Li Zhimei, Cui Tao, Shi Weixiong, Wang Qun
Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, China National Clinical Research Center for Neurological Diseases, Beijing, China.
Medicine (Baltimore). 2016 Jul;95(28):e4244. doi: 10.1097/MD.0000000000004244.
We summarized the clinical characteristics of patients presenting with seizures and limbic encephalitis (LE) associated with leucine-rich glioma inactivated-1 protein antibody (LGI1) in order help recognize and treat this condition at its onset.We analyzed clinical, video electroencephalogram (VEEG), magnetic resonance imaging (MRI), and laboratory data of 10 patients who presented with LGI1-LE and followed up their outcomes from 2 to 16 (9.4 ± 4.2) months.All patients presented with seizures onset, including faciobrachial dystonic seizure (FBDS), partial seizure (PS), and generalized tonic-clonic seizure (GTCS). Four patients (Cases 3, 5, 7, and 8) had mild cognitive deficits. Interictal VEEG showed normal patterns, focal slowing, or sharp waves in the temporal or frontotemporal lobes. Ictal VEEG of Cases 4, 5, and 7 showed diffuse voltage depression preceding FBDS, a left frontal/temporal origin, and a bilateral temporal origin, respectively. Ictal foci could not be localized in other cases. MRI scan revealed T2/fluid-attenuated inversion recovery (FLAIR) hyperintensity and evidence of edema in the right medial temporal lobe in Case 3, left hippocampal atrophy in Case 5, hyperintensities in the bilateral medial temporal lobes in Case 7, and hyperintensities in the basal ganglia and frontal cortex in Case 10. All 10 serum samples were positive for LGI1 antibody, but it was only detected in the cerebrospinal fluid (CSF) of 7 patients. Five patients (Cases 2, 4, 6, 7, and 8) presented with hyponatremia. One patient (Case 2) was diagnosed with small cell lung cancer. While responses to antiepileptic drugs (AEDs) were poor, most patients (except Case 2) responded favorably to immunotherapy.LGI1-LE may initially manifest with various types of seizures, particularly FBDS and complex partial seizures (CPS) of mesial temporal origin, and slowly progressive cognitive involvement. Clinical follow-up, VEEG monitoring, and MRI scan are helpful in early diagnosis. Immunotherapy is effective for the treatment of both seizure and LE associated with LGI1 antibody. Although mostly nonparaneoplastic, tumor screening is recommended in some cases.
我们总结了伴有富含亮氨酸胶质瘤失活-1蛋白抗体(LGI1)的癫痫发作和边缘性脑炎(LE)患者的临床特征,以便在疾病发作时帮助识别和治疗该病症。我们分析了10例LGI1-LE患者的临床、视频脑电图(VEEG)、磁共振成像(MRI)及实验室数据,并对其2至16(9.4±4.2)个月的预后进行了随访。所有患者均有癫痫发作起病,包括面臂肌张力障碍性发作(FBDS)、部分性发作(PS)和全身强直阵挛性发作(GTCS)。4例患者(病例3、5、7和8)有轻度认知缺陷。发作间期VEEG显示正常图形、局灶性慢波或颞叶或额颞叶尖波。病例4、5和7的发作期VEEG分别显示FBDS前弥漫性电压降低、左侧额叶/颞叶起源和双侧颞叶起源。其他病例发作期病灶无法定位。MRI扫描显示,病例3右侧颞叶内侧T2/液体衰减反转恢复序列(FLAIR)高信号及水肿,病例5左侧海马萎缩,病例7双侧颞叶内侧高信号,病例10基底节和额叶皮质高信号。所有10份血清样本LGI1抗体均为阳性,但仅在7例患者的脑脊液(CSF)中检测到。5例患者(病例2、4、6