Benucci Maurizio, Tramacere Luciana, Infantino Maria, Manfredi Mariangela, Grossi Valentina, Damiani Arianna, Gobbi Francesca Li, Piccininni Maristella, Zaccara Gaetano, Cincotta Massimo
Rheumatology Unit, S. Giovanni di Dio Hospital, Florence, Italy.
Neurology Unit, S. Giovanni di Dio Hospital, Florence, Italy.
Case Rep Neurol Med. 2020 Feb 14;2020:5697670. doi: 10.1155/2020/5697670. eCollection 2020.
We report the case of a 64-year-old man who presented with subacute memory, balance impairment, behavioral and mood changes, and epileptic seizures. Magnetic resonance imaging (MRI) showed bilateral hippocampal abnormalities. Brain [F]-FDG fluorodeoxyglucose positron emission tomography (PET) revealed hypometabolism in both the temporal lobe as well as in the left insular and parietal regions. The clinical and neuroradiological picture and the detection of anti-CASPR2 antibodies in serum oriented the diagnosis towards autoimmune limbic encephalitis. Intravenous high-dose steroid and immunoglobulin treatments were ineffective. We did not use rituximab for the presence of antibodies to HbcAg positivity. Tocilizumab given intravenously 8 mg/kg once a month for six months and then subcutaneously 162 mg every week for six months resulted in clinical and neuroradiological improvement. These data support the efficacy of tocilizumab in autoimmune limbic encephalitis associated with anti-CASPR2 antibodies, which has been sporadically reported in the literature.
我们报告了一例64岁男性患者,其表现为亚急性记忆、平衡障碍、行为和情绪改变以及癫痫发作。磁共振成像(MRI)显示双侧海马异常。脑[F]-氟脱氧葡萄糖正电子发射断层扫描(PET)显示颞叶以及左侧岛叶和顶叶区域代谢减低。临床和神经放射学表现以及血清中抗接触蛋白相关蛋白2(CASPR2)抗体的检测使诊断倾向于自身免疫性边缘叶脑炎。静脉注射高剂量类固醇和免疫球蛋白治疗无效。由于存在乙肝核心抗原(HbcAg)抗体阳性,我们未使用利妥昔单抗。每月静脉注射一次托珠单抗8 mg/kg,共6个月,然后每周皮下注射162 mg,共6个月,导致临床和神经放射学改善。这些数据支持托珠单抗在与抗CASPR2抗体相关的自身免疫性边缘叶脑炎中的疗效,这在文献中已有零星报道。