A 53-year-old man, complaining of left calf and hip claudication, was treated with surgery of the occluded common femoral artery. After incision in the artery, gelatinous material came out from the intramural cavity. All the contained material was evacuated, and definitive diagnosis of cystic adventitial disease was confirmed postoperatively. Twenty days later, he complained of identical claudication again. Follow-up study suggested the recurrence. Therefore, the artery replacement with polytetrafluoroethylene graft was performed. Pathologic examinations showed that the adventitial cyst lining cells expressed macrophage markers (CD68 and CD14), while fibroblast-like cells were not found on the lining. Cystic adventitial disease was not derived from synovium in this case.