Oguni M, Oguni H, Kozasa M, Fukuyama Y
Department of Pediatrics, Tokyo Women's Medical College, Japan.
Brain Dev. 1992 Nov;14(6):412-6. doi: 10.1016/s0387-7604(12)80351-2.
We report here a patient with frequent nocturnal attacks, characterized by paroxysmal dystonic posturing of the left arm and contraction of the lower face, lasting 20-30 seconds. The attacks occurred in clusters of up to 20 spasms during the NREM stage of sleep. Interictal EEG showed clear-cut epileptic discharges in the right frontal region. Repeated ictal video-EEG recordings revealed only arousal pattern immediately before and during seizures except in those which evolved to left sided clonic hemiconvulsions. The dystonic attacks here described share many features of nocturnal paroxysmal dystonia (NPD) of Lugaresi and Cirignotta in common, including nocturnal periodic occurrence, absence of ictal EEG changes during attacks, and favorable response to CBZ, except for their unilateral nature. It was assumed that our case may be a lateralized variant of NPD and would further support the hypothesis that NPD is one particular form of frontal lobe epilepsy.
我们在此报告一名患者，其频繁出现夜间发作，表现为左臂阵发性肌张力障碍姿势及下脸部收缩，持续20 - 30秒。发作成簇出现，在非快速眼动睡眠阶段多达20次痉挛。发作间期脑电图显示右侧额叶区域有明确的癫痫放电。多次发作期视频脑电图记录显示，除了那些演变为左侧阵挛性半身惊厥的发作外，在发作前和发作期间仅显示觉醒模式。这里描述的肌张力障碍发作与Lugaresi和Cirignotta的夜间阵发性肌张力障碍（NPD）有许多共同特征，包括夜间周期性发作、发作期间无发作期脑电图变化以及对卡马西平（CBZ）反应良好，只是其具有单侧性。据推测，我们的病例可能是NPD的一种单侧变异型，这将进一步支持NPD是额叶癫痫的一种特殊形式这一假说。
