An increased incidence of small bowel lymphoma in patients with long-standing celiac sprue is well documented in the literature. Less common is the association of adenocarcinoma of the small intestine. We report a patient with celiac sprue who initially responded to a gluten-free diet. Eighteen months later, diarrhea, abdominal cramps, and bloating was found to have its origin in partial small bowel obstruction. At laparotomy, two distinct adenocarcinomas of the jejunum were resected. Celiac patients who initially respond to gluten withdrawal and subsequently suffer exacerbation while adhering to strict dietary therapy should be carefully evaluated for evidence of a small bowel malignancy.