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19 November 2015 : Case report  Japan

Spontaneous Remission in a Case of Giant Cell Myocarditis with Preserved Left Ventricular Ejection Fraction

Unusual clinical course, Challenging differential diagnosis, Unusual or unexpected effect of treatment, Diagnostic / therapeutic accidents, Educational Purpose (only if useful for a systematic review or synthesis)

Misa HayaseBDE, Koichi NagashimaABCDEF, Mahoto KatoC, Daisuke FukamachiB, Kazuki IsoB, Masaru AraiB, Yoshihiro NakamuraB, Yukino IwasawaB, Haruna NishimakiCD, Yoshiaki KusumiCD, Yasuo OkumuraE, Satoshi KunimotoC, Atsushi HirayamaCD

DOI: 10.12659/AJCR.895253

Am J Case Rep 2015; 16:823-826

Abstract

BACKGROUND: Giant cell myocarditis (GCM) is rapidly progressive fulminant myocarditis causing death or requiring cardiac transplantation despite various immunosuppression therapies.

CASE REPORT: A 28-year-old woman with progressive shortness of breath and palpitation following an upper respiratory infection was referred to our institution. On admission, transthoracic echocardiography (TTE) revealed a preserved left ventricular ejection fraction (LVEF) with mildly impaired LV diastolic function despite extensive ECG abnormalities, a mildly elevated troponin I concentration, and moderately elevated N-terminal pro-brain natriuretic peptide (NT-pro-BNP) concentration. The diagnosis of GCM was made by endomyocardial biopsy (EMB), which revealed extensive fibrosis and inflammatory infiltration with multinucleated giant cells, as well as scattered eosinophils and lymphocytes in the absence of granuloma formation. However, the patient’s symptoms began to improve without any specific therapy within 2 weeks, followed by the normalization of the ECG abnormalities, TTE-determined diastolic function, and troponin I and NT-pro-BNP concentrations. In sub-acute phase, 18F-fluorodeoxyglucose positron emission tomography showed no evidence of inflammation, and repeat EMB showed a significant decrease in the inflammatory infiltration and fibrosis, including absence of giant cells. Given the favorable clinical course, the patient was discharged without medications. At the 6-month follow-up, the patient had no LV functional impairment, cardiovascular events, or arrhythmia.

CONCLUSIONS: We encountered a rare case of atypical GCM in which clinical and histologic remission was achieved without immunosuppression therapy. There seems to be a population of GCM patients who improve without immunosuppression therapy. In monitoring GCM patients, clinicians should be aware of the possibility of spontaneous remission.

Keywords: Biopsy, Diagnosis, Differential, Echocardiography, Electrocardiography, Giant Cells - pathology, Magnetic Resonance Imaging, Cine, Myocarditis - physiopathology, Myocardium - pathology, Positron-Emission Tomography, Remission, Spontaneous, Stroke Volume - physiology, Ventricular Function, Left - physiology

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923