The authors report their experience in the management of a rare case of bilateral popliteal artery entrapment syndrome occurred to their observation. This unusual pathological entity, arising mostly in young subjects and unilaterally, in this case was bilateral and developed in a 51 years old patient. The clinical case is fully documented by multiple Echo-Doppler and arteriographic examinations which clearly demonstrate the peculiar characteristics of the disease. Finally the patient received a two stage surgical treatment with a good global result. The Authors stress the importance of a correct diagnostic approach to this unusual disease to avoid the possibility to misdiagnose it.