Background: A subset of celiac patients shows a high risk for small bowel malignancies. Aims: To select celiac patients considered at risk and evaluate the diagnostic yield of enteroscopy in this context. Methods: Celiac patients were enrolled from a tertiary referral centre during the period June 2011-June 2013, based on the following criteria: (i) patients diagnosed when aged 50+ and with poor response to gluten-free dieting; (ii) low dietary compliance; (iii) alarm symptoms. The patients underwent small bowel capsule endoscopy and/or double-balloon enteroscopy. Control populations were represented by the 165 non-celiac patients undergoing capsule endoscopy for obscure gastrointestinal bleeding, and the 815,362-strong population of the Italian province of Varese as a registered cohort. Results: Fifty-three patients (19% males, mean age 43.6. ±. 17.4 years) were evaluated. Two jejunal adenocarcinomas and one ileal neuro-endocrine tumour were diagnosed by enteroscopy (the diagnostic yield for malignancies in the selected population being 5.7%). In the non-celiac controls the detection rate of small bowel tumours by capsule endoscopy was 0.6% (P= 0.04). When compared to the registered population, the relative risk for intestinal malignancy was 1282 (95% CI, 407-4033; P