糖皮质激素和肿瘤坏死因子-α抑制剂治疗口腔和生殖器溃疡伴炎性软骨综合征患者的长期喉部表现

IF 2.2 Q3 RHEUMATOLOGY
Journal of Rheumatic Diseases Pub Date : 2025-07-01 Epub Date: 2025-03-10 DOI:10.4078/jrd.2024.0144
Tomohiro Suzuki, Toshihiko Komai, Misaki Koyama, Kento Koda, Rumi Ueha, Hirofumi Shoda, Keishi Fujio
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引用次数: 0

摘要

口腔和生殖器溃疡伴炎性软骨(MAGIC)综合征是一种罕见的疾病,以复发性多软骨炎(RP)和behet病(BD)为特征,可累及多器官,包括炎症和软骨组织破坏。本报告描述了一名MAGIC综合征患者,他对长期喉部炎症的免疫抑制治疗有反应,并首次描述了一名人类白细胞抗原(HLA)-A26阳性的MAGIC综合征患者。这里我们提出一个49岁的男性复发性口腔和生殖器溃疡,声音嘶哑,吞咽困难。喉镜检查显示双侧声带不动,磁共振增强成像(MRI)显示环状软骨、杓状软骨和耳廓软骨有炎症。根据RP和BD标准,患者被诊断为MAGIC综合征。大剂量皮质类固醇和阿达木单抗生物类似药治疗可显著改善患者声音嘶哑和软骨炎症的MRI表现。本病例强调了免疫抑制治疗对MAGIC综合征长期喉部累及的有效应用,潜在地避免了气管切开术等侵入性干预的需要。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Resolution of long-lasting laryngeal manifestations using glucocorticoids and tumor necrosis factor-α inhibitor therapy in a patient with mouth and genital ulcers with inflamed cartilage syndrome.

Mouth and genital ulcers with inflamed cartilage (MAGIC) syndrome is a rare disorder characterized by features of both relapsing polychondritis (RP) and Behçet's disease (BD), with multi-organ involvement, including inflammation and destruction of cartilaginous tissues. This report describes a patient with MAGIC syndrome who responded to immunosuppressive therapy for long-lasting laryngeal inflammation and provides the first description of a patient with MAGIC syndrome positive for human leukocyte antigen (HLA)-A26. Here we present a 49-year-old male with recurrent oral and genital ulcers, hoarseness, and swallowing difficulties. Laryngoscopy showed bilateral vocal fold immobility, and contrast-enhanced magnetic resonance imaging (MRI) demonstrated inflammation in the cricoid, arytenoid, and auricular cartilages. The patient was diagnosed with MAGIC syndrome based on the presence of both RP and BD criteria. Treatment with high-dose corticosteroids and adalimumab biosimilar resulted in significant improvement in hoarseness and MRI findings of cartilage inflammation. This case highlights the effective use of immunosuppressive therapy for managing long-lasting laryngeal involvement in MAGIC syndrome, potentially avoiding the need for invasive interventions such as tracheostomy.

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来源期刊
CiteScore
2.30
自引率
5.00%
发文量
39
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