Department of Forensic Medicine, Defense Institute of Forensic Science, Criminal Investigation Command, Ministry of National Defense, Seoul, Korea.
Department of Pathology and Translational Genomics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.
J Korean Med Sci. 2023 Mar 13;38(10):e78. doi: 10.3346/jkms.2023.38.e78.
We present an autopsy case of a 19-year-old man with a history of epilepsy whose unwitnessed sudden death occurred unexpectedly in the night. About 4 years before death, he was diagnosed with unilateral optic neuritis (ON). Demyelinating disease was suspected, but he was lost to follow up after the recovery. Six months before death, he received a second dose of mRNA coronavirus disease 2019 (COVID-19) vaccine. Three months before death, he experienced epileptic seizures for the first time. Seventeen days before death, he was infected with COVID-19, which showed self-limited course under home isolation. Several days before death, he complained of seizures again at night. Autopsy revealed multifocal gray-tan discoloration in the cerebrum. Histologically, the lesions consisted of active and inactive demyelinated plaques in the perivenous area of the white matter. Perivascular lymphocytic infiltration and microglial cell proliferation were observed in both white matter and cortex. The other major organs including heart and lung were unremarkable. Based on the antemortem history and postmortem findings, the cause of death was determined to be multiple sclerosis with suspected exacerbation. The direct or indirect involvement of cortex and deep gray matter by exacerbated multiple sclerosis may explain the occurrence of seizures. Considering the absence of other structural abnormalities except the inflammatory demyelination of the cerebrum, fatal arrhythmia or laryngospasm in the terminal epileptic seizure may explain his sudden unexpected death in the benign circumstances. In this case, the onset of seizure was preceded by COVID-19 vaccination, and the exacerbation of seizure was preceded by COVID-19 infection, respectively. Literature reporting first manifestation or relapse of multiple sclerosis temporally associated with COVID-19 vaccination or infection are reviewed.
我们报告了一例 19 岁癫痫病史患者的尸检病例，其夜间未经目击的猝死出乎意料地发生。大约在死亡前 4 年，他被诊断为单侧视神经炎（ON）。怀疑是脱髓鞘疾病，但在康复后他就失去了随访。在死亡前 6 个月，他接受了第二剂 mRNA 新冠病毒 2019（COVID-19）疫苗。在死亡前 3 个月，他第一次出现癫痫发作。在死亡前 17 天，他感染了 COVID-19，在家隔离下自行好转。在死亡前几天，他再次在夜间抱怨发作。尸检显示大脑多处呈灰-棕灰色变色。组织学上，病变由脑白质静脉周围区的活动和不活动脱髓鞘斑块组成。在白质和皮质中均观察到血管周围淋巴细胞浸润和小胶质细胞增生。包括心脏和肺在内的其他主要器官无明显异常。基于生前病史和尸检结果，死因被确定为多发性硬化症伴疑似恶化。恶化的多发性硬化症直接或间接累及皮质和深部灰质，可能解释了癫痫发作的发生。考虑到除大脑炎症性脱髓鞘外没有其他结构异常，终末期癫痫发作中的致命心律失常或喉痉挛可能解释了他在良性情况下的意外猝死。在这种情况下，癫痫发作的发作先于 COVID-19 疫苗接种，癫痫发作的恶化先于 COVID-19 感染。回顾了报告 COVID-19 疫苗接种或感染与多发性硬化症首次表现或复发时间相关的文献。
