Clinical Characteristics and Visual Outcomes of Optic Neuritis in Pediatric Myelin Oligodendrocyte Glycoprotein Antibody-Associated Disease.

IF 2.1 4区 医学 Q3 CLINICAL NEUROLOGY
Takhmina Abdul, Arina Nisanova, Anjalee Chopra, Ryan Bugsch, Akash Virupakshaiah, Naga Pradyumna Kothapalli, Ketaki Milind Panse, Emmanuelle Waubant, Nailyn Rasool, Shannon J Beres, Yin Allison Liu
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引用次数: 0

Abstract

Background: Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is a neuro-immunological disorder that can cause optic neuritis (ON) and significant visual impairment. Our objective was to investigate clinical characteristics and visual outcomes in children with MOGAD-ON.

Methods: Retrospective cohort study of MOG-IgG-positive children (onset age 18 years and younger) meeting 2023 international MOGAD panel criteria who presented with ON and received neuro-ophthalmic care between 2003 and 2023 at 3 academic centers in Northern California was conducted. We captured demographic and clinical characteristics, disease course, and presenting MOG-IgG titers through chart review. Ophthalmic metrics, including logarithmic minimum angle of resolution best-corrected visual acuity (logMAR BCVA), retinal nerve fiber layer (RNFL) and ganglion cell-inner plexiform layer thicknesses, and automated visual field mean deviation (AVF MD) at diagnosis, 1-month, 6-month, 1-year, and final (2+ years) visits, were obtained.

Results: This study included 49 ON-affected eyes of 29 patients (20 with bilateral involvement), with 19 (66%) being female. The median age at first diagnosis was 9 years (range 1-18). The median follow-up was 40 months (0-119). Age younger than 8 years at MOGAD onset was predictive of recurrent ON (β1=-1.61, P = 0.03). LogMAR BCVA improved from 1.4 at diagnosis to 0.2 at 1 month and 0.07 at the final visit (P < 0.0001). In total, 29 eyes (59%) of 22 patients (76%) achieved complete visual recovery (BCVA LogMAR 0). RNFL thickness decreased from mean 130 µm at diagnosis to 6 months (65 µm, P < 0.05). Average AVF MD improved from -12.8 dB at diagnosis to -5.4 dB at the final visit (P = 0.02). Higher presenting MOG-IgG titers were associated with worse presenting visual fields (r = -0.96, P = 0.04).

Conclusions: Children with MOGAD-associated ON often return to normal visual acuity despite significant optic atrophy. Most RNFL thinning occurs in the first 6 months but continues beyond 2 years after MOGAD onset. Younger children were at greater risk of ON relapse. A prospective longitudinal study on pediatric MOGAD-ON is warranted to validate these findings and identify biomarkers for visual outcome prediction.

小儿髓鞘少突胶质细胞糖蛋白抗体相关疾病视神经炎的临床特点和视力结果
背景:髓鞘少突胶质细胞糖蛋白抗体相关疾病(MOGAD)是一种神经免疫疾病,可导致视神经炎(ON)和严重的视力损害。我们的目的是调查加迪加- on患儿的临床特征和视力结果。方法:回顾性队列研究的mog - igg阳性儿童(发病年龄18岁及以下)符合2023年国际MOGAD小组标准,2003年至2023年期间在北加州3个学术中心出现ON并接受神经眼科护理。我们捕获了人口统计学和临床特征、病程,并通过图表回顾呈现了MOG-IgG滴度。获得眼科指标,包括诊断时、1个月、6个月、1年和最后(2+年)就诊时的对数最小分辨率最佳矫正视力角(logMAR BCVA)、视网膜神经纤维层(RNFL)和神经节细胞-内丛状层厚度,以及自动视野平均偏差(AVF MD)。结果:本研究纳入29例患者的49只眼(20例双侧受累),其中19例(66%)为女性。首次诊断的中位年龄为9岁(范围1-18岁)。中位随访时间为40个月(0-119)。MOGAD发病年龄小于8岁可预测ON复发(β1=-1.61, P = 0.03)。LogMAR BCVA从诊断时的1.4提高到1个月时的0.2和最后一次就诊时的0.07 (P < 0.0001)。22例患者(76%)中有29只眼(59%)实现了完全视力恢复(BCVA LogMAR 0)。RNFL厚度从诊断时的平均130µm下降到6个月(65µm, P < 0.05)。平均AVF MD从诊断时的-12.8 dB改善到最后一次就诊时的-5.4 dB (P = 0.02)。呈现MOG-IgG滴度越高,呈现视野越差(r = -0.96, P = 0.04)。结论:患有摩加迪沙相关性ON的儿童尽管有明显的视神经萎缩,但通常能恢复正常的视力。大多数RNFL变薄发生在MOGAD发病后的前6个月,但持续超过2年。年龄较小的儿童复发的风险更大。一项针对儿童摩加迪松的前瞻性纵向研究有必要验证这些发现,并确定用于视觉结果预测的生物标志物。
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来源期刊
Journal of Neuro-Ophthalmology
Journal of Neuro-Ophthalmology 医学-临床神经学
CiteScore
2.80
自引率
13.80%
发文量
593
审稿时长
6-12 weeks
期刊介绍: The Journal of Neuro-Ophthalmology (JNO) is the official journal of the North American Neuro-Ophthalmology Society (NANOS). It is a quarterly, peer-reviewed journal that publishes original and commissioned articles related to neuro-ophthalmology.
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