Eikeland Birte
Primary Health Clinic, A. Bergs vei 31, 5089 Bergen, Norway.
Case Rep Neurol Med. 2020 Jul 29;2020:3579419. doi: 10.1155/2020/3579419. eCollection 2020.
Research in the last few years has indicated that most voltage-gated potassium channel- (VGKC-) complex antibodies without leucine-rich glioma-inactivated protein 1 or contactin-associated protein-like 2 antibody specificity lack pathogenic potential and are not clear markers for autoimmune inflammation. Here we report on a patient with double-negative VGKC who developed severe peripheral nerve hyperexcitability, central nervous system symptoms with agitation and insomnia, dysautonomia, and systemic symptoms with weight loss, itch, and skin lesions. The disease started acutely one month after an episode of enteroviral pericarditis and responded well to immunotherapy. The patient is presumed to have developed a postinfectious immunotherapy-responsive autoimmune disease. In the setting of anti-VGKC positivity, it seems likely that anti-VGKC contributed to the pathogenesis of the patient's symptoms of nerve hyperexcitability and that the disease was caused by an acquired autoimmune effect on the neuronal kinetics of VGKC. It is still unknown whether or not there are unidentified extracellular molecular targets within the VGKC-complex, i.e., a novel surface antigen and a pathogenic antibody that can cause affected individuals to develop a peripheral nerve hyperexcitability syndrome. This case highlights the fact that less well-characterized autoimmune central and peripheral nervous system syndromes may have infectious triggers.
过去几年的研究表明,大多数不具有富含亮氨酸胶质瘤失活蛋白1或接触蛋白相关蛋白样2抗体特异性的电压门控钾通道(VGKC)复合物抗体缺乏致病潜力,并非自身免疫性炎症的明确标志物。在此,我们报告一例双阴性VGKC患者,该患者出现严重的周围神经兴奋性增高、伴有激越和失眠的中枢神经系统症状、自主神经功能障碍以及伴有体重减轻、瘙痒和皮肤病变的全身症状。该病在肠道病毒性心包炎发作后1个月急性起病,对免疫治疗反应良好。推测该患者患有一种感染后免疫治疗反应性自身免疫性疾病。在抗VGKC阳性的情况下,抗VGKC似乎促成了患者神经兴奋性增高症状的发病机制,且该病是由对VGKC神经元动力学的后天自身免疫效应所致。VGKC复合物内是否存在未被识别的细胞外分子靶点,即一种可导致受累个体发生周围神经兴奋性增高综合征的新型表面抗原和致病抗体,目前仍不清楚。该病例凸显了一个事实,即特征尚不明确的自身免疫性中枢和周围神经系统综合征可能有感染诱因。