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

IF 3.5 2区医学 Q2 ENDOCRINOLOGY & METABOLISM

European Thyroid Journal Pub Date : 2024-04-11 Print Date: 2024-04-01 DOI:10.1530/ETJ-23-0236

Ilaria Muller, Sara Maioli, Mirco Armenti, Laura Porcaro, Nicola Currò, Elisabetta Iofrida, Lorenzo Pignataro, Jacopo Manso, Caterina Mian, Jens Geginat, Mario Salvi

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引用次数: 0

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.

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使用单次低剂量利妥昔单抗成功治疗阿来珠单抗诱发的甲状腺眼病。

背景多发性硬化症（MS）患者在接受阿仑妥珠单抗（ALTZ；抗CD52）治疗后，经常会出现继发性甲状腺自身免疫，尤其是巴塞杜氏病（GD）。甲状腺眼病（TED）也可能发生，而利妥昔单抗（RTX；抗 CD20）是一种合适的治疗方法。方法对一名同时接受 ALTZ 和 RTX 治疗的患者的血液和甲状腺衍生淋巴细胞进行免疫分型。结果一位37岁的女性多发性硬化症患者在接受ALTZ治疗三年后出现类固醇耐药的活动性中重度TED，并对单次500毫克剂量的静脉注射RTX治疗产生了成功的反应。RTX治疗前外周B细胞数量较少，治疗后立即完全耗尽。ALTZ治疗后四年和RTX治疗后一年的随访分析显示，B细胞持续耗竭，外周血和甲状腺切除术中获得的甲状腺组织中的T调节细胞相对减少。结论 RTX疗法成功灭活了一名因之前接受ALTZ治疗而导致B细胞数量较低的患者的TED。RTX治疗一年后，甲状腺和外周血中的B细胞仍在减少，这可能是两种治疗的累积效应。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

European Thyroid Journal Medicine-Endocrinology, Diabetes and Metabolism

CiteScore

6.70

自引率

2.10%

发文量

156

期刊介绍： The ''European Thyroid Journal'' publishes papers reporting original research in basic, translational and clinical thyroidology. Original contributions cover all aspects of the field, from molecular and cellular biology to immunology and biochemistry, from physiology to pathology, and from pediatric to adult thyroid diseases with a special focus on thyroid cancer. Readers also benefit from reviews by noted experts, which highlight especially active areas of current research. The journal will further publish formal guidelines in the field, produced and endorsed by the European Thyroid Association.