Simabukuro Mateus Mistieri, Nóbrega Paulo Ribeiro, Pitombeira Milena, Cavalcante Wagner Cid Palmeira, Grativvol Ronnyson Susano, Pinto Lécio Figueira, Castro Luiz Henrique Martins, Nitrini Ricardo
Neurology Division, Hospital das Clínicas da Faculdade de Medicina da Universidade de São Paulo, SP, Brazil.
Hospital Geral de Fortaleza, Fortaleza, CE, Brazil.
Dement Neuropsychol. 2016 Oct-Dec;10(4):351-357. doi: 10.1590/s1980-5764-2016dn1004016.
Creutzfeldt-Jakob Disease (CJD) is the prototypical cause of rapidly progressive dementia (RPD). Nonetheless, efforts to exclude reversible causes of RPD that mimic prion disease are imperative. The recent expanding characterization of neurological syndromes associated with antibodies directed against neuronal cell surface or sympathic antigens, namely autoimmune encephalitis is shifting paradigms in neurology. Such antigens are well known proteins and receptors involved in synaptic transmission. Their dysfunction results in neuropsychiatric symptoms, psychosis, seizures, movement disorders and RPD. Faciobrachial dystonic seizure (FBDS) is a novel characterized type of seizure, specific for anti-LGI1 encephalitis.
In order to improve clinical recognition we report the cases of two Brazilian patients who presented with characteristic FDBS (illustrated by videos) and anti-LGI1 encephalitis.
We have included all patients with FBDS and confirmed anti-LGI1 encephalitis and video records of FDBS in two tertiary Brazilian centers: Department of Neurology of Hospital das Clínicas, Sao Paulo University, Sao Paulo, Brazil and Hospital Geral de Fortaleza, Fortaleza, Brazil between January 1, 2011 and December 31, 2015.
Both patients presented with clinical features of limbic encephalitis associated with FBDS, hyponatremia and normal CSF. None of them presented with tumor and both showed a good response after immunotherapy.
FBDSs may be confounded with myoclonus and occurs simultaneously with rapid cognitive decline. Unawareness of FDBS may induce to misdiagnosing a treatable cause of RPD as CJD.
克雅氏病(CJD)是快速进展性痴呆(RPD)的典型病因。尽管如此,排除模仿朊病毒病的RPD可逆病因的努力势在必行。最近,与针对神经元细胞表面或交感神经抗原的抗体相关的神经综合征(即自身免疫性脑炎)的特征不断扩展,正在改变神经病学的范式。此类抗原是参与突触传递的知名蛋白质和受体。它们的功能障碍会导致神经精神症状、精神病、癫痫、运动障碍和RPD。面臂肌张力障碍性癫痫(FBDS)是一种新型的癫痫类型,是抗LGI1脑炎所特有的。
为了提高临床识别能力,我们报告了两名巴西患者的病例,他们表现出特征性的FBDS(有视频展示)和抗LGI1脑炎。
我们纳入了2011年1月1日至2015年12月31日期间在巴西两个三级医疗中心(巴西圣保罗大学临床医院神经科和巴西福塔雷萨综合医院)出现FBDS且确诊为抗LGI1脑炎的所有患者以及FBDS的视频记录。
两名患者均表现出与FBDS、低钠血症和脑脊液正常相关的边缘性脑炎临床特征。他们均未出现肿瘤,且在免疫治疗后均显示出良好反应。
FBDS可能与肌阵挛混淆,并与快速认知衰退同时发生。对FBDS缺乏认识可能导致将RPD的可治疗病因误诊为CJD。