Sasikumar Sruthi, Vincent Jino, Gopinath Siby, Nambiar Vivek, Umesh Saraf Udit, Kannoth Sudheeran, Antony Dayana, Mathai Annamma, Anandakuttan Anandkumar
Neuroimmunology Laboratory, Department of Neurology, Amrita Institute of Medical Sciences, Amrita Viswavidyapeetham University, Ponekkara PO, Kochi, Kerala, 682041, India.
Neuroimmunology Laboratory, Amrita Institute of Medical Sciences, Amrita Viswavidyapeetham University, Kochi, Kerala, 682041, India.
Acta Neurol Belg. 2022 Oct;122(5):1323-1328. doi: 10.1007/s13760-022-02018-8. Epub 2022 Jul 18.
Extrapyramidal symptoms are seen in patients with leucine-rich glioma-inactivated 1 (LGI1) antibody-positive patients infrequently and this can be successfully treated with immunotherapy. This is a retrospective hospital-based study from 2013 to 2021 at a tertiary care referral hospital in South India. LGI1 antibody-positive cases with Faciobrachio-crural dystonic seizures [FBCDS] were identified by reviewing electronic medical records and Neuroimmunology laboratory register. Clinical and laboratory details and treatment outcomes were analysed. There was a total of 23 patients who were positive for LGI1 antibody. Of these, three cases had FBCDS (2 males, age range 30-76 years). Upon reviewing the records they had additional asymmetric parkinsonian features. All had similar presentations with progressive slowness of activities and gait and later went on to have paroxysmal events of sudden falls with vocalization. Prolonged VEEG monitoring captured the habitual event, which were confirmed to be FBCDS. MRI did not show significant structural abnormalities, CSF showed elevated proteins and normal cell in two and lymphocytic pleocytosis in one, PET scans ruled out malignancy. Of the three patients, two were completely relieved of FBCDS with immunosuppression and there was complete resolution of extrapyramidal features in all. Thus, the patients in our series of FBCDS showed additional features of parkinsonism which responded well to immunotherapy. Involvement of basal ganglia can explain all the manifestations of this phenotype. This series reveals a unique phenotype of the LGI1 antibody.
锥体外系症状在富含亮氨酸的胶质瘤失活1(LGI1)抗体阳性患者中很少见,且可通过免疫疗法成功治疗。这是一项于2013年至2021年在印度南部一家三级医疗转诊医院进行的基于医院的回顾性研究。通过查阅电子病历和神经免疫学实验室登记册,确定了患有面臂-股肌张力障碍性癫痫发作[FBCDS]的LGI1抗体阳性病例。分析了临床和实验室细节以及治疗结果。共有23例患者LGI1抗体呈阳性。其中,3例患有FBCDS(2例男性,年龄范围30 - 76岁)。查阅记录时发现他们还有其他不对称帕金森样特征。所有患者均有类似表现,即活动和步态逐渐迟缓,随后出现伴有发声的突然跌倒的阵发性事件。长时间视频脑电图监测捕捉到了习惯性事件,经证实为FBCDS。MRI未显示明显结构异常,脑脊液检查显示2例蛋白升高、细胞正常,1例淋巴细胞增多,PET扫描排除恶性肿瘤。3例患者中,2例通过免疫抑制完全缓解了FBCDS,所有患者的锥体外系特征均完全消失。因此,我们系列FBCDS患者表现出帕金森样的额外特征,对免疫疗法反应良好。基底神经节受累可以解释该表型的所有表现。本系列揭示了LGI1抗体的一种独特表型。