Department ofDermatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama, Japan.
Department ofChild Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan.
J Dermatol. 2018 May;45(5):592-595. doi: 10.1111/1346-8138.14253. Epub 2018 Feb 11.
We report two rare cases of childhood epilepsy patients who developed ethosuximide-induced Stevens-Johnson syndrome (SJS). Unlike typical SJS, the initial eruption of both patients presented well-demarcated, infiltrating firm papules mainly on the cheeks and the extensor aspects of the arms (case 1), and multiple vesicles on the soles and oral aphthosis (case 2), which closely mimicked viral exanthema. We diagnosed both patients with ethosuximide-induced SJS, based on the dosing period and the positive results of drug-induced lymphocyte stimulation test. Systemic corticosteroids are usually selected as a standard therapy for SJS, despite controversial results regarding their effectiveness. In case 1, an i.v. pulse therapy of methylprednisolone (30 mg/kg, 3 days consecutively) was initiated on day 7 from the onset of illness, and an i.v. immunoglobulin (400 mg/kg, 5 days consecutively) was added the following day. In case 2, an i.v. prednisone treatment (1 mg/kg, for 1 week) was initiated on day 4 from the onset. Eventually, the early therapeutic interventions resulted in good outcomes in both patients.
我们报告了两例儿童癫痫患者发生乙琥胺诱导的史蒂文斯-约翰逊综合征（SJS）的罕见病例。与典型的 SJS 不同，这两例患者的初始皮疹表现为界限清楚、浸润性坚实丘疹，主要位于脸颊和手臂伸侧（病例 1），以及足底多形性水疱和口腔阿弗他溃疡（病例 2），这些表现类似于病毒性出疹。我们根据用药时间和药物诱导的淋巴细胞刺激试验的阳性结果，诊断这两例患者均为乙琥胺诱导的 SJS。尽管关于其疗效存在争议，但全身皮质类固醇通常被选为 SJS 的标准治疗方法。在病例 1 中，在发病后第 7 天开始静脉注射甲泼尼龙（30mg/kg，连续 3 天），第 2 天开始静脉注射免疫球蛋白（400mg/kg，连续 5 天）。在病例 2 中，在发病后第 4 天开始静脉注射泼尼松龙治疗（1mg/kg，持续 1 周）。最终，这两例患者的早期治疗干预均取得了良好的效果。
