Background <p>Takayasu arteritis is a large-vessel vasculitis, in addition to giant cell arteritis. Various post-operative complications associated with the cardiac macrovasculature have been reported. Detachment of the prosthetic valve, pseudoaneurysm formation, and dilatation of the aortic root are well-known post-operative complications associated with vasculitis syndromes, including Takayasu arteritis. Here, we report a rare complication involving aortic bioprosthetic valve dislodgement in the left ventricular outflow tract due to Takayasu arteritis.</p> Case presentation <p>A 76-year-old female underwent aortic valve replacement with a 21-mm Carpentier–Edwards Perimount valve for severe aortic regurgitation and a coronary artery bypass graft from the left internal thoracic artery to the left anterior descending artery for ischemic heart disease. Fourteen years after the initial surgery, echocardiography revealed severe aortic valve sclerosis due to structural valve deterioration of the bioprosthesis. Upon scrutiny, the bioprosthetic aortic valve was found to have dislodged into the left ventricular outflow tract. We performed re-implantation of the bioprosthetic aortic valve and replacement of the ascending aorta.</p> Conclusions <p>Although dislodgement of the bioprosthetic aortic valve is an extremely rare complication associated with Takayasu arteritis, the possibility that it could occur should be considered when treating the post-operative patients.</p>