神经结节病脊髓炎的多中心回顾性队列研究:当前观察和未来方向。

IF 3.9 2区 医学 Q1 CLINICAL NEUROLOGY
Giovanna S Manzano, Denis Balaban, Yihan Zhang, Brian Healy, Bart K Chwalisz, Michael Levy, Nagagopal Venna, Barney J Stern, Carlos A Pardo, Paula Barreras, Nicole Bou Rjeily, Eoin P Flanagan, Vyanka Redenbaugh, Allen J Aksamit, Spencer Hutto, Max Herman, Sally El Sammak, Elsa C Rodriguez, Laura Snider, Hannah Rains, Mayra Montalvo, Torge Rempe, Sergi Martinez Ramirez, Lucas Horta, Stacey Clardy, Jennifer Lord, Tracey A Cho, Lama Abdel Wahed, Joseph R Berger, Rohini D Samudralwar, Noellie Rivera Torres, David B Clifford, Steven Richard Dunham, Masoud Majed, Aram Zabeti, Samuel Marcucci, Yang Mao-Draayer, Jon Doty, Paunel B Agyei, Shamik Bhattacharyya
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引用次数: 0

摘要

目的:神经结节病脊髓炎的最佳治疗方法尚不确定。我们描述了事件神经结节病脊髓炎,并通过MRI和临床量表评估治疗反应。方法:回顾性分析13个学术医疗中心发生的可能或确定的成人神经结节病脊髓炎。对病例进行初步治疗分析。主要结果是治疗后6个月的T1造影后钆增强分辨率。次要观察指标为改良Rankin量表(mRS)和扩展残疾状态量表(EDSS)自最低点至最终随访时的变化。结果:共发现202例患者(中位诊断年龄:47岁(IQR 39-55);男:女:1.3:1)。中位最低点mRS和EDSS分别为2 (IQR 2-3)和4 (IQR 2.5-6)。在初始治疗时,129例(63.9%)接受延长糖皮质激素≥4周(A1组),36例(17.8%)接受糖皮质激素1组,21例(10.4%)接受糖皮质激素加结节病定向免疫抑制剂(E), 16例(7.9%)接受糖皮质激素加非结节病定向药物(F)。167例影像充足的患者,6个月时对比增强分辨率无显著差异(A1 27/106 (25.5%), B1 9/28 (32.1%), E 5/19 (26.3%), F 5/14 (35.7%);费雪精确p = 0.76)。中位随访46.5个月(IQR 18-91.3)后,各治疗组mRS和EDSS的变化无显著差异(Kruskal-Wallis p = 0.69和0.63)。解释:不同的初始免疫抑制策略与6个月或临床尺度的MRI对比增强分辨率无关(mRS, EDSS)。然而,由于回顾性设计、不平衡的队列、MRI二元结果的不敏感性和神经结节病治疗反应的可用临床量表,结论受到限制。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
A Multi-Center Retrospective Cohort Study of Neurosarcoidosis Myelitis: Current Observations and Future Directions.

Objective: The optimal treatment for neurosarcoidosis myelitis is uncertain. We characterize incident neurosarcoidosis myelitis and assess treatment response by MRI and clinical scales.

Methods: Incident probable or definite neurosarcoidosis myelitis in adults was retrospectively identified from 13 academic medical centers. Cases were analyzed by initial treatment. The primary outcome was T1 post-contrast gadolinium enhancement resolution at 6 months post-treatment. Secondary outcomes were changes in modified Rankin scale (mRS) and Expanded Disability Status Scale (EDSS) from nadir to final follow-up.

Results: Two hundred two patients were identified (median diagnosis age: 47 years (IQR 39-55); male: female 1.3:1). Median nadir mRS and EDSS were 2 (IQR 2-3) and 4 (IQR 2.5-6). At initial treatment, 129 (63.9%) received prolonged corticosteroids ≥ 4 weeks (group A1), 36 (17.8%) received corticosteroids < 4 weeks (B1), 21 (10.4%) received corticosteroids plus sarcoidosis-directed immunosuppressant (E), and 16 (7.9%) received corticosteroids plus non-sarcoidosis-directed agents (F). In 167 cases with sufficient imaging, there were no significant differences in contrast enhancement resolution at 6 months (A1 27/106 (25.5%), B1 9/28 (32.1%), E 5/19 (26.3%), F 5/14 (35.7%); Fisher's exact p = 0.76). There were no significant differences in changes in mRS or EDSS among treatment groups (Kruskal-Wallis p = 0.69 and 0.63, respectively) after median follow-up of 46.5 months (IQR 18-91.3).

Interpretation: Different initial immunosuppression strategies did not correlate with MRI contrast enhancement resolution at 6 months or clinical scales (mRS, EDSS). However, conclusions are limited by retrospective design, imbalanced cohorts, and insensitivity of binary MRI outcomes and available clinical scales for treatment response in neurosarcoidosis.

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来源期刊
Annals of Clinical and Translational Neurology
Annals of Clinical and Translational Neurology Medicine-Neurology (clinical)
CiteScore
9.10
自引率
1.90%
发文量
218
审稿时长
8 weeks
期刊介绍: Annals of Clinical and Translational Neurology is a peer-reviewed journal for rapid dissemination of high-quality research related to all areas of neurology. The journal publishes original research and scholarly reviews focused on the mechanisms and treatments of diseases of the nervous system; high-impact topics in neurologic education; and other topics of interest to the clinical neuroscience community.
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