Baba Shimpei, Okanishi Tohru, Nozaki Toshiki, Ichikawa Naoki, Sakakura Kazuki, Nishimura Mitsuyo, Yonekawa Takahiro, Enoki Hideo, Fujimoto Ayataka
Department of Child Neurology, Comprehensive Epilepsy Center, Seirei Hamamatsu General Hospital, Hamamatsu 430-8558, Japan.
Epilepsy and Surgery, Comprehensive Epilepsy Center, Seirei Hamamatsu General Hospital, Hamamatsu 430-8558, Japan.
Brain Sci. 2021 Jun 22;11(7):827. doi: 10.3390/brainsci11070827.
Neonatal thalamic hemorrhage is a strong risk factor for developing encephalopathy with continuous spikes and waves during sleep (ECSWS), even when not accompanied by widespread cortical destruction. The efficacy and indication of resective epilepsy surgery in such patients has not yet been reported. A 4-year-old boy was diagnosed with ECSWS based on strong epileptiform activation during sleep and neurocognitive deterioration. He had a history of left thalamic hemorrhage related to a straight sinus thrombosis during the newborn period. He presented with daily absence seizures that were refractory to medical treatment. At age 5, he underwent intracranial electroencephalogram (EEG) recording using depth and subdural strip electrodes placed in the left thalamus and over bilateral cortex, respectively. Interictal and ictal epileptiform discharges were observed in the thalamus, always preceded by discharges in the left or right parietal lobe. Left hemispherotomy successfully normalized the EEG of his unaffected hemisphere and extinguished his seizures. This is the first case report documenting resective epilepsy surgery in a patient with ECSWS due to neonatal thalamic injury without widespread cerebral destruction. Based on intracranial EEG findings, his injured thalamus did not directly generate the EEG abnormalities or absence seizures on its own. Patients with ipsilateral neonatal thalamic injury and even mild lateralized cortical changes may be candidates for resective or disconnective surgery for ECSWS.
新生儿丘脑出血是发生睡眠期持续性棘慢波癫痫性脑病(ECSWS)的一个重要危险因素,即使没有广泛的皮质破坏也是如此。此类患者切除性癫痫手术的疗效和适应证尚未见报道。一名4岁男孩因睡眠期强烈的癫痫样放电和神经认知功能恶化被诊断为ECSWS。他有新生儿期左侧丘脑出血病史,与直窦血栓形成有关。他每天发作失神发作,药物治疗无效。5岁时,他接受了颅内脑电图(EEG)记录,分别在左侧丘脑和双侧皮质放置了深部电极和硬膜下条形电极。在丘脑中观察到发作间期和发作期癫痫样放电,总是先于左侧或右侧顶叶放电。左侧大脑半球切除术成功地使他未受影响半球的脑电图恢复正常,并终止了他的癫痫发作。这是第一例记录因新生儿丘脑损伤而非广泛脑破坏导致的ECSWS患者进行切除性癫痫手术的病例报告。根据颅内脑电图结果,他受损的丘脑本身并未直接产生脑电图异常或失神发作。同侧新生儿丘脑损伤甚至伴有轻度皮质侧化改变的患者可能是ECSWS切除性或离断性手术的候选者。