'Comprehensive Aortic Root and Valve Reconstruction' technique, which was first introduced in 2005 involves aortic root reduction using prosthetic rings in order to preserve the native aortic sinuses in patients having aortic regurgitation associated with aortic root dilatation. We report a case of isolated aortic sinus aneurysm in a Marfan syndrome patient following the aortic root preserving surgery in the presence of ascending aorta aneurysm and annuloaortic ectasia. Re-operation consisted of aortic sinus resection and replacement with an artificial graft, and coronary reimplantation using a button technique. Close follow-up is essential for patients who underwent aortic root preserving surgery to appropriately manage this kind of complication.