Popliteal entrapment syndrome should be considered as a cause of lower limb vascular insufficiency in young adults. It is often caused by musculotendinous structures and bony abnormalities are rarely implicated. Early diagnosis and prompt surgical treatment are the key factors for a better prognosis. This report describes a rare case of popliteal artery thrombosis cause by an osteochondroma treated by surgical resection with good results.