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The occurrence of Brucella endocarditis following Bentall operation is a rare and life threatening condition, particularly when it is complicated by development of a pseudoaneurysm. Here we present a 40-year-old veterinarian with bicuspid aortic valve, who developed type A aortic root dissection and required Bentall operation. His past medical history was positive for an episode of treated brucellosis. Four months after the operation, he developed signs, symptoms and serological findings of brucellosis for the second time and was treated with antibiotics. Then after he was well untill about 14 days prior to his recent admission, when he again developed hip pain, fever, shortness of breath, profound fatigue and weakness. His transthoracic echocardiography was suggestive of endocarditis. A transesophageal echocardiogram revealed detachment of valve-conduit from the annulus and the mitral-aortic intervalvular fibrosa and the presence of a large aortic pseudoaneurysm with multiple vegetations attached to its Dacron walls. A huge pseudoaneurysm was detected at surgery and the whole valve-conduit was replaced with a 25 mm homograft. Blood and vegetation cultures turned to be positive for Brucella melitensis. Early echocardiographic diagnosis and prompt surgical intervention were helpful for survival of our patient who had aortic pseudoaneurysm complicated by Brucella endocarditis.