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

1
Polymyositis Presenting With Nontraumatic Rhabdomyolysis and Dysphagia: A Case Report.多发性肌炎伴非创伤性横纹肌溶解症和吞咽困难:病例报告。
J Investig Med High Impact Case Rep. 2022 Jan-Dec;10:23247096221074589. doi: 10.1177/23247096221074589.
2
Favorable rituximab response in patients with refractory idiopathic inflammatory myopathies.利妥昔单抗治疗难治性特发性炎性肌病疗效良好。
Adv Rheumatol. 2018 Sep 18;58(1):31. doi: 10.1186/s42358-018-0030-z.
3
Development of a New Classification System for Idiopathic Inflammatory Myopathies Based on Clinical Manifestations and Myositis-Specific Autoantibodies.基于临床表现和肌炎特异性自身抗体的特发性炎性肌病新分类系统的建立。
JAMA Neurol. 2018 Dec 1;75(12):1528-1537. doi: 10.1001/jamaneurol.2018.2598.
4
2017 European League Against Rheumatism/American College of Rheumatology Classification Criteria for Adult and Juvenile Idiopathic Inflammatory Myopathies and Their Major Subgroups.2017 年欧洲抗风湿病联盟/美国风湿病学会成人和青少年特发性炎性肌病及其主要亚群分类标准。
Arthritis Rheumatol. 2017 Dec;69(12):2271-2282. doi: 10.1002/art.40320. Epub 2017 Oct 27.
5
Predictors of clinical improvement in rituximab-treated refractory adult and juvenile dermatomyositis and adult polymyositis.利妥昔单抗治疗难治性成人和青少年皮肌炎及成人多发性肌炎的临床改善的预测因素。
Arthritis Rheumatol. 2014 Mar;66(3):740-9. doi: 10.1002/art.38270.
6
Incidence and prevalence of idiopathic inflammatory myopathies in South Australia: a 30-year epidemiologic study of histology-proven cases.南澳大利亚特发性炎性肌病的发病率和流行率:组织学证实病例的 30 年流行病学研究。
Int J Rheum Dis. 2013 Jun;16(3):331-8. doi: 10.1111/j.1756-185X.2011.01669.x. Epub 2011 Oct 13.
7
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.
8
Intravenous immunoglobulins for steroid-refractory esophageal involvement related to polymyositis and dermatomyositis: a series of 73 patients.静脉注射免疫球蛋白治疗类固醇激素抵抗性皮肌炎/多发性肌炎相关食管受累:73 例患者系列研究。
Arthritis Care Res (Hoboken). 2010 Dec;62(12):1748-55. doi: 10.1002/acr.20325.
9
Estimating the prevalence of polymyositis and dermatomyositis from administrative data: age, sex and regional differences.利用行政数据估算多发性肌炎和皮肌炎的患病率:年龄、性别及地区差异
Ann Rheum Dis. 2009 Jul;68(7):1192-6. doi: 10.1136/ard.2008.093161. Epub 2008 Aug 19.
10
Efficacy of high-dose intravenous immunoglobulin therapy in Japanese patients with steroid-resistant polymyositis and dermatomyositis.大剂量静脉注射免疫球蛋白疗法对日本类固醇抵抗性多发性肌炎和皮肌炎患者的疗效。
Mod Rheumatol. 2008;18(1):34-44. doi: 10.1007/s10165-007-0013-0. Epub 2008 Jan 25.

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.

DOI:10.1097/MS9.0000000000002490
PMID:39359797
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11444562/
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。