A persistent median artery is a rare anatomical variant found in approximately 10% of the population. Carpal tunnel syndrome due to a thrombosed persistent median artery is exceptionally rare. A 29-year-old professional hockey player presented with a 2 months history of hand pain and intermittent paresthesias without vascular symptoms. Magnetic resonance imaging and angiography demonstrated a 2-cm thrombosis in a persistent median artery at the level of the carpal tunnel, with otherwise normal vascular anatomy.