Patch testing is an effective method for the diagnosis of carbamazepine-induced drug reaction, eosinophilia and systemic symptoms (DRESS) syndrome in an 8-year-old girl

Patch testing is an effective method for the diagnosis of carbamazepine-induced drug reaction, eosinophilia and systemic symptoms (DRESS) syndrome in an 8-year-old girl
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DOI:
10.1111/j.1440-0960.2012.00887.x
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发表时间:
2012-11-01
影响因子:
2
通讯作者:
Sackesen, Cansin
Sackesen, Cansin
中科院分区:
医学4区
文献类型:
--
作者:
Buyuktiryaki, Ayse Betul;Bezirganoglu, Handan;Sackesen, Cansin

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药物反应、嗜酸性粒细胞增多和全身症状(DRESS)是一种急性和危及生命的疾病,其特征为发热、皮疹和全身症状,包括淋巴结病、肝功能异常、间质性肾炎、肺和心脏浸润以及血液学异常伴嗜酸性粒细胞增多和非典型淋巴细胞。与DRESS最相关的药物是抗惊厥药、别嘌呤醇、米诺环素和磺胺类药物。这种综合征在儿童时期很少见,尽管大量儿童接受了抗惊厥治疗。一名8岁女孩因发热、淋巴结病和躯干皮疹入院。她有癫痫病史,5周前开始接受卡马西平治疗。实验室研究显示白色细胞计数为6200/mu L(正常,410011 200/mu L),嗜酸性粒细胞占22%,γ-谷氨酰转肽酶水平为296 U/L(正常,023 U/L)。感染和胶原蛋白疾病的实验室检查均在正常范围内。持续发热和斑丘疹伴全身脱屑,以及唇炎和面部血管性水肿的出现提示对卡马西平有超敏反应。用左乙拉西坦替代卡马西平。所有临床症状在皮质类固醇和抗组胺药治疗一周内改善。完全恢复后6周,进行卡马西平表皮斑贴试验,并在48小时观察到卡马西平诱导的阳性皮肤反应。卡马西平引起的DRESS综合征是一种罕见的儿童疾病。表皮斑贴试验是鉴别DRESS综合征诱导剂和鉴别安全抗惊厥药物的有用工具。
Drug reaction, eosinophilia and systemic symptoms (DRESS) is an acute and life-threatening disease, characterised by fever, rash and systemic symptoms, including lymphadenopathy, abnormal liver function, interstitial nephritis, pulmonary and cardiac infiltrates and haematological abnormalities with eosinophilia and atypical lymphocytes. The drugs mostly associated with DRESS are anticonvulsants, allopurinol, minocycline and sulfonamides. This syndrome is rarely seen in childhood even though a large number of children have anticonvulsant treatment. An 8-year-old girl was admitted with fever, lymphadenopathy and skin eruptions on her trunk. Her medical history was notable for epilepsy and carbamazepine treatment had been started 5 weeks previously. Laboratory studies showed a white cell count of 6200/mu L (normal, 410011 200/mu L) with 22% eosinophils and a gamma-glutamyl transpeptidase level of 296 U/L (normal, 023 U/L). Laboratory tests for infections and collagen diseases were in the normal range. Persistence of fever and maculopapular eruption with generalised desquamation and the appearance of cheilitis and facial angioedema suggested a hypersensitivity reaction to carbamazepine. The carbamazepine was replaced with levetiracetam. All clinical symptoms improved within a week with corticosteroids and antihistamine treatment. Six weeks after complete recovery an epicutaneous patch test with carbamazepine was performed and a carbamazepine-induced positive skin reaction was observed at 48-h. Carbamazepine-induced DRESS syndrome is a rare entity in children. An epicutaneous patch test is a useful tool for identifying the inducing agent for the DRESS syndrome and for identifying a safe anticonvulsant drug.