Spontaneous Remission in a Case of Giant Cell Myocarditis with Preserved Left Ventricular Ejection Fraction
Spontaneous Remission in a Case of Giant Cell Myocarditis with Preserved Left Ventricular Ejection Fraction
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DOI：
10.12659/ajcr.895253
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发表时间：
2015-01-01
影响因子：
1.2
通讯作者：
Hirayama, Atsushi
中科院分区：
文献类型：
--
作者：
Hayase, Misa;Nagashima, Koichi;Hirayama, Atsushi
病人：女性，28岁最终诊断：巨细胞心肌炎症状：进行性呼吸急促和心悸药物治疗：无临床操作：肌内膜活检。核磁共振PET专业：心脏病学目的：不寻常的临床病程背景：巨细胞心肌炎（GCM）是一种快速进行性暴发性心肌炎，导致死亡或需要心脏移植，尽管各种免疫抑制剂therapy.Case报告：一个28岁的女性进行性呼吸急促和心悸后，上呼吸道感染被提到我们的机构。入院时，经胸超声心动图（TTE）显示左心室射血分数（LVEF）保留，左心室舒张功能轻度受损，尽管存在广泛的ECG异常，肌钙蛋白I浓度轻度升高，N末端脑钠肽前体（NT-pro-BNP）浓度中度升高。GCM的诊断是通过肌内膜活检（EMB）进行的，EMB显示广泛的纤维化和炎性浸润，伴多核巨细胞，以及散在的嗜酸性粒细胞和淋巴细胞，无肉芽肿形成。然而，患者的症状在2周内开始改善，无需任何特定治疗，随后ECG异常、经胸超声心动图测定的舒张功能以及肌钙蛋白I和NT-pro-BNP浓度恢复正常。在亚急性期，F-18-氟脱氧葡萄糖正电子发射断层扫描显示无炎症证据，重复EMB显示炎性浸润和纤维化显著减少，包括巨细胞消失。鉴于良好的临床病程，患者出院，未用药。在6个月的随访中，患者没有左心室功能障碍，心血管事件，或arrhythm.Conclusions：我们遇到了一个罕见的情况下，非典型GCM的临床和组织学缓解实现无免疫抑制治疗。似乎有一群GCM患者在没有免疫抑制治疗的情况下改善。在监测GCM患者时，临床医生应了解自发缓解的可能性。
Patient: Female, 28Final Diagnosis: Giant cell myocarditisSymptoms: Progressive shortness of breath and palpitationMedication: NoneClinical Procedure: Endomyocardial biopsy . MRI . PETSpecialty: CardiologyObjective: Unusual clinical courseBackground: 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, F-18-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.