A 62-year-old patient with a history of an acute myeloid leukemia in follow-up, underwent an allotransplantation in February 2019 complicated by grade 4 acute intestinal GVHD and a previous sepsis episode caused by Staphylococcus epidermidis. In October 2019 the patient was affected by an Aspergillus Terreus endophthalmitis of the left eye treated with vitrectomy and voriconazole for three months. In June 2020, he arrived at the emergency department with a sudden onset of chest pain and palpitations. Upon examination, the emergency room staff discovered a third-degree heart block and a temporal pacemaker was implanted. The chest-abdomen CT revealed an aortic hematoma and periaortic effusion without signs of acute aortic dissection. The TEE confirmed the presence of a thrombosed pseudoaneurysm from the non-coronary sinus without signs of endocarditis. The heart team decided for an urgent non-coronary sinus replacement with Dacron prosthesis and definitive bicameral pacemaker implantation. During the operation a longitudinal aortotomy revealed a 1.5 cm breach linked to the pseudoaneurysm extending around the aortic root. The pseudoaneurysm was excised, and tissue samples were sent for examination. Empirical therapy with Meropenem and vancomycin was initiated in the early postoperative hours. Then the biopsy cultures revealed an infection caused by Aspergillus terreus (with persistent negative blood cultures), leading to the initiation of voriconazole and confirming the final diagnosis of aortic mycotic pseudoanuerysm. Moreover encephalic CT (negative), ophthalmological visit (no signs of inflammation) and oncohematologic evaluation were performed.The patient carried on her course in the Cardiac Rehabilitation and then into the Infective Disease department. Control TTE and TEE revealed the presence of endocarditis: a large vegetation and abscess on the non-coronary cusp caused a severe aortic regurgitation, abscess at the myoaortic junction extending towards the interatrial septum and a fistula between the right aortic cusp and the right atrium. The case was reassessed by the cardiac surgeons who didn’t recommend surgery. Given the good hemodynamic status, the patient was discharged at home to continue long life-term antifungal therapy. From dismissal, the patient continued regular cardiological visits with no evidence of recurrences of endocarditis.