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We conducted a study in order to assess the prevalence of extrapancreatic malignancies (EPMs) in a cohort of patients with intraductal papillary mucinous neoplasms (IPMNs) from northeastern Italy.A study was conducted in hospital Santa Maria della Misericordia, Udine, Italy. Hospital records were screened in order to identify newly diagnosed IPMN cases in the period from January 1, 2009, to December 31, 2015. We searched for EPMs diagnosed previous to, synchronous to, or after the IPMN. The ratio of the observed (O) number of patients with EPMs to the expected (E) was calculated.We identified 72 EPMs in 63 (31.8%) of 198 patients included. Among them, 51 (70.8%) were diagnosed previous to, 17 (23.6%) synchronous to, and 4 (5.6%) after the IPMN. Most frequently diagnosed were colorectal (12 patients [6.1%]), breast (8 patients [6.8%, in females]), renal cell (8 patients [4.0%]), and prostate cancer (7 patients [8.6%, in males]). The O/E ratios for EPMs were significantly increased for cancer in general (3.66; 95% confidence interval [CI], 2.39–5.37), renal cell (9.62; 95% CI, 1.98–28.10), prostate (4.91; 95% CI, 1.59–11.45), and breast cancer (3.16; 95% CI, 1.03–7.37).We report an increased risk of EPMs in patients with IPMN, especially for renal cell, prostate, colorectal, and breast cancer.