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Abstract Background An aberrant origin of the left coronary artery (LCA) from the right coronary cusp (RCC) is an extremely rare congenital anomaly. We here report on successful percutaneous coronary intervention (PCI) in a patient presenting with acute coronary syndrome and an aberrant origin of the LCA from the RCC. Case presentation A 50-year-old man presented at our emergency department with recurrent resting chest pain. Following unsuccessful attempts at visualizing the left coronary artery using Judkins left and Amplatz catheters, an aortogram using a pigtail catheter suggested anomalous left coronary artery origin and showed a significant occlusive lesion at proximal left anterior descending artery. A Judkins right 4 guiding catheter was placed around the left coronary ostium and exchanged for a Judkins left 3.5 guiding catheter after introducing a .014" coronary long wire into the left circumflex artery. With excellent angiographic visualization and guide support, a drug-eluting stent was then successfully implanted. Cardiac computed tomography (CT) demonstrated left coronary artery origin from right coronary cusp. Conclusion This report presents a case of LCA originating from the RCC accompanied with acute coronary syndrome that was treated with successful PCI.