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阿仑单抗诱导的甲状腺眼病经单次低剂量利妥昔单抗治疗成功。

Alemtuzumab-induced thyroid eye disease successfully treated with a single low dose of rituximab.

机构信息

Department of Clinical Sciences and Community Health, University of Milan, Italy.

Endocrinology Unit, Graves' Orbitopathy Center, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.

出版信息

Eur Thyroid J. 2024 Apr 11;13(2). doi: 10.1530/ETJ-23-0236. Print 2024 Apr 1.

Abstract

INTRODUCTION

Secondary thyroid autoimmunity, especially Graves' disease (GD), frequently develops in patients with multiple sclerosis (MS) following alemtuzumab treatment (ALTZ; anti-CD52). Thyroid eye disease (TED) can also develop, and rituximab (RTX; anti-CD20) is a suitable treatment.

CASE PRESENTATION

A 37-year-old woman with MS developed steroid-resistant active moderate-to-severe TED 3 years after ALTZ, that successfully responded to a single 500 mg dose of i.v. RTX. Before RTX peripheral B-cells were low, and were totally depleted immediately after therapy. Follow-up analysis 4 years post ALTZ and 1 year post RTX showed persistent depletion of B cells, and reduction of T regulatory cells in both peripheral blood and thyroid tissue obtained at thyroidectomy.

CONCLUSION

RTX therapy successfully inactivated TED in a patient with low B-cell count derived from previous ALTZ treatment. B-cell depletion in both thyroid and peripheral blood was still present 1 year after RTX, indicating a likely cumulative effect of both treatments.

摘要

简介

在接受阿仑单抗(抗 CD52)治疗后,多发性硬化症(MS)患者常会继发甲状腺自身免疫,特别是格雷夫斯病(GD)。甲状腺眼病(TED)也可能会发生,利妥昔单抗(RTX;抗 CD20)是一种合适的治疗药物。

病例介绍

一名 37 岁女性,在接受阿仑单抗治疗 3 年后出现了激素耐药性活动期中重度 TED,单次静脉注射 500mg 利妥昔单抗后病情得到缓解。RTX 治疗前外周血 B 细胞计数较低,治疗后即刻完全耗竭。在 ALTZ 治疗后 4 年和 RTX 治疗后 1 年的随访分析中,发现 B 细胞在外周血和甲状腺组织中持续耗竭,甲状腺组织中 T 调节细胞也减少。

结论

RTX 治疗成功地使一位先前接受过阿仑单抗治疗、B 细胞计数较低的 TED 患者的病情得到缓解。RTX 治疗 1 年后,甲状腺和外周血中的 B 细胞耗竭仍然存在,表明两种治疗药物可能存在累积效应。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a8d5/11046353/f6a724d71ebc/ETJ-23-0236fig1.jpg

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