Introduction: Symmetrical dilatation of the aortic root may be observed in patients with congenital heart diseases, either as an inherent characteristic of the condition or as a consequence of surgical manipulation during repair procedures. Furthermore, as a less common manifestation of dilatation, an aneurysm of the sinus of Valsalva may also develop. Case: We herein report a case of 37-year-old male who developed severe left pulmonary arterial stenosis and residual shunt following the surgical repair for closure of a ventricular septal defect. In this particular patient, there was a simultaneous presence of symmetric aortic root dilatation and an aneurysm in the right coronary sinus. The patient underwent an aortic root reimplantation procedure, which was performed concurrently with left pulmonary arterial plasty and closure of the residual ventricular septal defect. The postoperative course proceeded without complications, and there were no persistent residual cardiac defects observed in the patient. Observation: This case highlights a distinctive variation of aortic root dilatation, potentially unique to certain types of congenital heart diseases.
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