Department of Neurology, Juntendo University Faculty of Medicine, Japan.
Department of Neurology, Juntendo University Faculty of Medicine, Japan.
J Neuroimmunol. 2019 May 15;330:19-22. doi: 10.1016/j.jneuroim.2019.01.020. Epub 2019 Feb 1.
Neuromyelitis optica spectrum disorder (NMOSD) is an autoimmune disorder caused by antibody to aquaporin-4 (AQP4). NMOSD can infrequently present as a complication of posterior reversible encephalopathy syndrome (PRES). Moreover, few cases of NMOSD patients with brain hemorrhage have been reported. We report a rare case of PRES together with NMOSD recurrence, subsequent to pontine hemorrhage after intravenous methylprednisolone (IVMP) therapy. A 51-year-old Japanese woman, with a history of hypertension and dyslipidemia, and recurrent episodes of left visual acuity disorder related to AQP4-positive NMOSD, developed blindness in the left eye. Brain MRI showed a hyperintense lesion in pons. She was initially diagnosed with recurrence of NMOSD and 1000 mg of IVMP was administered for 3 days. After the 3rd course of IVMP, she developed left-sided sensory disturbance, and blood pressure was increased to 202/127 mmHg. Brain computed tomography (CT) showed pontine hemorrhage, and she was referred to our hospital again. We diagnosed PRES associated with NMOSD recurrence, along with development of pontine hemorrhage induced by the increase in blood pressure resulting from IVMP. The patient was treated with nicardipine to strictly control blood pressure, and tranexamic acid and glycerol for pontine hemorrhage and PRES. We also extended IVMP for 5 consecutive days in total, followed by plasmapheresis. After therapy, blindness in the left eye improved to light perception. Collectively, anti-AQP4 antibody could induce PRES together with recurrent NMOSD, and pontine hemorrhage could thus be induced by blood pressure increases resulting from IVMP.
视神经脊髓炎谱系疾病(NMOSD)是一种由抗水通道蛋白-4(AQP4)抗体引起的自身免疫性疾病。NMOSD 偶尔可作为后部可逆性脑病综合征(PRES)的并发症出现。此外,NMOSD 合并脑出血的病例报告较少。我们报告了一例罕见的 PRES 合并 NMOSD 复发病例,在静脉注射甲基泼尼松龙(IVMP)治疗后发生桥脑出血。一位 51 岁日本女性,有高血压和血脂异常病史,曾反复发作与 AQP4 阳性 NMOSD 相关的左眼视力障碍,左眼失明。脑部 MRI 显示桥脑高信号病变。最初诊断为 NMOSD 复发,给予 1000mg IVMP 治疗 3 天。第 3 个疗程 IVMP 后,出现左侧感觉障碍,血压升高至 202/127mmHg。脑 CT 显示桥脑出血,再次转入我院。我们诊断为 PRES 合并 NMOSD 复发,同时由于 IVMP 导致血压升高而发生桥脑出血。患者接受尼卡地平严格控制血压,并给予氨甲环酸和甘油治疗桥脑出血和 PRES。我们还将 IVMP 延长至连续 5 天,随后进行血浆置换。治疗后,左眼失明改善至光感。总之,抗 AQP4 抗体可引起 PRES 合并 NMOSD 复发,IVMP 导致的血压升高可引发桥脑出血。