Department of Pediatrics, St. Luke's International Hospital, 9-1 Akashi-cho, Chuo-ku, Tokyo, Japan, Division of Neurology, Saitama Children's Medical Center, 1-2 Shintoshin, Chuo-ku, Saitama, Japan, Department of Pediatrics and Adolescent Medicine, Tokyo Medical University, 6-7-1, Nishi-shinjuku, Shinjuku-ku, Tokyo, Japan.
Department of Pediatrics, St. Luke's International Hospital, 9-1 Akashi-cho, Chuo-ku, Tokyo, Japan.
Epileptic Disord. 2021 Dec 1;23(6):917-921. doi: 10.1684/epd.2021.1350.
Eyelid myoclonia with absences (EMA) is an epileptic syndrome characterised by eyelid myoclonia with or without absences, eye closure-induced paroxysms, and photosensitivity. The relationship between EMA and epileptic negative myoclonus has not previously been reported. Herein, we describe a case of a 10-year-old girl who presented with eyelid myoclonia, eye closure-induced paroxysms, and photosensitivity, which was compatible with the diagnosis of EMA. Ictal EEG depicted an eye closure-induced diffuse 3.0-4.5-Hz polyspike-and-wave complex, which was accompanied by eye fluttering (eyelid myoclonia). EMG disclosed a brief interruption (60-140 mseconds) of tonic contraction of the orbicularis oculi muscle, which was associated with the polyspike-and-wave complex on EEG. The findings led to the diagnosis of epileptic negative myoclonus. Eye closure-induced eyelid epileptic negative myoclonus, demonstrated in this patient, may be an atypical seizure type of EMA that represents an intermediate between eyelid myoclonia and epileptic negative myoclonus.
眼睑肌阵挛伴失神(EMA)是一种癫痫综合征,其特征为眼睑肌阵挛伴或不伴失神、闭眼诱发的发作以及光敏性。EMA 与癫痫性负性肌阵挛之间的关系以前尚未报道过。在此,我们描述了一例 10 岁女孩的病例,其表现为眼睑肌阵挛、闭眼诱发的发作和光敏性,符合 EMA 的诊断。发作期 EEG 显示闭眼诱发的弥漫性 3.0-4.5-Hz 多棘慢波复合波,伴有眼球颤动(眼睑肌阵挛)。肌电图显示眼轮匝肌的强直收缩短暂中断(60-140 毫秒),与 EEG 上的多棘慢波复合波相关。这些发现导致了癫痫性负性肌阵挛的诊断。在该患者中观察到的闭眼诱发的眼睑癫痫性负性肌阵挛可能是 EMA 的一种非典型发作类型,代表了眼睑肌阵挛和癫痫性负性肌阵挛之间的中间状态。