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Summary Background There is a lack of Tunisian data on the frequency and clinical significance of different coronary artery anomalies. Methods All patients who underwent coronary angiography from March 1996 to December 2006 were considered. Only patients with congenital anomalous aortic origin of the coronary artery were included. Results Among 7330 adult patients who underwent diagnostic coronary angiography, 20 (0.27%) patients (13 men; mean age 53.3 years) had anomalies of the coronary artery origin. The right coronary artery was the vessel involved most frequently ( n = 10); it originated separately from the left sinus of Valsalva (SV) in three patients and from the posterior sinus of Valsalva in one patient. In the other patients, it arose from the left main coronary artery or its branches in a single coronary artery originating from the left sinus of Valsalva. Isolated anomalous left circumflex artery was the second most frequent anomaly ( n = 6). Isolated anomalous left anterior descending artery was seen in one patient. A single coronary artery arising from the right SV was seen in three patients. Atheroslerotic lesions were seen in eight cases. Four patients underwent coronary revascularization; the remainder received medical management. All 20 patients are alive and had an uneventful follow-up (mean 34.2 months). Conclusions In Tunisia, the incidence of congenital anomalous aortic origin of the coronary artery in adults is 0.27%. The right coronary artery is involved most frequently. Medical management seems promising.