The occurrence of small-bowel cancer in Crohn's disease (CD) is a rare event. The risk seems to be greatest in patients with long-standing disease. Strictureplasty has proved to be a valuable alternative in the management of Crohn's strictures of the small-bowel. Critics and proponents of strictureplasty for selected patients with small-bowel Crohn's disease have voiced their concerns about cancer risk in the strictured or strictureplasty site. To date, there has been no clear or detailed report of such an occurrence. The authors report the first case of small-bowel adenocarcinoma arising at the site of a previous strictureplasty. In this patient, biopsies of the strictures at the original operation confirmed CD and excluded both cancer and dysplasia. Malignancy occurred seven years later at a strictureplasty site. The main clinical sign associated with the adenocarcinoma was severe, persistent anemia. The authors conclude that the risk of adenocarcinoma developing at the site of a previous strictureplasty for CD, although small, is real.