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Bicuspid aortic valves (BAV) can be associated with aortopathy and coronary anomalies. We report the case of a 60 year-old woman undergoing surgery for severe aortic stenosis due to BAV and an ascending aortic aneurysm. During the procedure, an uncommon anomalous origin of the left main coronary artery from the posterior commissure with intramural takeoff of the left coronary artery was found. Routine pre-operative coronary angiography had failed to identify this anomaly. To avoid ischemic events or left main coronary lesions, we placed the aortic bioprosthesis by respecting the commissures, not to occlude the anomalous coronary ostium. The association of BAV, aortopathy and coronary anomalies is a rare finding. Awareness of the anatomy of the coronary arteries in patients with BAV should be considered mandatory to avoid catastrophic consequences and to select the appropriate surgical procedure.

Aortopathy and coronary anomaly in bicuspid aortic valve: an uncommon tricky association