{"title":"Myositis associated with antimitochondrial autoantibodies presenting with early respiratory failure","authors":"Kentaro Kawama , Shinsuke Tobisawa , Akinori Uruha , Satoko Uruha , Hirotake Nishimura , Hiromi Onizuka , Kenta Masui , Saeko Yoshizawa , Tomoko Yamamoto , Takashi Komori , Keisuke Ishizawa , Kazushi Takahashi","doi":"10.1016/j.nmd.2025.105395","DOIUrl":null,"url":null,"abstract":"<div><div>Myositis associated with antimitochondrial M2 antibodies (AMA) is a distinct subtype of inflammatory myopathy characterized by axial muscle weakness and, occasionally, respiratory failure. This report highlights two cases involving patients presenting with severe respiratory failure while ambulant. Patient 1: An 82-year-old man was referred to our hospital for respiratory failure and truncal muscle weakness. Shortly after hospitalization, the patient succumbed to respiratory failure. Autopsy findings revealed inflammation predominantly affecting the respiratory muscles. Later, AMA were detected. Patient 2: A 69-year-old woman with a nine-year history of muscle weakness presented with chronic respiratory failure requiring mechanical ventilation. A biceps brachii muscle biopsy revealed mild necrosis. Corticosteroid therapy was successful in weaning the patient off ventilator support. Notably, both patients showed mild limb muscle weakness, contrasting the severity of their respiratory insufficiency. Myositis associated with AMA should be considered in the differential diagnosis of diseases causing respiratory failure in ambulatory patients.</div></div>","PeriodicalId":19135,"journal":{"name":"Neuromuscular Disorders","volume":"52 ","pages":"Article 105395"},"PeriodicalIF":2.7000,"publicationDate":"2025-05-27","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/S0960896625001221","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
Myositis associated with antimitochondrial M2 antibodies (AMA) is a distinct subtype of inflammatory myopathy characterized by axial muscle weakness and, occasionally, respiratory failure. This report highlights two cases involving patients presenting with severe respiratory failure while ambulant. Patient 1: An 82-year-old man was referred to our hospital for respiratory failure and truncal muscle weakness. Shortly after hospitalization, the patient succumbed to respiratory failure. Autopsy findings revealed inflammation predominantly affecting the respiratory muscles. Later, AMA were detected. Patient 2: A 69-year-old woman with a nine-year history of muscle weakness presented with chronic respiratory failure requiring mechanical ventilation. A biceps brachii muscle biopsy revealed mild necrosis. Corticosteroid therapy was successful in weaning the patient off ventilator support. Notably, both patients showed mild limb muscle weakness, contrasting the severity of their respiratory insufficiency. Myositis associated with AMA should be considered in the differential diagnosis of diseases causing respiratory failure in ambulatory patients.
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.