Neurology Unit, Department of Neuroscience, Bambino Gesù Children's Hospital, Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS), Rome, Italy.
Child Neurology and Psychiatry Unit, Department of System Medicine, Tor Vergata University of Rome, Rome, Italy.
Front Immunol. 2022 Aug 12;13:987968. doi: 10.3389/fimmu.2022.987968. eCollection 2022.
Bickerstaff brainstem encephalitis (BBE) is a rare, immune-mediated disease characterized by the acute onset of external ophthalmoplegia, ataxia, and consciousness disturbance. It has a complex multifactorial etiology, and a preceding infectious illness is seen in the majority of cases. Immune-mediated neurological syndromes following COVID-19 vaccination have been increasingly described. Here we report the case of a child developing BBE 2 weeks after COVID-19 vaccination. Despite nerve conduction studies and CSF analysis showing normal results, BBE was diagnosed on clinical ground and immunotherapy was started early with a complete recovery. Later, diagnosis was confirmed by positive anti-GQ1b IgG in serum. Even if there was a close temporal relationship between disease onset and COVID-19 vaccination, our patient also had evidence of a recent Mycoplasma pneumoniae infection that is associated with BBE. Indeed, the similarity between bacterial glycolipids and human myelin glycolipids, including gangliosides, could lead to an aberrantly immune activation against self-antigens (i.e., molecular mimicry). We considered the recent Mycoplasma pneumoniae infection a more plausible explanation of the disease onset. Our case report suggests that suspect cases of side effects related to COVID-19 vaccines need a careful evaluation in order to rule out well-known associated factors before claiming for a causal relationship.
比克斯特夫脑炎(Bickerstaff brainstem encephalitis,BBE)是一种罕见的、免疫介导的疾病,其特征为急性起病的眼外肌瘫痪、共济失调和意识障碍。它具有复杂的多因素病因,大多数情况下存在前驱感染性疾病。COVID-19 疫苗接种后出现的免疫介导性神经系统综合征已越来越多地被描述。在这里,我们报告了一例儿童在 COVID-19 疫苗接种后 2 周发生 BBE 的病例。尽管神经传导研究和 CSF 分析显示正常结果,但根据临床表现诊断为 BBE,并早期开始免疫治疗,完全康复。后来,血清中抗 GQ1b IgG 阳性证实了诊断。尽管疾病发作与 COVID-19 疫苗接种之间存在密切的时间关系,但我们的患者也有近期肺炎支原体感染的证据,该感染与 BBE 有关。实际上,细菌糖脂与包括神经节苷脂在内的人类髓鞘糖脂之间的相似性可能导致针对自身抗原的异常免疫激活(即分子模拟)。我们认为近期肺炎支原体感染更可能是疾病发作的原因。我们的病例报告表明,与 COVID-19 疫苗相关的疑似副作用病例需要进行仔细评估,以便在声称因果关系之前排除已知的相关因素。