Watanabe Eri, Fujita Takako, Shimono Masayuki, Koike Haruki, Yasumoto Sawa, Hirose Shinichi
Department of Pediatrics, School of Medicine, Fukuoka University, Japan.
Nakatsu Municipal Hospital, Japan.
eNeurologicalSci. 2018 Aug 23;12:36-38. doi: 10.1016/j.ensci.2018.08.001. eCollection 2018 Sep.
We report a case of recurrent neuropathy with predominant autonomic and sensory involvement whose serum was positive for anti-ganglionic acetylcholine receptor (anti-gAChR) antibodies, a diagnostic marker of autoimmune autonomic ganglionopathy. An 11-year-old girl complained of numbness and limb pain after gastroenteritis. Although hyperalgesia and autonomic dysfunctions, such as orthostatic intolerance and gastrointestinal dysmotility subsequently developed, these symptoms faded after a few days. Similar sensory and autonomic impairments recurred three times within 12 months after the first episode. The sensory and autonomic symptoms were rapidly ameliorated by the administration of intravenous immunoglobulin (IVIg) at the second and third relapse; however, the symptoms persisted even after the administration of IVIg at the fourth relapse. The residual symptoms disappeared after methylprednisolone pulse therapy. The patient's serum was found to be positive for anti-gAChR antibodies at the second relapse, and was negative after methylprednisolone pulse therapy. Further studies are needed to clarify the efficacy of treatment and the nosological position in the spectrum of neuropathies that are associated with autonomic and sensory impairments.
我们报告一例以自主神经和感觉受累为主的复发性神经病病例,其血清抗神经节乙酰胆碱受体(anti-gAChR)抗体呈阳性,这是自身免疫性自主神经节病的诊断标志物。一名11岁女孩在患肠胃炎后出现肢体麻木和疼痛。随后出现痛觉过敏和自主神经功能障碍,如体位性不耐受和胃肠动力障碍,但这些症状在数天后消退。首次发作后的12个月内,类似的感觉和自主神经损伤复发了3次。在第二次和第三次复发时,静脉注射免疫球蛋白(IVIg)迅速改善了感觉和自主神经症状;然而,在第四次复发时,即使给予IVIg,症状仍持续存在。甲基强的松龙冲击治疗后残留症状消失。在第二次复发时发现患者血清抗gAChR抗体呈阳性,甲基强的松龙冲击治疗后转为阴性。需要进一步研究以阐明治疗效果以及在与自主神经和感觉损伤相关的神经病谱系中的疾病分类位置。