Cystic adventitial disease is a rare disease that usually affects the popliteal artery and it is a rare cause of non-atherosclerotic stenosis. It is most commonly found in young or middle-aged men with intermittent claudication. Herein we present a histologically proven case of cystic adventitial disease of the popliteal artery in a 53-year-old man and we describe the imaging findings on gray-scale, on Doppler US and triplex sonography, on computed tomography with 3D reconstructions and on digital subtraction angiography.