Neuroimmunological Clinic, Institute of Pediatric Neurology, Schneider Children's Medical Center of Israel, Petah Tikva 4920235, Israel; Sackler School of Medicine, Tel Aviv University, Tel Aviv 6997801, Israel.
Department of Neurology, Rambam Health Care Campus, Haifa 3525408, Israel; Neuroimmunology Laboratory, Department of Neurology, Rambam Health Care Campus and Ruth and Bruce Rapaport Faculty of Medicine, Technion - Israel Institute of Technology, Haifa 3525408, Israel.
Mult Scler Relat Disord. 2023 Nov;79:105034. doi: 10.1016/j.msard.2023.105034. Epub 2023 Oct 1.
Distinguishing between MOG-associated disease (MOGAD) and multiple sclerosis (MS) presents a considerable challenge, as there are instances of overlapping clinical presentations. This complexity is further magnified in cases where patients concurrently exhibit both anti-myelin oligodendrocyte glycoprotein (anti-MOG) positivity and detectable oligoclonal bands (OCBs) This retrospective study investigates the clinical and imaging attributes of dual-positive patients, those with both anti-MOG positivity and OCBs, The study aims to show potential areas of overlap between multiple sclerosis (MS) and MOGAD.
Utilizing data gathered from three medical centers, we evaluated a cohort of 45 patients, stratifying them into two groups: those exclusively positive for anti-MOG antibodies and those displaying dual positivity. Our analysis encompassed a wide range of clinical and imaging parameters. The statistical techniques employed comprised Fisher's Exact Test along with Benjamini-Hochberg correction to ensure robustness of the findings.
The study involved 45 patients with anti-MOG antibodies; 30 exhibited isolated anti-MOG positivity without OCBs, while 15 were dual-positive. The first group's average age was 10±7 years, compared to 28±17 years in the double-positive group (p = 0.001). CSF analysis showed no significant differences in pleocytosis, protein levels, or opening pressure between the groups. In the exclusive anti-MOG positivity cohort, 9 out of 15 patients received IVIG treatment; a larger subgroup with dual positivity chose anti-CD20 treatment. Notably, papilledema incidence was higher in the single-positive group (p = 0.014). Optic nerve enhancement (p = 0.0038) and nerve thickening (p = 0.0017) were markedly elevated in the single-positive population, with a trend towards pre-chiasmatic lesions (p = 0.06). Double-positive cases exhibited more polyfocal presentation (p = 0.013) and higher attacks per case (p = 0.002, HR=10.2, 95 % CI: 2.19 to 49.23). The double-positive group had more brain lesions (p = 0.0063) but no significant distinctions in other aspects.
The results emphasize the challenges inherent in differentiating between MS and a more MOGAD. While the data suggest two plausible scenarios-either falling within the spectrum of MS or representing an intensified MOGAD-we recognize the need for stronger evidence to definitively classify these instances. This study underscores the imperative for thorough investigations to ascertain whether these cases align with the MS spectrum or denote an inflammatory variant of MOGAD.
MOG 相关性疾病（MOGAD）与多发性硬化症（MS）之间的鉴别极具挑战性，因为存在临床特征重叠的情况。在同时表现出抗髓鞘少突胶质细胞糖蛋白（anti-MOG）阳性和可检测到寡克隆带（OCBs）的病例中，这种复杂性进一步加剧。本回顾性研究调查了双重阳性患者（即同时存在抗-MOG 阳性和 OCBs 的患者）的临床和影像学特征。研究旨在展示 MS 和 MOGAD 之间的潜在重叠区域。
利用来自三个医学中心的数据，我们评估了一组 45 名患者，将他们分为两组：仅抗-MOG 抗体阳性组和双重阳性组。我们的分析涵盖了广泛的临床和影像学参数。采用 Fisher's Exact Test 与 Benjamini-Hochberg 校正相结合的统计学方法，以确保结果的稳健性。
研究共纳入 45 名抗-MOG 抗体阳性患者；30 名患者表现为单纯抗-MOG 阳性而无 OCBs，15 名患者为双重阳性。第一组的平均年龄为 10±7 岁，而双重阳性组为 28±17 岁（p=0.001）。CSF 分析显示两组之间细胞增多症、蛋白水平或开放压均无显著差异。在仅抗-MOG 阳性组中，15 名患者中有 9 名接受了 IVIG 治疗；而更大的双重阳性亚组选择了抗-CD20 治疗。值得注意的是，单阳性组的视乳头水肿发生率更高（p=0.014）。视神经增强（p=0.0038）和神经增粗（p=0.0017）在单阳性人群中显著升高，且视交叉前病变呈趋势（p=0.06）。双重阳性病例表现出更多的多灶性表现（p=0.013）和更高的每个病例发作次数（p=0.002，HR=10.2，95%CI：2.19 至 49.23）。双重阳性组的脑病变更多（p=0.0063），但其他方面无显著差异。
结果强调了在 MS 和更典型的 MOGAD 之间进行鉴别所固有的挑战。虽然数据表明存在两种可能的情况——要么属于 MS 谱，要么代表更强化的 MOGAD——但我们认识到需要更强有力的证据来明确分类这些情况。本研究强调了进行彻底调查的必要性，以确定这些病例是否与 MS 谱一致，还是表示 MOGAD 的炎症变体。
