From the Departments of Neurology (E.A.G., P.K., B.L.B.), Radiology (L.B.), Children's Hospital of Philadelphia, PA; Department of Neurology (A.B.-O.), University of Pennsylvania, PA; Division of Neurology (E.A.Y.), The Hospital for Sick Children, Toronto, Canada; McConnell Brain Imaging Centre (D.L.A.), Montreal Neurological Institute, Montreal, Canada; Department of Neurology (S.N.), McGill University, Montreal, Canada; Departments of Medicine and Community Health Sciences (R.A.M.), Max Rady College of Medicine, Rady Faculty of Health Sciences, University of Manitoba, Winnipeg, Canada; and Department of Medicine (G.F.), University of Ottawa, Ottawa Hospital Research Institute, Ottawa, Canada.
Neurol Neuroimmunol Neuroinflamm. 2024 Nov;11(6):e200319. doi: 10.1212/NXI.0000000000200319. Epub 2024 Oct 23.
Recent studies suggest that the choroid plexus (CP) may function as a site of access of inflammatory cells into the CNS in multiple sclerosis (MS). Pediatric-onset MS (POMS) is characterized by a high inflammatory burden, as evidenced by a high relapse rate and volume of T2 lesions, making patients with POMS an informative population to evaluate choroid plexus volume (CPV). The objectives of the study were (1) to evaluate CPV at symptom onset in participants with POMS compared with healthy controls (HCs); (2) to evaluate changes in CPV in the first year of disease in participants with POMS; and (3) to evaluate associations between CPV, brain volumes, relapse activity, and disability in participants with POMS.
Baseline 1.5T MRI scans were acquired from 23 participants with POMS and 23 age-matched and sex-matched HCs; 18 participants with POMS also had 12-month follow-up MRI scans. The CP of the lateral ventricles was segmented manually. CP and brain structure volumes were normalized for total intracranial volume. The number of relapses, T2 and gadolinium-enhancing T1 lesion counts, and Expanded Disability Status Scale (EDSS) scores at 12 months were also analyzed. Baseline CPVs were compared between groups using the Wilcoxon exact test, and CPV change from baseline to 12 months in participants with POMS was compared using the Wilcoxon signed-rank test. The relationship between CPV and brain volumetric measures, T2 lesion volumes, lesion count, number of relapses, and EDSS scores was assessed through Spearman correlation.
The median normalized CPV was 1.51 × 10 (interquartile range [IQR]: 1.32-1.76) in POMS baseline scans and 1.21 × 10 (IQR: 1.1-1.47) in HC scans ( = 0.001). CPV did not significantly change at 12 months in the 18 participants with POMS with follow-up scans ( = 0.352). CPV in participants with POMS and HCs correlated with lateral ventricular volume ( = 0.012 for both groups) but did not correlate with brain and T2 lesion volumes or lesion count at baseline, nor with relapse activity or EDSS scores at 12 months in participants with POMS.
CPV measured at baseline is greater in participants with POMS than in HCs. Baseline CPV did not predict higher disease activity or worse neurologic outcomes over 1 year. While higher CPV may be an early feature of inflammation in MS, its strong correlation with ventricular volumes could also reflect enlargement secondary to the mechanical attachment of CP to the ventricular wall.
最近的研究表明，脉络丛（CP）可能是多发性硬化症（MS）中炎症细胞进入中枢神经系统的途径之一。儿科发病的多发性硬化症（POMS）的炎症负担较高，这表现在较高的复发率和 T2 病变体积上，这使得 POMS 患者成为评估脉络丛体积（CPV）的一个有意义的人群。本研究的目的是：（1）比较 POMS 患者在症状发作时与健康对照组（HCs）的 CPV；（2）评估 POMS 患者在疾病的第一年 CPV 的变化；（3）评估 POMS 患者的 CPV 与脑容量、复发活动和残疾之间的相关性。
对 23 名 POMS 患者和 23 名年龄和性别匹配的 HCs 进行了基线 1.5T MRI 扫描；18 名 POMS 患者还进行了 12 个月的随访 MRI 扫描。通过手动对侧脑室 CP 进行分割。将 CP 和脑结构体积标准化为总颅内体积。还分析了 12 个月时的复发次数、T2 和钆增强 T1 病变计数和扩展残疾状态量表（EDSS）评分。使用 Wilcoxon 精确检验比较组间 CPV 的差异，使用 Wilcoxon 符号秩检验比较 POMS 患者基线到 12 个月 CPV 的变化。通过 Spearman 相关分析评估 CPV 与脑容积测量、T2 病变体积、病变计数、复发次数和 EDSS 评分之间的关系。
在 POMS 基线扫描中，中位数标准化 CPV 为 1.51×10（四分位距[IQR]：1.32-1.76），在 HCs 扫描中为 1.21×10（IQR：1.1-1.47）（ = 0.001）。在有随访扫描的 18 名 POMS 患者中，12 个月时 CPV 没有显著变化（ = 0.352）。POMS 患者和 HCs 的 CPV 与侧脑室容积相关（两组均为 = 0.012），但与基线时的脑和 T2 病变体积或病变计数不相关，也与 POMS 患者 12 个月时的复发活动或 EDSS 评分不相关。
与 HCs 相比，POMS 患者的 CPV 在基线时更高。基线 CPV 不能预测 1 年内更高的疾病活动或更差的神经学结局。虽然较高的 CPV 可能是 MS 中炎症的早期特征，但它与脑室容积的强烈相关性也可能反映了 CP 与脑室壁机械附着引起的扩大。
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