Peutz-Jeghers syndrome (PJS), is a rare autosomal dominant hereditary disorder. Intestinal hamartomatous polyps in association with melanocytic pigmentation of the skin and mucous membranes characterize PJS. Patients with PJS often have complications associated with the polyps, such as intestinal obstruction, intussusception, acute or chronic gastrointestinal bleeding, and the development of various types of cancer. Enteroclysis, small bowel follow-through, and push enteroscopy are generally used to identify the presence and location of small bowel polyps. Wireless capsule endoscopy (CE) has been confirmed as a feasible, safe, and sensitive test for the surveillance of small bowel polyps in patients with PJS and could replace radiographic small bowel surveillance. Here, a case of PJS newly diagnosed by CE is reported. The patient, a 30-year-old man who had developed rectal polyps associated with anal extrusion 7 years previously, had six episodes of hematochezia. (Intest Res 2010;8:181-186)