Neuroimmunology and Multiple Sclerosis Research (IJ, JVMH, SL, RM, AL, and SS), Neurology Clinic, University Hospital Zurich, University of Zurich, Zurich, Switzerland; Department of Ophthalmology (JVMH, KPW, and KL), University Hospital Zurich, Zurich, Switzerland; Department of Ophthalmology (MP), Luzerner Kantonsspital, Lucerne, Switzerland; Clinical Department of Neurology (MR), Medical University of Innsbruck, Innsbruck, Austria; and Neurology Clinic (MW, KPW), University Hospital Zurich, University of Zurich, Zurich, Switzerland.
J Neuroophthalmol. 2019 Mar;39(1):3-7. doi: 10.1097/WNO.0000000000000669.
Recurrent optic neuritis (rON) associated with myelin oligodendrocyte glycoprotein (MOG)-specific antibodies has been initially reported to show a better clinical outcome than aquaporin-4 (AQP4)-seropositive ON in neuromyelitis optica spectrum disorder (NMOSD). Here, we characterize clinical and neuroimaging findings in severe cases of MOG antibody-positive and AQP4 antibody-negative bilateral rON.
Three male adults with rON (ages 18, 44, and 63 years) were evaluated with optical coherence tomography (OCT), MRI, cerebrospinal fluid (CSF), and serological studies.
All patients experienced >7 relapses of ON with severe reduction of visual acuity and partial response to steroid treatment. Optic nerves were affected bilaterally, although unilateral relapses were more frequent than simultaneous bilateral recurrences. Patients were MOG-seropositive but repeatedly tested negative for AQP4 antibodies. OCT showed severe thinning of the peripapillary retinal nerve fiber layer. On MRI, contrast-enhancing lesions extended over more than half the length of the optic nerve. CSF analyses during ON episodes were normal. Severe visual deficits accumulated over time in 2 of 3 patients, despite immunosuppressive therapy.
MOG-seropositive and AQP4-seronegative rON may be associated with an aggressive disease course and poor functional and structural outcomes. In contrast to previous reports, the severity and pattern of retinal and optic nerve damage closely resembled phenotypes commonly observed in AQP4-seropositive rON without fulfilling current diagnostic criteria for NMOSD.
最初报道称,与髓鞘少突胶质细胞糖蛋白 (MOG) 特异性抗体相关的复发性视神经炎 (rON) 比视神经脊髓炎谱系障碍 (NMOSD) 中水通道蛋白 4 (AQP4) 阳性 ON 的临床结局更好。在此,我们对 MOG 抗体阳性和 AQP4 抗体阴性双侧 rON 的严重病例的临床和神经影像学表现进行了特征描述。
对 3 名 rON 男性成人(年龄分别为 18 岁、44 岁和 63 岁)进行光学相干断层扫描 (OCT)、MRI、脑脊液 (CSF) 和血清学研究评估。
所有患者均经历了 7 次以上的 ON 复发,视力严重下降,对类固醇治疗仅有部分反应。视神经受到双侧影响,尽管单侧复发比同时双侧复发更常见。患者 MOG 血清阳性,但反复检测 AQP4 抗体均为阴性。OCT 显示视盘周围视网膜神经纤维层严重变薄。在 MRI 上,增强病变延伸超过视神经长度的一半。ON 发作期间的 CSF 分析正常。尽管进行了免疫抑制治疗,但 3 名患者中有 2 名的严重视力缺陷随时间逐渐累积。
MOG 血清阳性和 AQP4 血清阴性 rON 可能与侵袭性疾病过程和不良功能及结构结局相关。与之前的报道不同,视网膜和视神经损伤的严重程度和模式与常见的 AQP4 阳性 rON 表型非常相似,但不符合 NMOSD 的当前诊断标准。