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Celiac disease (CD) has been established as being associated with several thyroid diseases. However, occurrence of thyroid epithelial cancer in celiac patients has been rarely described.We describe the prevalence of thyroid carcinoma detected in a cohort of Italian celiac patients, with the aim to carry out a prospective analysis of the risk of celiac patients to develop thyroid carcinoma.The study population included all CD patients diagnosed at the Collaborating Centers of the Italian Registry of CD between January 1, 1982 and December 31, 2006. Upon diagnosis of CD and at every subsequent clinical control, the Collaborating Centers filled in a validated form for each CD patient reporting information about demographic data, possible occurrence of a thyroid disease, and adherence to a gluten-free diet.Of the 1757 celiac patients enrolled, 6 developed a papillary thyroid carcinoma during the follow-up period (mean: 18.1 y). The Standardized Incidence Ratio resulted 2.55 (95% confidence interval=0.93-5.55; P<0.01). The mean age of diagnosis of CD in patients who developed thyroid carcinoma was rather low (40 y) and not statistically different from that of those who did not develop thyroid carcinoma. The number of gluten-containing diet per month did not correlate with the development of a thyroid carcinoma.There is a 2.5-fold increased risk of papillary cancer of thyroid for celiac patients. A prompt diagnosis of CD and a strict adherence to gluten-free diet do not seem to protect from the development of this malignancy.