Concurrent nodular lymphocytic myositis and myasthenia gravis. A case report

IF 2.7 4区医学 Q2 CLINICAL NEUROLOGY

Neuromuscular Disorders Pub Date : 2024-10-05 DOI:10.1016/j.nmd.2024.105211

Eleonora S D'Ambrosio , Matti D Allen , Burcak Ozes , Zarife Sahenk

{"title":"Concurrent nodular lymphocytic myositis and myasthenia gravis. A case report","authors":"Eleonora S D'Ambrosio , Matti D Allen , Burcak Ozes , Zarife Sahenk","doi":"10.1016/j.nmd.2024.105211","DOIUrl":null,"url":null,"abstract":"<div><div>Localized painful nodules associated with focal myositis have been previously documented, either as isolated occurrences or in conjunction with systemic illnesses, such as polymyositis [<span><span>[1]</span></span>, <span><span>[2]</span></span>, <span><span>[3]</span></span>, <span><span>[4]</span></span>]. These nodules typically manifest over the course of 2-8 weeks and exhibit histological features consistent with inflammatory myositis. Here we present a unique case of a patient with a history of myasthenia gravis, who experienced exacerbation of myasthenic symptoms, accompanied by the development of palpable, tender nodules on her thighs and proximal weakness. Muscle biopsy revealed circumscribed mononuclear infiltrates predominantly composed of CD3+ and CD4+ lymphocytes. Furthermore, we detail the patient's rapid and sustained response to treatment with steroids (both intravenous pulse and maintenance therapy) and methotrexate. To the best of our knowledge, this represents the first documented case of nodular lymphocytic myositis concurrent with an exacerbation of myasthenia gravis, in a patient with no prior history of significant immunosuppression or chronic infection.</div></div>","PeriodicalId":19135,"journal":{"name":"Neuromuscular Disorders","volume":"45 ","pages":"Article 105211"},"PeriodicalIF":2.7000,"publicationDate":"2024-10-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Neuromuscular Disorders","FirstCategoryId":"3","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S0960896624017073","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}

引用次数: 0

Abstract

Localized painful nodules associated with focal myositis have been previously documented, either as isolated occurrences or in conjunction with systemic illnesses, such as polymyositis [[1], [2], [3], [4]]. These nodules typically manifest over the course of 2-8 weeks and exhibit histological features consistent with inflammatory myositis. Here we present a unique case of a patient with a history of myasthenia gravis, who experienced exacerbation of myasthenic symptoms, accompanied by the development of palpable, tender nodules on her thighs and proximal weakness. Muscle biopsy revealed circumscribed mononuclear infiltrates predominantly composed of CD3+ and CD4+ lymphocytes. Furthermore, we detail the patient's rapid and sustained response to treatment with steroids (both intravenous pulse and maintenance therapy) and methotrexate. To the best of our knowledge, this represents the first documented case of nodular lymphocytic myositis concurrent with an exacerbation of myasthenia gravis, in a patient with no prior history of significant immunosuppression or chronic infection.

查看原文本刊更多论文

并发结节性淋巴细胞性肌炎和重症肌无力。病例报告

以前曾有过与局灶性肌炎相关的局部疼痛结节的记录，这些结节可能是单独出现的，也可能与多发性肌炎等全身性疾病同时出现[[1], [2], [3], [4]]。这些结节通常在 2 至 8 周内出现，其组织学特征与炎性肌炎一致。我们在此介绍一例独特的病例，患者曾患重症肌无力，肌无力症状加重，大腿上出现可触及的触痛性结节，近端无力。肌肉活检显示，周身单核浸润主要由 CD3+ 和 CD4+ 淋巴细胞组成。此外，我们还详细介绍了患者对类固醇（静脉脉冲疗法和维持疗法）和甲氨蝶呤治疗的快速和持续反应。据我们所知，这是有据可查的首例结节性淋巴细胞性肌炎并发重症肌无力症加重的病例，患者之前没有明显的免疫抑制或慢性感染病史。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

求助全文

约1分钟内获得全文求助全文

来源期刊

Neuromuscular Disorders 医学-临床神经学

CiteScore

4.60

自引率

3.60%

发文量

543

审稿时长

53 days

期刊介绍： This international, multidisciplinary journal covers all aspects of neuromuscular disorders in childhood and adult life (including the muscular dystrophies, spinal muscular atrophies, hereditary neuropathies, congenital myopathies, myasthenias, myotonic syndromes, metabolic myopathies and inflammatory myopathies). The Editors welcome original articles from all areas of the field: • Clinical aspects, such as new clinical entities, case studies of interest, treatment, management and rehabilitation (including biomechanics, orthotic design and surgery). • Basic scientific studies of relevance to the clinical syndromes, including advances in the fields of molecular biology and genetics. • Studies of animal models relevant to the human diseases. The journal is aimed at a wide range of clinicians, pathologists, associated paramedical professionals and clinical and basic scientists with an interest in the study of neuromuscular disorders.