Background: Sinus of Valsalva aneurysm (SVA) is an uncommon cardiac defect accounting for only 1% of congenital cardiac anomalies and the most common complication is ruptured into the atrium or ventricle. Very late recurrence of ruptured SVA after patch repair is extremely rare.
Case presentation: We present a case of 57-year-old man had received repair for ruptured Sinus of Valsalva aneurysm at 19 ages. In the clinics, he presented with exertional dyspnea and leg swelling. The serial examination disclosed he had bicuspid aortic valve and very late rupture of SVA connecting to right atrium. After surgical repair again, he was discharged smoothly.
Conclusion: A very late recurrence of ruptured SVA after surgical repair was rare. We reported a case with unique echocardiographic presentation and a successful repair.