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A 37-year-old man developed bilateral sensorineural hearing loss with tinnitus 14 months prior to admission to our hospital. He had no febrile symptoms such as fever and fatigue despite an increase in the serum level of C-reactive protein. Contrast-enhanced computed tomography imaging demonstrated diffuse wall thickening of the ascending aorta and aortic arch, leading to a diagnosis of TKA. He was treated with prednisolone, concomitantly with methotrexate, resulting in complete recovery of auditory acuity. This report suggests that sensorineural hearing loss may develop as the initial and main manifestation in TKA. Furthermore, it is necessary to initiate appropriate immunosuppressive therapy based on the definite diagnosis of TKA for restoring auditory function. 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