Antimitochondrial antibodies-positive myositis accompanied by cardiac involvement

Yamanaka, Tetsuo; Fukatsu, Toru; Ichinohe, Yoshimaro; Hirata, Yasunobu

doi:10.1136/bcr-2016-218469

Cited by 11 publications

(14 citation statements)

References 20 publications

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“…Indeed, we found that the clinical features in patients with AMAs showed great variability. Firstly, although AMAs only occurred in adult IIM patients, the age at onset ranged from 23 years to 86 years and the mean age at onset ranged from the fourth decade to the seventh decade [3][4][5]7,8,[20][21][22][23][24][25][26] . Besides, the average disease duration varied from 10 months (in our study) to 6.5 years, and the prevalence of chronic disease duration varied from approximately 30% (in our study) to 85% [3,5,6] .…”

Section: Discussionmentioning

confidence: 99%

Idiopathic inflammatory myopathies with anti-mitochondrial antibodies: Clinical features and treatment outcomes in a Chinese cohort

Hou

Liu

Luo

et al. 2019

Neuromuscular Disorders

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Anti-mitochondrial antibodies, the hallmark of primary biliary cirrhosis, have been detected in many patients with idiopathic inflammatory myopathies and these anti-mitochondrial-antibody-associated idiopathic inflammatory myopathies frequently show unique characteristics. We detected anti-mitochondrial antibodies in Chinese idiopathic inflammatory myopathy and summarized the clinical findings of these antimitochondrial-antibody-positive patients. Of 136 patients, seven (5.15%) were found to be anti-mitochondrial-antibody-positive. Primary biliary cirrhosis was present in 2 of these 7 patients, chronic disease duration in 2 patients and asymmetrical muscle weakness in 4 patients. The mean disease course was 8.58 months, and the mean creatine kinase level was 2256.53 U/L. Myositis-specific antibodies were found in 3 patients. According to clinical features and muscle histopathological findings, 3 patients were classified as dermatomyositis, 2 as possible polymyositis and 2 as necrotizing autoimmune myopathy. Of the 6 anti-mitochondrial-antibody-positive patients receiving follow-ups of 12-83 months, they all showed marked clinical improvement. Our study indicates that anti-mitochondrial antibodies are relatively rare in Chinese idiopathic inflammatory myopathy patients. These patients generally show various clinical features and have favorable treatment outcomes. Anti-mitochondrial antibody testing may be helpful to confirm the diagnosis of idiopathic inflammatory myopathy, especially in patients with atypical manifestations.

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Section: Discussionmentioning

confidence: 99%

Idiopathic inflammatory myopathies with anti-mitochondrial antibodies: Clinical features and treatment outcomes in a Chinese cohort

Hou

Liu

Luo

et al. 2019

Neuromuscular Disorders

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“…Although AMAs are most commonly found in association with PBC, the presence of the antibody has been linked to other autoimmune conditions, such as Sjögren's syndrome, scleroderma, and autoimmune thyroid disease ( 2 ). Inflammatory myopathy occurring in association with AMA is rare, but there has been growing recognition for this clinical entity ( 3 - 8 ).…”

Section: Discussionmentioning

confidence: 99%

“…Corticosteroids are considered as the first-line therapy for dermatomyositis, polymyositis, and IMNM ( 6 , 8 , 10 ). However, there is no established treatment for AMA-associated myopathy with cardiac involvement.…”

Section: Discussionmentioning

confidence: 99%

“…Although the presence of these antibodies has been linked to other autoimmune conditions, such as Sjögren's syndrome, scleroderma, and autoimmune thyroid disease, inflammatory myopathy occurring in association with AMA is rare ( 2 ). However, this clinical entity is being recognized increasingly frequently ( 3 - 8 ), and cardiac involvement was reported in approximately 30% of cases with AMA-positive myopathy ( 3 ).…”

Section: Introductionmentioning

confidence: 99%

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Antimitochondrial Antibody-associated Myopathy with Slowly Progressive Cardiac Dysfunction

et al. 2021

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A 45-year-old woman was referred to our hospital for the evaluation of proximal muscle weakness and serum creatinine kinase elevation. She had atrial fibrillation and left ventricular asynergy. She was diagnosed with myopathy, accompanied by cardiomyopathy of unknown etiology. She was treated with prednisolone. After long-term follow-up and a detailed examination, the patient was diagnosed with antimitochondrial antibody (AMA)-associated myopathy with cardiac involvement. Although the patient received medical treatment, including beta-blockers and prednisolone, her cardiac function deteriorated progressively. Physicians should consider AMA-associated myopathy when diagnosing myopathies of unknown etiology. The presence of cardiac involvement should be proactively investigated in AMA-associated myopathy.

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“…Hence, the initially sparse muscle symptoms and prolonged diagnostic process with step-wise exclusion of genetic -, other rheumatic-, ischemic- and malignant diseases, before AMA testing provided a final diagnosis, caused a treatment delay, which might in part explain the insufficient cardiac responsiveness to immune suppressants in our patient. However, a diminished cardiac versus muscular response to immunosuppressants in AMA-IM has been observed in previous cases, across genders, disease durations and drug types [ 1 , 2 , 4 , 5 ]. Taken together, this might point to a need for more aggressive strategies to reverse the cardiac manifestations.…”

Section: Discussionmentioning

confidence: 77%

Cardiac arrest in anti-mitochondrial antibody associated inflammatory myopathy

Højgaard

Witting

Rossing

et al. 2021

Oxford Medical Case Reports

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Insight into predictors of cardiac involvement in inflammatory myopathies is sparse. A negative prognostic role of anti-mitochondrial antibodies (AMA) has been noticed and is supported by the current case. We describe a male patient who at the age 40 suffered a cardiac arrest and over the following months experienced progressive heart failure, arrhythmias and proximal muscle weakness. Clinical, genetic and serologic testing and repeated imaging- and histopathological investigations resulted in a diagnosis of AMA-associated, necrotizing, inflammatory myositis with cardiac involvement. Besides a cardiac resynchronization therapy defibrillator, heart failure and antiarrhythmic drugs the patient received successive immunosuppressants, which improved skeletal muscle strength but not cardiac disease progression. At age 45 he died from end-stage heart failure. Clinicians must be aware of AMA-associated myositis as a cause of unclarified heart disease, even in patients with initially sparse extra-cardiac manifestations. Further knowledge of treatment strategies is highly needed for this disease entity.

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Antimitochondrial antibodies-positive myositis accompanied by cardiac involvement

Cited by 11 publications

References 20 publications

Idiopathic inflammatory myopathies with anti-mitochondrial antibodies: Clinical features and treatment outcomes in a Chinese cohort

Idiopathic inflammatory myopathies with anti-mitochondrial antibodies: Clinical features and treatment outcomes in a Chinese cohort

Antimitochondrial Antibody-associated Myopathy with Slowly Progressive Cardiac Dysfunction

Cardiac arrest in anti-mitochondrial antibody associated inflammatory myopathy

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