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Clinical features of anti-mitochondrial M2 antibody-positive myositis : case series of 17 patients

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Title: Clinical features of anti-mitochondrial M2 antibody-positive myositis : case series of 17 patients
Authors: Nagai, Azusa Browse this author
Nagai, Toshiyuki Browse this author →KAKEN DB
Yaguchi, Hiroaki Browse this author
Fujii, Shintaro Browse this author
Uwatoko, Hisashi Browse this author
Shirai, Shinichi Browse this author
Horiuchi, Kazuhiro Browse this author
Iwata, Ikuko Browse this author
Matsushima, Masaaki Browse this author
Ura, Shigehisa Browse this author
Anzai, Toshihisa Browse this author →KAKEN DB
Yabe, Ichiro Browse this author →KAKEN DB
Keywords: Anti-mitochondrial M2 antibody
Inflammatory myopathy
Weight loss
Cardiac complication
Respiratory failure
Chronic clinical course
Issue Date: 15-Nov-2022
Publisher: Elsevier
Journal Title: Journal of the neurological sciences
Volume: 442
Start Page: 120391
Publisher DOI: 10.1016/j.jns.2022.120391
Abstract: Objective: In 2012, a large number of myositis cases with anti-mitochondrial M2 (AMA-M2) antibody, which had well been known as the serological hallmark for primary biliary cholangitis (PBC), were reported in Japan. Recently, some case series from Japan, France, America, China and India have shown that approximately 2.5% to 19.5% of patients with myositis have AMA-M2 antibody. The objective of this study was to clarify the prevalence, clinical features, treatment outcome, and severity determinants of AMA-M2 positive myositis. Methods: This study was a multicenter observational study. We enrolled patients who were diagnosed with myositis during a ten-year period between 2012 and 2021. Results: Of the total of 185 patients with inflammatory myopathy, 17 patients were positive for AMA-M2 anti-body. The typical symptoms were weakness mainly involving paravertebral muscles, weight loss, respiratory failure, and cardiac complications. Thirteen of the 17 patients had cardiac complications. A strong correlation was found between respiratory failure and modified Rankin Scale (mRS) score. A strong correlation was also found between respiratory failure and body weight, indicating that weight loss can be an indicator of potential progression of respiratory failure. Six of the 17 patients were complicated by malignancy. Conclusions: This study showed significant correlations between % vital capacity (VC), body mass index (BMI), and mRS score in patients with AMA-M2-positive myositis. Immunotherapy often improved CK level and res-piratory dysfunction. We therefore propose that %VC and BMI should be monitored as disease indicators in treatment of AMA-M2-positive myositis.
Rights: © 2022, Elsevier. This manuscript version is made available under the CC-BY-NC-ND 4.0 license
Type: article (author version)
Appears in Collections:医学院・医学研究院 (Graduate School of Medicine / Faculty of Medicine) > 雑誌発表論文等 (Peer-reviewed Journal Articles, etc)

Submitter: 矢部 一郎

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