Myxovirus resistance protein 1–expressing fatal myocarditis in a patient with anti-MDA5 antibody–positive dermatomyositis

Sakamoto, Rika; Kotobuki, Yorihisa; Iga, Saki; Nojima, Satoshi; Deno, Rikako; Hanaoka, Yuma; Tonomura, Kyoko; Kiyohara, Eiji; Nakagawa, Yukinobu; Ueda‐Hayakawa, Ikuko; Arase, Noriko; Fujimoto, Manabu

doi:10.1093/rheumatology/keab637

Cited by 5 publications

(3 citation statements)

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“…In our study, ventricular wall dyskinesia was observed in 50% patients who have MI, and three of them had a disease duration longer than 6 months and were complicated with severe systolic dysfunction presenting as significantly decreased left ventricular ejection fraction (LVEF). This phenomenon is consistent with two former reported cases (a 55-year-old man with a 7-month disease duration and a 48-year-old man with a 6-month disease duration) with a severe LVEF decline ( 6 , 8 ). The results suggest that the MI of anti-MDA5 Ab+ DM/CADM predisposes patients to severe cardiac systolic dysfunction along with disease duration.…”

Section: Discussionsupporting

confidence: 93%

“…Documented cardiac manifestations in IIM patients are diverse including myocardial ischemia, arrhythmias, conduction defects, cardiomyopathies, pericardium diseases, and pulmonary hypertension (5), whereas reports of cardiac involvement in anti-MDA5 Ab+ dermatomyositis/clinically amyopathic dermatomyositis (DM/CADM) patients are scarce. In the past decades, only three cases of anti-MDA5 Ab+ DM patients with severe myocardial defects have been reported (6)(7)(8). A recent study revealing distinctive electrocardiography (ECG) changes in anti-MDA5 Ab+ DM/ CADM patients (9) further indicates cardiac involvement of anti-MDA5 Ab+ DM/CADM patients.…”

Section: Introductionmentioning

confidence: 99%

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Myocardial involvement is not rare in anti-melanoma differentiation-associated gene 5 antibody-positive dermatomyositis/clinically amyopathic dermatomyositis: a retrospective study

Zhou

Lai

et al. 2022

Front. Immunol.

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ObjectivesStudies concerning myocardial involvement (MI) in patients with anti-melanoma differentiation-associated gene 5 antibody-positive dermatomyositis/clinically amyopathic dermatomyositis (anti-MDA5 Ab+ DM/CADM) are scarce. We aimed to characterize MI in our anti-MDA5 Ab+ DM/CADM cohort and to investigate its association with prognosis.MethodsIn this single-center retrospective study, anti-MDA5 Ab+ hospitalized DM/CADM patients who underwent transthoracic echocardiography (TTE) were enrolled. Myocardial involvement was diagnosed according to abnormal cardiac structure and function detected by TEE. Clinical features and cardiac examination findings of patients with MI were analyzed. Clinical features, laboratory findings, complications, and treatments were compared between MI and non-MI, deceased, and survival patients. Logistic regression analysis was used to explore the independent risk factors for the occurrence of MI and prognostic factors for these patients.ResultsSeventy-six hospitalized patients with anti-MDA5 Ab+ DM/CADM were enrolled. Twelve (15.8%) patients were diagnosed with MI. Of the 12 patients, three underwent cardiac magnetic resonance imaging (CMR) and late gadolinium enhancement (LGE) were noted for them. TEE revealed that eight (66.7%) patients had left atrial and/or ventricular enlargement, three (25.0%) had cardiac hypertrophy, six (50.0%) had diffuse ventricular wall dyskinesia, and seven (58.3%) had diastolic dysfunction. Six (50.0%) patients with MI developed heart failure (HF) during treatment. Of the 12 patients, one patient died of HF caused by myocarditis, three died of infection, and four died of exacerbation of rapidly progressive interstitial lung disease (RP-ILD). Logistic regression analysis revealed that dysphagia (OR 3.923, 95% CI 1.085, 14.181), NT-proBNP >600 pg/ml (OR 18.333, 95% CI 1.508, 222.875), and increased peripheral white blood cells (OR 1.201, 95% CI 1.003, 1.438) were risk factors for the occurrence of MI, but plasma albumin (OR 0.892, 95% CI 0.796, 0.999) was a protective factor. Both MI (OR 5.984, 95% CI 1.174, 30.496) and RP-ILD (OR 11.875, 95% CI 2.796, 50.411) were independent risk factors for the mortality of these anti-MDA5 Ab+ DM/CADM patients.ConclusionMyocardial involvement is not rare and is an independent poor prognostic factor of anti-MDA5 Ab+ DM/CADM patients. Cardiac abnormality screening is necessary for them.

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Section: Discussionsupporting

confidence: 93%

Section: Introductionmentioning

confidence: 99%

Myocardial involvement is not rare in anti-melanoma differentiation-associated gene 5 antibody-positive dermatomyositis/clinically amyopathic dermatomyositis: a retrospective study

Zhou

Lai

et al. 2022

Front. Immunol.

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“…Mx1 (myxovirus resistance 1) belong to an important antiviral protein in the innate immune responses of vertebrates to microbial pathogens. Previous study has reported the fatal myocarditis in a patient with anti-MDA5 antibody–positive dermatomyositis accompanied with Myxovirus resistance protein 1 [ 54 ].…”

Section: Discussionmentioning

confidence: 99%

Construction and evaluation of immune-related diagnostic model in patients with heart failure caused by idiopathic dilated cardiomyopathy

Xu,

Wu,

Chen

2024

BMC Cardiovasc Disord

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Objective The purpose of the study was to construct the potential diagnostic model of immune-related genes during the development of heart failure caused by idiopathic dilated cardiomyopathy. Method GSE5406 and GSE57338 were downloaded from the GEO website (https://www.ncbi.nlm.nih.gov/geo/). CIBERSORT was used for the evaluation of immune infiltration in idiopathic dilated cardiomyopathy (DCM) of GSE5406. Differently expressed genes were calculated by the limma R package and visualized by the volcano plot. The immune-related genes were downloaded from Immport, TISIDB, and InnateDB. Then the immune-related differential genes (IRDGs) were acquired from the intersection. Protein–protein interaction network (PPI) and Cytoscape were used to visualize the hub genes. Three machine learning methods such as random forest, logical regression, and elastic network regression model were adopted to construct the prediction model. The diagnostic value was also validated in GSE57338. Results Our study demonstrated the obvious different ratio of T cell CD4 memory activated, T cell regulatory Tregs, and neutrophils between DCM and control donors. As many as 2139 differential genes and 274 immune-related different genes were identified. These genes were mainly enriched in lipid and atherosclerosis, human cytomegalovirus infection, and cytokine-cytokine receptor interaction. At the same time, as many as fifteen hub genes were identified as the IRDGs (IFITM3, IFITM2, IFITM1, IFIT3, IFIT1, HLA-A, HLA-B, HLA-C, ADAR, STAT1, SAMHD1, RSAD2, MX1, ISG20, IRF2). Moreover, we also discovered that the elastic network and logistic regression models had a higher diagnostic value than that of random forest models based on these hub genes. Conclusion Our study demonstrated the pivotal role of immune function during the development of heart failure caused by DCM. This study may offer new opportunities for the detection and intervention of immune-related DCM.

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Anti-MDA5 antibody-positive dermatomyositis: pathogenesis and clinical progress

Lu,

Peng,

Wang

2023

Nat Rev Rheumatol

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Myxovirus resistance protein 1–expressing fatal myocarditis in a patient with anti-MDA5 antibody–positive dermatomyositis

Cited by 5 publications

References 7 publications

Myocardial involvement is not rare in anti-melanoma differentiation-associated gene 5 antibody-positive dermatomyositis/clinically amyopathic dermatomyositis: a retrospective study

Myocardial involvement is not rare in anti-melanoma differentiation-associated gene 5 antibody-positive dermatomyositis/clinically amyopathic dermatomyositis: a retrospective study

Construction and evaluation of immune-related diagnostic model in patients with heart failure caused by idiopathic dilated cardiomyopathy

Anti-MDA5 antibody-positive dermatomyositis: pathogenesis and clinical progress

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