Coronary arteries originating from the opposite coronary cusp and crossing the path between the aorta and the pulmonary artery are associated with ischemia and sudden cardiac death. An increased prevalence of these cases may be attributed to diagnostic advances in computed tomographic angiography (CTA). We report a retrospective review of ten patients referred for surgical intervention from March 2008 to present. Nine patients were diagnosed with right coronary arteries arising from the left coronary cusp and one patient with a left coronary artery arising from the right coronary cusp. Seven patients were male and the median age was 40 years (range, 21 to 51). Symptoms included atypical chest pain, tachy-arrythmias, diaphoresis, and dyspnea on exertion. CTA demonstrated anomalous coronary arteries arising from the opposite coronary cusp and traveling between the aorta and the pulmonary artery. Surgical intervention was performed on all ten patients with no mortality and only one re-operation requiring bypass grafting. The sixth patient in the series had concomitant atherosclerotic disease, requiring left internal mammary artery grafting to the left anterior descending coronary artery. Cardiopulmonary bypass (CPB) was utilized with moderate hypothermia in all ten patients, with retrograde and/or coronary ostial cardioplegia administration. At routine surgical follow-up, all patients were without original presenting symptoms. Patients with anomalous coronary arteries arising from the opposite coronary cusp are at risk of acute myocardial infarction and sudden cardiac death. Surgical unroofing is a viable option for this patient population and avoids coronary artery bypass grafting. Since March 2008, we have operated on ten patients presenting with this anomaly and have had excellent short-term results. Further long-term follow-up is necessary.