Myopericarditis in a case of anti-signal recognition particle (anti-SRP) antibody-positive myopathy

Tanaka, Mariko; Gamou, Naoki; Shizukawa, Hirohiko; Tsuda, Etsuko; Shimohama, Shun

doi:10.5692/clinicalneurol.cn-000916

Cited by 5 publications

(8 citation statements)

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“…Case of anti-SRP-positive patient with IIM, who developed inflammatory cardiomyopathy was described [20]. In other case report, anti-SRP-positive myopathy led to myopericarditis [21]. In our study, all the three patients with anti-SRP antibodies declared irregular heartbeat/chest pounding.…”

Section: Discussionsupporting

confidence: 56%

Interleukin-33/suppression of tumorigenicity 2 (IL-33/ST2) axis in idiopathic inflammatory myopathies and its association with laboratory and clinical parameters: a pilot study

et al. 2020

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Idiopathic inflammatory myopathies (IIM) are rare connective tissue diseases, which can lead to internal organ involvement. IL-33/ST2 pathway is involved in the pathogenesis of numerous diseases including autoimmune disorders. IL-33 fulfils cardioprotective function, while soluble ST2 (sST2) is a decoy receptor that reduces protective impact of IL-33. The aim of the study was to evaluate the concentrations of sST2 and IL-33 in sera of patients with IIM and evaluate its associations with the clinical course of the disease. Patients with IIM as well as age-and sex-matched healthy controls were recruited. Concentrations of sST2 and IL-33 were assessed with ELISA in sera of both patients and controls. Patients were asked to fill in the questionnaires concerning clinical symptoms and physical functioning. Concentrations of sST2 and IL-33 were correlated with the results of laboratory tests and clinical symptoms. Concentrations of sST2 were significantly higher in IIM group than in healthy subjects (median sST2 in IIM 26.51 vs in healthy controls 21.39; p = 0.03). In the majority of patients, IL-33 concentrations did not exceed the detection limit. Anti-SRP-positive patients presented significantly higher concentrations of sST2 as compared to anti-SRP-negative patients (p = 0.04). In patients with anti-Ro52 antibodies, sST2 concentrations were significantly lower than in anti-Ro52-negative patients (p = 0.02). Concentrations of sST2 correlated with the degree of disability evaluated with Health Assessment Questionnaire. sST2 is increased in patients with IIM and its concentration correlates with the degree of disability. In patients with anti-SRP antibodies, levels of sST2 are exceptionally high. KeywordsIdiopathic inflammatory myopathy · ST2 · IL-33 · Myositis Rheumatology INTERNATIONAL Electronic supplementary material The online version of this article (https ://doi.org/10.

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Section: Discussionsupporting

confidence: 56%

Interleukin-33/suppression of tumorigenicity 2 (IL-33/ST2) axis in idiopathic inflammatory myopathies and its association with laboratory and clinical parameters: a pilot study

et al. 2020

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“…We identified six case series reporting on patients with myopericarditis with anti-SRP-antibodies [9][10][11][18][19][20]. Three of the cases showed pericardial effusion like the present case [10,11,18], and two showed diastolic left ventricular dysfunction [18,20]. Three reports showed that the initial symptoms were related to heart failure [10,11,19].…”

Section: Discussionmentioning

confidence: 87%

“…They often require aggressive and prolonged immunomodulation [5]. In patients with anti-SRP-antibody-positive myopathy, skin rash, interstitial lung disease, arthritis, and cardiac involvement are reported as extramuscular features [6,[8][9][10][11]. However, recent data suggest that the cardiac involvement rate is relatively low [5,6,12,13].…”

Section: Introductionmentioning

confidence: 99%

Secondary cardiac involvement in anti-SRP-antibody-positive myopathy: an 87-year-old woman with heart failure symptoms as the first clinical presentation

et al. 2020

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Background: Necrotizing myopathy (NM) is defined by the dominant pathological feature of necrosis of muscle fibers without substantial lymphocytic inflammatory infiltration. Anti-signal recognition particle (SRP)-antibodypositive myopathy is related to NM. Anti-SRP-antibody-positive myopathy can comorbid with other disorders in some patients, however, comorbidity with malignant tumor and myopericarditis has still not been reported. Case presentation: An 87-year-old woman with dyspnea on exertion and leg edema was referred to our hospital because of suspected heart failure and elevated serum creatine kinase level. Upon hospitalization, she developed muscle weakness predominantly in the proximal muscles. Muscle biopsy and immunological blood test led to the diagnosis of anti-SRP-antibody-positive myopathy. A colon carcinoma was also found and surgically removed. The muscle weakness remained despite the tumor resection and treatment with methylprednisolone. Cardiac screening revealed arrhythmia and diastolic dysfunction with pericardial effusion, which recovered with intravenous immunoglobulin (IVIg) treatment. Conclusions:We reported the first case of anti-SRP-positive myopathy comorbid with colon carcinoma and myopericarditis. This case is rare in the point that heart failure symptoms were the first clinical presentation. The underlying mechanism is still not clear, however, physicians should be carefully aware of the neoplasm and cardiac involvement in anti-SRP-antibody positive-myopathy patients and should consider farther evaluation and management.

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“…7 Tanaka et al found anti-SRP myopathy with myopericarditis in a patient with marked pericardial effusion, ventricular extrasystoles and non-sustained ventricular tachycardia. 8 In patient 1, sinus insufficiency was observed before the anti-SRP antibody was detected, and this rapidly worsened with increasing CK. Subsequently, myalgia and limb muscle weakness appeared, and anti-SRP antibodies were detected; therefore, a close relationship between deterioration in sinus insufficiency and anti-SRP was suspected.…”

Section: Discussionmentioning

confidence: 94%

“…Tanaka et al . found anti‐SRP myopathy with myopericarditis in a patient with marked pericardial effusion, ventricular extrasystoles and non‐sustained ventricular tachycardia …”

Section: Discussionmentioning

confidence: 97%

Three cases of immune‐mediated necrotizing myopathy with anti‐signal recognition particle antibodies: Extramuscular symptoms preceding progressive muscle weakness

Seino

Nakamura

Hirohata

et al. 2018

Clinical & Exp Neuroim

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Background Immune‐mediated necrotizing myopathy with an anti‐signal recognition particle antibody (anti‐SRP myopathy) is generally refractory to treatment. Anti‐SRP myopathy is characterized by muscle weakness as the main symptom, but is often complicated by extramuscular symptoms. Rarely, the extramuscular symptoms precede muscle weakness. In the present report, we describe the clinical presentation and medical history of three patients with extramuscular symptoms. Focusing on the extramuscular symptoms, anti‐SRP antibody‐positive myopathy can be diagnosed at an early stage. It might therefore be treatable before muscle weakness progresses. Case presentation Patient 1 was a 68‐year‐old woman who suffered progressive muscle weakness after a 2‐year history of creatine kinase elevation, vitiligo, 1‐year history of easy fatigability, sinus insufficiency and a 6‐month history of myalgia. She was followed up in the cardiology department. Patient 2 was a 77‐year‐old woman who showed muscle weakness and respiratory failure after a 3‐day history of malaise, loss of appetite and creatine kinase elevation. She initially visited the internal medicine department and was suspected of having heat stroke at the first visit. Patient 3 was a 48‐year‐old man who had a gastric ulcer and progressive weakness after a 1‐year history of anorexia and severe weight loss. He first visited the gastroenterology department. Conclusions All three patients visited the internal medicine department before the neurology department. It is important to consider anti‐SRP myopathy as a differential diagnosis for patients presenting with elevated creatine kinase levels and extramuscular symptoms.

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Myopericarditis in a case of anti-signal recognition particle (anti-SRP) antibody-positive myopathy

Cited by 5 publications

References 5 publications

Interleukin-33/suppression of tumorigenicity 2 (IL-33/ST2) axis in idiopathic inflammatory myopathies and its association with laboratory and clinical parameters: a pilot study

Interleukin-33/suppression of tumorigenicity 2 (IL-33/ST2) axis in idiopathic inflammatory myopathies and its association with laboratory and clinical parameters: a pilot study

Secondary cardiac involvement in anti-SRP-antibody-positive myopathy: an 87-year-old woman with heart failure symptoms as the first clinical presentation

Three cases of immune‐mediated necrotizing myopathy with anti‐signal recognition particle antibodies: Extramuscular symptoms preceding progressive muscle weakness

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