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泼尼松龙单药治疗使巨细胞性心肌炎完全缓解:1例T2高信号区与钆增强延迟不匹配显示轻度炎症的病例

Complete remission of giant cell myocarditis by prednisolone monotherapy: A case with mild inflammation demonstrated by mismatch between T2-high intensity areas and late gadolinium enhancement.

作者信息

Soma Takanobu, Kinjo Takahiko, Goto Shintaro, Sasaki Shingo, Tomita Hirofumi

机构信息

Department of Cardiology, Hirosaki University Graduate School of Medicine, Hirosaki, Japan.

Department of Pathology, Hirosaki University Graduate School of Medicine, Hirosaki, Japan.

出版信息

J Cardiol Cases. 2024 Jan 15;29(4):182-185. doi: 10.1016/j.jccase.2023.12.007. eCollection 2024 Apr.

Abstract

UNLABELLED

Giant cell myocarditis (GCM) is a potentially lethal subtype of myocarditis. Herein, we report a case of a 22-year-old woman with GCM who was successfully treated with prednisolone monotherapy. The patient had a fever and shortness of breath and was referred to our hospital. Laboratory test results revealed elevated troponin I levels. Cardiac magnetic resonance (CMR) showed high intensity in the inferoseptal segment of the left ventricle on T2-weighted short tau inversion recovery imaging without late gadolinium enhancement (LGE), suggesting predominant edema rather than necrosis. The patient was diagnosed with GCM based on an endomyocardial biopsy, which revealed lymphocyte infiltration and multinucleated giant cells in the absence of granuloma formation. Subsequently, the patient received intravenous methylprednisolone at 1000 mg/day for 3 days followed by oral prednisolone at 30 mg/day, which normalized troponin levels. Follow-up CMR revealed improved cardiac inflammation; therefore, the patient was discharged without prescribing another immunosuppressive agent. Prednisolone was tapered and terminated three years after discharge. The patient went one year without medication and had no recurrence of GCM on follow-up. This case highlights the presence of mild GCM, successfully treated by steroid monotherapy, in which the mismatch between high-intensity T2 areas and LGE suggests mild inflammation.

LEARNING OBJECTIVE

Giant cell myocarditis (GCM) is potentially lethal and usually requires multiple immunosuppressive agents. Here, we report a patient with GCM with preserved left ventricular ejection fraction. Cardiac magnetic resonance revealed focal high T2 signal intensity areas without late gadolinium enhancement, indicating myocardial edema without necrosis. The patient remained in remission with prednisolone monotherapy for 2 years. Our report indicates that "mild" GCM may be treated with prednisolone monotherapy.

摘要

未标注

巨细胞性心肌炎(GCM)是心肌炎的一种潜在致命亚型。在此,我们报告一例22岁患有GCM的女性患者,该患者接受泼尼松龙单一疗法成功治愈。患者出现发热和呼吸急促症状,随后转诊至我院。实验室检查结果显示肌钙蛋白I水平升高。心脏磁共振成像(CMR)在T2加权短反转时间反转恢复成像上显示左心室下间隔段高强度信号,无延迟钆增强(LGE),提示主要为水肿而非坏死。根据心内膜心肌活检结果,患者被诊断为GCM,活检显示淋巴细胞浸润和多核巨细胞,无肉芽肿形成。随后,患者接受静脉注射甲泼尼龙1000毫克/天,共3天,随后口服泼尼松龙30毫克/天,肌钙蛋白水平恢复正常。随访CMR显示心脏炎症有所改善;因此,患者出院时未再开具其他免疫抑制剂。出院三年后泼尼松龙逐渐减量并停用。患者停药一年,随访时GCM未复发。该病例突出了轻度GCM的存在,通过类固醇单一疗法成功治愈,其中高强度T2区域与LGE之间的不匹配提示轻度炎症。

学习目标

巨细胞性心肌炎(GCM)具有潜在致命性,通常需要多种免疫抑制剂治疗。在此,我们报告一例左心室射血分数保留的GCM患者。心脏磁共振显示局灶性高T2信号强度区域,无延迟钆增强,表明心肌水肿而非坏死。该患者接受泼尼松龙单一疗法缓解2年。我们的报告表明“轻度”GCM可用泼尼松龙单一疗法治疗。

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