Liu Xiaorui, Jiang Tiejia, Xu Lu, Zhang Weiran, Gao Feng
Department of Neurology, Children's Hospital, Zhejiang University School of Medicine, Hangzhou 310052, China.
Epilepsy Center, Children's Hospital, Zhejiang University School of Medicine, Hangzhou 310052, China.
Epilepsy Behav Rep. 2024 Dec 3;29:100732. doi: 10.1016/j.ebr.2024.100732. eCollection 2025 Mar.
We presented a 7-year-old boy with refractory Epileptic Encephalopathy with Spike-and-Wave Activation in Sleep (EE-SWAS) successfully managed with a combination of propofol and midazolam. His seizures began at age 2, initially controlled by multiple antiseizure medications (ASMs) for almost three years. At age 5, seizures recurred with electroencephalography (EEG) showing electrical status epilepticus in sleep (ESES) and a spike-wave index (SWI) of 85 %. High-dose methylprednisolone pulse therapy initially reduced the SWI to 50 %, but it relapsed to 80 % within six months. Despite further treatments, including methylprednisolone, midazolam infusion, and four combined ASMs, the SWI persisted between 75 % and 85 %, leading to progressively worsening cognitive impairment and subsequently a somnolent state with nearly continuous discharges. During hospitalization, a combination of propofol and midazolam significantly improved the condition, reducing the SWI to 50 % upon completion of the treatment period. Over a three-year follow-up, no ESES or seizures were reported, and cognitive function notably improved. Currently, there is no consensus on the treatment of ESES, which is sometimes refractory to medication and can result in partially irreversible cognitive impairment. Propofol in combination with midazolam has demonstrated effective suppression of ESES phenomena, presenting a promising treatment strategy for refractory ESES.
我们报告了一名7岁患有睡眠期棘波-慢波激活型难治性癫痫性脑病(EE-SWAS)的男孩,通过丙泊酚和咪达唑仑联合治疗成功得到控制。他的癫痫发作始于2岁,最初由多种抗癫痫药物(ASM)控制了近三年。5岁时,癫痫复发,脑电图(EEG)显示睡眠期癫痫性电持续状态(ESES),棘波-慢波指数(SWI)为85%。大剂量甲泼尼龙冲击疗法最初将SWI降至50%,但在六个月内又复发至80%。尽管进行了包括甲泼尼龙、咪达唑仑输注和四种ASM联合使用在内的进一步治疗,SWI仍持续在75%至85%之间,导致认知障碍逐渐恶化,随后进入嗜睡状态且几乎持续放电。住院期间,丙泊酚和咪达唑仑联合使用显著改善了病情,治疗期结束时SWI降至50%。在三年的随访中,未报告ESES或癫痫发作,认知功能明显改善。目前,对于ESES的治疗尚无共识,其有时对药物治疗难治,并可导致部分不可逆的认知障碍。丙泊酚与咪达唑仑联合已证明能有效抑制ESES现象,为难治性ESES提供了一种有前景的治疗策略。