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度普利尤单抗诱发的嗜酸性肉芽肿性多血管炎并发周围神经性疼痛:一例报告及文献综述

Dupilumab-induced Eosinophilic Granulomatosis with Polyangiitis Complicated by Peripheral Neuropathic Pain: a Case Report and Literature Review.

作者信息

Wu Jiajun, Li Linlin, Ten Weidong, Wang Yuchen, Liu Ruiqi, Hu Binbin, Tan Jun, Dong Feilin, Shi Kaiwen, Zhang Haibo, Su Lizhong, Hu Weiming

机构信息

Otolaryngology & Head and Neck Center, Cancer Center, Department of Otolaryngology, Zhejiang Provincial People's Hospital (Affiliated People's Hospital), Hangzhou Medical College, Hangzhou, 310014, Zhejiang, China.

Zhejiang Provincial Clinical Research Center for Head & Neck Cancer, Hangzhou, 310014, China.

出版信息

J Clin Immunol. 2025 Jul 24;45(1):114. doi: 10.1007/s10875-025-01914-x.

DOI:10.1007/s10875-025-01914-x
PMID:40705100
Abstract

PURPOSE

Eosinophilic Granulomatosis with Polyangiitis (EGPA) is a rare vasculitis characterized by increased eosinophils in human tissues and peripheral blood. In this case, we present a 53-year-old female patient with EGPA. By this case and literature review, we want to explain the early manifestations, diagnosis, and management of EGPA, which will help clinicians to understand the disease and attach importance to the possibility of dupilumab-induced EGPA, to improve the early diagnosis rate of EGPA, and reduce misdiagnosis and missed diagnosis.

METHODS

The diagnostic criteria for EGPA established by the American Rheumatology Association (ACR) in 2022 were used; these criteria encompass clinical presentation, laboratory tests, and pathological biopsy. In addition, we conducted a comprehensive literature review on this case.

RESULT

We present a 53-year-old female patient who developed severe peripheral neuropathic pain after the administration of dupilumab for the treatment of refractory asthma and sinusitis. The patient's symptoms, laboratory examination findings, and nasopharyngeal biopsy pathology results collectively support the diagnosis of EGPA. When dupilumab was converted to mepolizumab combined with glucocorticoid, her peripheral neuropathic pain and asthma symptoms were dramatically relieved. Our literature review also provides a detailed discussion on the relationship between Dupilumab and EGPA.

CONCLUSION

We present a case of EGPA with peripheral neuropathic pain induced by Dupilumab, and mepolizumab has a good therapeutic effect on this patient. Our literature review shows that although dupilumab is effective in treating eosinophilic airway inflammatory diseases, clinicians must pay attention to the possibility of dupilumab inducing or aggravating EGPAs.

摘要

目的

嗜酸性肉芽肿性多血管炎(EGPA)是一种罕见的血管炎,其特征是人体组织和外周血中嗜酸性粒细胞增多。在此病例中,我们报告一名53岁的EGPA女性患者。通过该病例及文献回顾,我们旨在阐述EGPA的早期表现、诊断及治疗,这将有助于临床医生了解该疾病,并重视度普利尤单抗诱导EGPA的可能性,提高EGPA的早期诊断率,减少误诊和漏诊。

方法

采用美国风湿病学会(ACR)2022年制定的EGPA诊断标准,该标准涵盖临床表现、实验室检查及病理活检。此外,我们对该病例进行了全面的文献回顾。

结果

我们报告一名53岁女性患者,在使用度普利尤单抗治疗难治性哮喘和鼻窦炎后出现严重的外周神经病理性疼痛。患者的症状、实验室检查结果及鼻咽活检病理结果共同支持EGPA的诊断。当将度普利尤单抗换为美泊利单抗联合糖皮质激素治疗后,其外周神经病理性疼痛和哮喘症状明显缓解。我们的文献回顾还对度普利尤单抗与EGPA之间的关系进行了详细讨论。

结论

我们报告了一例由度普利尤单抗诱发外周神经病理性疼痛的EGPA病例,美泊利单抗对该患者有良好的治疗效果。我们的文献回顾表明,尽管度普利尤单抗在治疗嗜酸性气道炎症性疾病方面有效,但临床医生必须注意度普利尤单抗诱发或加重EGPA的可能性。

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本文引用的文献

1
Characteristics of Severe Asthma Clinic Patients With Eosinophilic Granulomatosis With Polyangiitis.伴有嗜酸性粒细胞性肉芽肿性多血管炎的重度哮喘门诊患者的特征
J Allergy Clin Immunol Pract. 2025 Feb;13(2):361-368.e2. doi: 10.1016/j.jaip.2024.10.013. Epub 2024 Oct 16.
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Mesenchymal stem cell therapy in eosinophilic granulomatosis with polyangiitis-related lower limb gangrene: a case report.间充质干细胞治疗嗜酸性肉芽肿伴多血管炎相关下肢坏疽:病例报告。
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Exploring the immunopathology of type 2 inflammatory airway diseases.
探索 2 型炎症性气道疾病的免疫病理学。
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Cardiac involvement in eosinophilic granulomatosis with polyangiitis: acute eosinophilic myocarditis and chronic inflammatory cardiomyopathy.嗜酸性肉芽肿性多血管炎的心脏受累:急性嗜酸性粒细胞性心肌炎和慢性炎症性心肌病。
Rheumatology (Oxford). 2025 Feb 1;64(2):722-731. doi: 10.1093/rheumatology/keae085.
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Combination of monoclonal antibodies targeting type 2 inflammation for severe asthma and eosinophilic granulomatosis with polyangiitis.针对 2 型炎症的单克隆抗体联合治疗用于严重哮喘和嗜酸性肉芽肿性多血管炎。
Autoimmun Rev. 2024 Mar;23(3):103503. doi: 10.1016/j.autrev.2023.103503. Epub 2023 Dec 13.
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A case of dupilumab combination therapy for exacerbation of atopic dermatitis in a patient with eosinophilic granulomatosis with polyangiitis treated with mepolizumab.一例美泊利珠单抗治疗嗜酸性肉芽肿性多血管炎患者中重度特应性皮炎恶化后接受度普利尤单抗联合治疗的病例。
Mod Rheumatol Case Rep. 2023 Dec 29;8(1):159-162. doi: 10.1093/mrcr/rxad059.
7
Eosinophilic granulomatosis with polyangiitis after treatment with dupilumab.使用度普利尤单抗治疗后发生的嗜酸性肉芽肿性多血管炎。
J Allergy Clin Immunol Glob. 2022 Apr 30;1(3):180-182. doi: 10.1016/j.jacig.2022.03.006. eCollection 2022 Aug.
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Eosinophilic Granulomatosis with Polyangiitis: Latest Findings and Updated Treatment Recommendations.嗜酸性肉芽肿性多血管炎:最新研究发现与更新的治疗建议
J Clin Med. 2023 Sep 15;12(18):5996. doi: 10.3390/jcm12185996.
9
Dupilumab for relapsing or refractory sinonasal and/or asthma manifestations in eosinophilic granulomatosis with polyangiitis: a European retrospective study.度普利尤单抗用于治疗嗜酸性肉芽肿性多血管炎的复发性或难治性鼻窦和/或哮喘表现:一项欧洲回顾性研究。
Ann Rheum Dis. 2023 Dec;82(12):1587-1593. doi: 10.1136/ard-2023-224756. Epub 2023 Sep 21.
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Successful switching from mepolizumab to dupilumab in a patient with EGPA in remission phase and persistent nasal polyposis.一名处于缓解期且患有持续性鼻息肉的嗜酸性肉芽肿性多血管炎(EGPA)患者成功从美泊利单抗转换为度普利尤单抗治疗。
Rheumatology (Oxford). 2024 Mar 1;63(3):e96-e98. doi: 10.1093/rheumatology/kead479.