[Siltuximab as a salvage therapy in multi-refractory adult-onset Still's disease: A case series].

La Revue de medecine interne Pub Date : 2025-05-23 DOI:10.1016/j.revmed.2025.05.002

Julie Moreau, Amélie Leurs, Emmanuel Ledoult, Sébastien Sanges, Pierre Quartier, Thibault Comont, David Launay, Eric Hachulla, Louis Terriou

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Abstract

Introduction: Adult-onset Still's disease (AOSD) is a systemic auto-inflammatory disorder characterized by a cytokine storm. Four major cytokines can be specifically targeted in severe, refractory or corticosteroid-dependent forms: interleukin 1 (IL-1), interleukin 6 (IL-6), interleukin 18 (IL-18), and Tumor necrosis factor alpha (TNFa). According to the French national protocol for adult-onset Still's disease, anti-IL-6 agents are the second-line of treatment after corticosteroids. We report here on the efficacy and safety of siltuximab prescribed off-label in patients with multi-refractory forms.

Observations/results: These 4 patients, aged 24 to 74 years old, had polycyclic and inflammatory AOSD. The introduction of siltuximab was proposed after failure or poor tolerance of several lines of treatment. A partial or complete clinico-biological response was observed in all 4 patients.

Conclusion: Siltuximab is a possible salvage treatment for multi-refractory AOSD.

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[西妥昔单抗作为多难治性成人发病斯蒂尔氏病的补救性治疗：一个病例系列]。

成人发病斯蒂尔氏病（AOSD）是一种以细胞因子风暴为特征的全身性自身炎症性疾病。有四种主要的细胞因子可以以严重的、难治性的或皮质类固醇依赖的形式特异性靶向：白细胞介素1 （IL-1）、白细胞介素6 （IL-6）、白细胞介素18 （IL-18）和肿瘤坏死因子α (TNFa)。根据法国成人发病斯蒂尔氏病的国家方案，抗il -6药物是皮质类固醇之后的二线治疗。我们在此报告西妥昔单抗在标签外治疗多重难治性患者的有效性和安全性。观察/结果：4例患者，年龄24 ~ 74岁，均为多环性炎性AOSD。引入西妥昔单抗是在几种治疗方法失败或耐受性差后提出的。所有4例患者均有部分或完全的临床生物学反应。结论：西妥昔单抗是治疗多重难治性AOSD的一种可能的挽救性治疗方法。

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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La Revue de medecine interne

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