Cystic disease of the popliteal artery is a rare disorder in which most cases involve the formation of an adventitial cyst that disturbs the popliteal artery blood flow. We present herein the case of a patient presenting with popliteal artery occlusion due to compression by a cyst which formed at the media of the popliteal artery. The onset occurred during a baseball game in which he played catcher. Preoperative magnetic resonance imaging demonstrated a communication of the cyst with the adjacent knee joint. This unusual case could provide important clues to help identify the pathogenesis of this disease.