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托珠单抗治疗对静脉用免疫球蛋白治疗反应不佳的高度复发性 MO-GAD 患者的疗效:病例系列研究。

Efficacy of tocilizumab in highly relapsing MOGAD with an inadequate response to intravenous immunoglobulin therapy: A case series.

机构信息

Department of Neurology, Research Institute and Hospital of National Cancer Center, Goyang, Republic of Korea.

Department of Neurology, Research Institute and Hospital of National Cancer Center, Goyang, Republic of Korea; Department of Neurology, Sanggye Paik Hospital, Inje University College of Medicine, Seoul, Republic of Korea.

出版信息

Mult Scler Relat Disord. 2024 Nov;91:105859. doi: 10.1016/j.msard.2024.105859. Epub 2024 Aug 30.

DOI:10.1016/j.msard.2024.105859
PMID:39236649
Abstract

BACKGROUND

Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is an inflammatory disease affecting the central nervous system that may require long-term immunotherapy in relapsing cases. While immunotherapies utilized in neuromyelitis optica spectrum disorder have shown varying efficacy in MOGAD, intravenous immunoglobulin G (IVIG) recently emerged as a promising treatment. Tocilizumab, a monoclonal antibody that targets the interleukin-6 (IL-6) receptor, has been reported to be effective in refractory MOGAD in several case studies, where tocilizumab was introduced primarily due to rituximab failure.

METHODS

This retrospective study was conducted in a single center and focused on MOGAD patients receiving tocilizumab therapy, who have shown limited response to various immunotherapies, including intravenous immunoglobulin G (IVIG) maintenance.

RESULTS

This study included four patients, three adults, and one child. They experienced a median of 9 attacks (range 6-9) throughout their disease course despite at least two immunotherapies. All patients transitioned to tocilizumab after experiencing a median of two relapses (range 1-3) while on IVIG maintenance for a median of 21.9 months (range 21.3-49.6 months). Following the monthly tocilizumab administration at a dose of 8g/kg, all patients remained relapse-free with a median follow-up duration of 25.0 months (range 9.8-51.3 months) without reported adverse events.

CONCLUSION

Targeting the IL-6 pathway appears to offer therapeutic benefits in highly relapsing MOGAD patients who poorly respond to IVIG maintenance therapy.

摘要

背景

髓鞘少突胶质细胞糖蛋白抗体相关疾病(MOGAD)是一种影响中枢神经系统的炎症性疾病,在复发病例中可能需要长期免疫治疗。虽然视神经脊髓炎谱系疾病中使用的免疫疗法在 MOGAD 中的疗效各不相同,但静脉注射免疫球蛋白 G(IVIG)最近成为一种有前途的治疗方法。托珠单抗是一种针对白细胞介素-6(IL-6)受体的单克隆抗体,在几项病例研究中被报道对难治性 MOGAD 有效,在这些病例中,托珠单抗主要是由于利妥昔单抗治疗失败而引入。

方法

本回顾性研究在一家单中心进行,重点关注接受托珠单抗治疗的 MOGAD 患者,这些患者对包括 IVIG 维持在内的各种免疫疗法反应有限。

结果

这项研究包括 4 名患者,3 名成人和 1 名儿童。尽管他们接受了至少两种免疫疗法,但在疾病过程中他们经历了中位数为 9 次发作(范围 6-9)。所有患者在接受 IVIG 维持治疗中位数为 21.9 个月(范围 21.3-49.6 个月)时经历了中位数为 2 次复发(范围 1-3)后,转为托珠单抗治疗。每月给予 8g/kg 的托珠单抗剂量后,所有患者均未复发,中位随访时间为 25.0 个月(范围 9.8-51.3 个月),无不良事件报告。

结论

靶向 IL-6 途径似乎为对 IVIG 维持治疗反应不佳的高度复发性 MOGAD 患者提供了治疗益处。

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