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白细胞介素-17抑制剂司库奇尤单抗使复发性多软骨炎病情缓解:一例报告

IL-17 Inhibitor Secukinumab Achieves Remission in Relapsing Polychondritis: A Case Report.

作者信息

Neycheva Stefka, Kamburova Adriana

机构信息

Department of Rheumatology, Military Medical Academy, Sofia, Bulgaria.

出版信息

Am J Case Rep. 2025 Mar 20;26:e946916. doi: 10.12659/AJCR.946916.

DOI:10.12659/AJCR.946916
PMID:40108787
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11932544/
Abstract

BACKGROUND Relapsing polychondritis is a rare autoimmune disease of unknown etiology. There are no defined protocols for treatment, and its management depends on the physician's knowledge and empirical experience. Various reports often highlight the insufficient efficacy of conventional disease-modifying anti-rheumatic drugs, and report controversial results associated with the use of different biologic agents. CASE REPORT The patient was a 32-year-old woman, previously diagnosed with ankylosing spondylitis. Four years after the initial diagnosis and following her second delivery, the patient presented with a refractory and aggressive relapsing polychondritis, which posed a significant challenge for treatment. Two months prior to the onset of the chondritis, the patient exhibited high disease activity of ankylosing spondylitis, with ASDAS-CRP 3.5 and BASDAI 6.0. After treatment failure using a combination of an anti-TNFalpha inhibitor and methotrexate, we achieved remission of both co-existing autoimmune diseases using an IL-17 inhibitor (secukinumab). CONCLUSIONS Further investigations are needed to better understand the pathogenesis of this rare condition. The existence of various reports on the controversial effects of treatment with different biologic agents, including IL-17 antagonists, raises several questions, including whether the onset of relapsing polychondritis is a side effect of the use of biologics or if it is an autoimmune disease that occurs independently of the treatment. This is the first time that the use of an anti-IL-17 agent is reported to have successfully suppressed an aggressive, progressive form of this disease, without any symptom-free period before the initiation of secukinumab.

摘要

背景 复发性多软骨炎是一种病因不明的罕见自身免疫性疾病。目前尚无明确的治疗方案,其管理依赖于医生的知识和经验。各种报告经常强调传统抗风湿药物疗效不足,并报告了使用不同生物制剂的争议性结果。病例报告 该患者为一名32岁女性,既往诊断为强直性脊柱炎。初次诊断四年后且在第二次分娩后,患者出现难治性且侵袭性的复发性多软骨炎,这对治疗构成了重大挑战。在软骨炎发作前两个月,患者强直性脊柱炎疾病活动度较高,ASDAS-CRP为3.5,BASDAI为6.0。在使用抗TNFα抑制剂和甲氨蝶呤联合治疗失败后,我们使用IL-17抑制剂(司库奇尤单抗)使两种并存的自身免疫性疾病均得到缓解。结论 需要进一步研究以更好地了解这种罕见疾病的发病机制。关于不同生物制剂(包括IL-17拮抗剂)治疗的争议性效果的各种报告引发了几个问题,包括复发性多软骨炎的发作是否是使用生物制剂的副作用,或者它是否是一种独立于治疗发生的自身免疫性疾病。这是首次报道使用抗IL-17药物成功抑制了这种疾病的侵袭性、进行性形式,且在开始使用司库奇尤单抗之前没有无症状期。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d200/11932544/0b018410787e/amjcaserep-26-e946916-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d200/11932544/265223b769f4/amjcaserep-26-e946916-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d200/11932544/3dff501dd3e3/amjcaserep-26-e946916-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d200/11932544/0b018410787e/amjcaserep-26-e946916-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d200/11932544/265223b769f4/amjcaserep-26-e946916-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d200/11932544/3dff501dd3e3/amjcaserep-26-e946916-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d200/11932544/0b018410787e/amjcaserep-26-e946916-g003.jpg

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本文引用的文献

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Relapsing polychondritis: clinical updates and new differential diagnoses.复发性多软骨炎:临床更新及新的鉴别诊断。
Nat Rev Rheumatol. 2024 Jun;20(6):347-360. doi: 10.1038/s41584-024-01113-9. Epub 2024 May 2.
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Autoimmunity and Autoinflammation: Relapsing Polychondritis and VEXAS Syndrome Challenge.自身免疫与自身炎症:复发性多软骨炎与 VEXAS 综合征的挑战。
Int J Mol Sci. 2024 Feb 13;25(4):2261. doi: 10.3390/ijms25042261.
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Relapsing Polychondritis in a Patient with Ankylosing Spondylitis under Two Different Tumor Necrosis Factor Alpha Inhibitors Treatment.
两种不同肿瘤坏死因子 α 抑制剂治疗下强直性脊柱炎伴发复发性多软骨炎 1 例
Clin Ter. 2022 Apr 4;173(2):97-98. doi: 10.7417/CT.2022.2400.
4
A Rare Case of Ankylosing Spondylitis Coexisting with Relapsing Polychondritis, Antiphospholipid Syndrome, and Myelodysplastic Syndrome.一例强直性脊柱炎合并复发性多软骨炎、抗磷脂综合征和骨髓增生异常综合征的罕见病例。
Intern Med. 2022 Aug 1;61(15):2367-2371. doi: 10.2169/internalmedicine.8784-21. Epub 2022 Jan 13.
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Assessment of TNF-α inhibitors in airway involvement of relapsing polychondritis: A systematic review.肿瘤坏死因子-α抑制剂在复发性多软骨炎气道受累中的评估:一项系统评价。
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Relapsing Polychondritis following Treatment with Secukinumab for Ankylosing Spondylitis: Case Report and Review of the Literature.司库奇尤单抗治疗强直性脊柱炎后发生复发性多软骨炎:病例报告及文献复习
Case Rep Rheumatol. 2018 Jul 2;2018:6760806. doi: 10.1155/2018/6760806. eCollection 2018.
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Refractory relapsing polychondritis: challenges and solutions.难治性复发性多软骨炎:挑战与解决方案
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