Abstract: 　Objective　To explore the clinical significance of anomalous origin of coronary artery from the aorta. Methods　The clinical data of a case of anomalous origin of coronary artery from the aorta which was misdiagnosed as fulminant myocarditis in the early stage of the disease were retrospectively analyzed and relative literature were reviewed. Results　A 9-year-old girl had syncope after exercise and was found to have the significant elevation of myocardial enzyme and troponin. Electrocardiogram showed myocardial ischemia changes and the cardiac ultrasonography indicated decreased cardiac function. It was diagnosed as the left coronary artery from the right coronary sinus by the CT angiography. Conclusion　The anomalous origin of coronary artery is a rare cardiovascular malformation. Some patients have a potential risk of sudden death.