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利妥昔单抗治疗难治性多发性肌炎合并吞咽困难和发音障碍患者的快速反应:一例报告

Prompt response to rituximab in a patient with resistant polymyositis with complications of dysphagia and dysphonia: a case report.

作者信息

Habes Yousef Mahmoud Nimer, Mohammad Sadeh Saif Ibraheem, Saada Layth Jamil Mohammad, Mali Ali Jamil Ali, Abdulrazzak Mohammed, Attawna Saed I Y

机构信息

Faculty of Medicine, Al-Quds University, Jerusalem.

Faculty of Medicine, University of Aleppo, Aleppo, Syria.

出版信息

Ann Med Surg (Lond). 2024 Aug 22;86(10):6227-6230. doi: 10.1097/MS9.0000000000002490. eCollection 2024 Oct.

Abstract

INTRODUCTION AND IMPORTANCE

Polymyositis is an inflammatory process, primarily affecting proximal muscles, characterized by elevated muscle enzymes and distinctive electromyography patterns.

CASE PRESENTATION

The authors present a case of a 33-year-old male patient experiencing complications of polymyositis, including pharyngeal and laryngeal involvement leading to dysphagia and dysphonia. Steroids and intravenous immunoglobulin (IVIG) therapy proved ineffective. Subsequently, rituximab was administered, resulting in significant improvement in dysphagia, dysphonia, and proximal muscles within 3 days of the initial rituximab dose. Additionally, there was a remarkable decrease in creatine phosphokinase (CPK) levels.

CLINICAL DISCUSSION

Immune-mediated myopathies (IMM) are rare diseases characterized by muscle inflammation and weakness. This case of probable polymyositis, diagnosed through clinical features and elevated CPK, was complicated by the patient's lack of response to glucocorticoids and IVIG therapy. Remarkably, rituximab treatment led to rapid improvement in muscle strength and symptoms, highlighting its potential effectiveness in refractory cases of polymyositis.

CONCLUSIONS

Primary treatment for cases of polymyositis typically involves the use of glucocorticoids. However, approximately half of the patients do not respond adequately to corticosteroids alone. Alternatives, in such cases, encompass IVIG therapy and rituximab.

摘要

引言与重要性

多发性肌炎是一种炎症性疾病,主要累及近端肌肉,其特征为肌肉酶升高和独特的肌电图模式。

病例介绍

作者报告了一例33岁男性患者,患有多发性肌炎并发症,包括咽喉部受累导致吞咽困难和发音障碍。类固醇和静脉注射免疫球蛋白(IVIG)治疗无效。随后给予利妥昔单抗治疗,在首次使用利妥昔单抗剂量后的3天内,吞咽困难、发音障碍和近端肌肉有显著改善。此外,肌酸磷酸激酶(CPK)水平显著下降。

临床讨论

免疫介导性肌病(IMM)是罕见疾病,其特征为肌肉炎症和无力。该例可能的多发性肌炎通过临床特征和CPK升高得以诊断,患者对糖皮质激素和IVIG治疗无反应,病情复杂。值得注意的是,利妥昔单抗治疗使肌肉力量和症状迅速改善,突出了其在难治性多发性肌炎病例中的潜在疗效。

结论

多发性肌炎病例的主要治疗通常使用糖皮质激素。然而,约一半患者仅使用皮质类固醇治疗反应不佳。在此类情况下,替代治疗包括IVIG治疗和利妥昔单抗。

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本文引用的文献

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Rituximab treatment in patients with refractory inflammatory myopathies.利妥昔单抗治疗难治性炎性肌病。
Rheumatology (Oxford). 2011 Dec;50(12):2206-13. doi: 10.1093/rheumatology/ker088. Epub 2011 May 13.

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