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First case of serrated lesion of the duodenal papilla in a patient with serrated colonic polyposis. A serrated polyposis syndrome was diagnosed in a 26-year-old female presenting with gastrointestinal symptoms. Screening for other lesions of the gastrointestinal tract showed a serpiginous looking papilla, described as possibly dysplastic. Histological analysis of biopsies showed a serrated lesion. This case describes the first known association between a duodenal serrated lesion and serrated polyposis syndrome. Upper GI screening is probably of little interest in this setting. In patients with upper GI serrated lesions, we recommend screening colonoscopy.