Background: A 38-year-old man with a history of recurrent genital ulceration initially diagnosed as genital herpes was admitted after presenting with paroxysmal nocturnal dyspnea. Echocardiography revealed aneurysm formation of the sinus of Valsalva. On diagnosis of an aneurysm of the sinus of Valsalva associated with Behcet's disease (BD), surgeon repaired the affected sinus only, and continuous steroid therapy maintained the integrity.