Successful Steroid Therapy for Heart Failure Due to Myocarditis Associated With Primary Biliary Cirrhosis

Matsumoto, Kensuke; Tanaka, Hidekazu; Yamana, Shota; Kaneko, Akira; Tsuji, Takayuki; Ryo, Keiko; Sekiguchi, Kenji; Kawakami, Fumi; Kawai, Hiroya; Hirata, Ken–ichi

doi:10.1016/j.cjca.2011.12.010

Cited by 26 publications

(26 citation statements)

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“…Among the nine AMAs, AMA-M7 has been detected in patients with acute and chronic cardiomyopathies ( 12 , 13 ). AMA-M2 was also found in some cardiomyopathy patients ( 6 , 7 ). Notably, Varga et al described the presence of skeletal mitochondrial damage without significant inflammation in PBC patients ( 14 ), suggesting a direct disturbance of the muscular energy metabolism by the AMAs.…”

Section: Discussionmentioning

confidence: 82%

“…Although the mechanism underlying the development of AMA-M2-associated myopathy has yet to be clarified, it is considered likely that mitochondrial dysfunction and autoimmunity, which are maintained by the predominant autoreactive T cells, are associated with the skeletal muscular damage ( 8 ). Furthermore, concomitant cardiac involvement has been reported in patients with AMA-M2-associated myopathy, as revealed by ventricular dysfunction and/or various arrhythmias ( 5 , 6 ). Among patients with such arrhythmias, the prevalence of supraventricular arrhythmias has been reported to be high ( 5 ), which might suggest a preferential atrial myocardial involvement.…”

Section: Introductionmentioning

confidence: 99%

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Anti-mitochondrial M2 Antibodies Enhance the Risk of Supraventricular Arrhythmias in Patients with Elevated Hepatobiliary Enzyme Levels

et al. 2017

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Objective Supraventricular arrhythmias are commonly detected in patients with anti-mitochondrial antibody M2 (AMA-M2)-associated myopathy. However, the prevalence of supraventricular arrhythmias in unselected AMA-M2-positive patients and the impact of AMA-M2 on supraventricular arrhythmias have yet to be fully investigated. Methods We analyzed 384 patients (116 men; age, 60 [48-69] years), who underwent AMA-M2 testing following the detection of elevated hepatobiliary enzymes. Supraventricular arrhythmias involving atrial fibrillation, atrial flutter, atrial tachycardia, sick sinus syndrome, and atrial standstill were confirmed by a 12-lead electrocardiogram, 24-hour ambulatory monitoring, and physician-assigned diagnoses within the three years before and two years after the AMA-M2 test. Results Seventy-seven (20%) patients were positive for AMA-M2. The prevalence of supraventricular arrhythmias among AMA-M2-positive patients was higher than that among AMA-M2-negative patients (14% vs. 6%, p=0.008). A univariate analysis showed that supraventricular arrhythmias were associated with AMA-M2 positivity, aging, congestive heart failure, and the CHADS2 score. The multivariate analysis determined that AMA-M2 positivity was an independent risk factor for supraventricular arrhythmias (odds ratio 3.52, p=0.011). Among the AMA-M2-positive patients, the AMA-M2 titer did not differ to a statistically significant extent, regardless of the presence or absence of supraventricular arrhythmias. Multiple supraventricular arrhythmias with extremely low atrial deflections was a characteristic finding in AMA-M2-positive patients with supraventricular arrhythmias. Conclusion AMA-M2 enhances the risk of supraventricular arrhythmias, indicating the possible involvement of the atrial myocardium and the formation of an arrhythmogenic substrate. The results highlight the need for clinical attention to supraventricular arrhythmias in AMA-M2-positive patients.

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Section: Discussionmentioning

confidence: 82%

Section: Introductionmentioning

confidence: 99%

Anti-mitochondrial M2 Antibodies Enhance the Risk of Supraventricular Arrhythmias in Patients with Elevated Hepatobiliary Enzyme Levels

et al. 2017

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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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“…There is only one previously reported case of this disease in which myocardial biopsy was performed 19. According to this report, the biopsy revealed interstitial fibrosis.…”

Section: Discussionmentioning

confidence: 81%

“…However, it is unclear whether this improvement was due to PSL, as it may have resulted from an improvement in the tachycardia-induced cardiomyopathy. While improvement in cardiac involvement with recovery of muscle strength induced by PSL has been reported,19 there have also been some reports of cases in which cardiac involvement remained unchanged,13 17 and cases in which arrhythmia did not improve and actually worsened, requiring a pacemaker and catheter ablation,12 or implantable cardioverter defibrillator implantation 18. From these reports,12 13 17–19 and the present case, in this disease, cardiac involvement might be hard to respond to PSL than myositis.…”

Section: Discussionmentioning

confidence: 99%

Antimitochondrial antibodies-positive myositis accompanied by cardiac involvement

et al. 2017

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We report a 55-year-old man who experienced proximal muscle weakness accompanied by the atrial flutter (AFL) with 1:1 conduction. Detailed examination revealed elevated antimitochondrial antibodies (AMA) and creatine kinase (CK). AFL was converted to sinus rhythm by cardioversion. He was diagnosed as AMA-positive myositis-associated AFL and was treated by prednisolone. Although his muscle weakness and CK level improved, AFL with 1:1 conduction reappeared. Therefore, radiofrequency catheter ablation (RFCA) was needed to treat the AFL, resulting in maintenance of sinus rhythm. This case report describes cardiac involvement in a patient with AMA-positive myositis.

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Successful Steroid Therapy for Heart Failure Due to Myocarditis Associated With Primary Biliary Cirrhosis

Cited by 26 publications

References 4 publications

Anti-mitochondrial M2 Antibodies Enhance the Risk of Supraventricular Arrhythmias in Patients with Elevated Hepatobiliary Enzyme Levels

Anti-mitochondrial M2 Antibodies Enhance the Risk of Supraventricular Arrhythmias in Patients with Elevated Hepatobiliary Enzyme Levels

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

Antimitochondrial antibodies-positive myositis accompanied by cardiac involvement

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