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Both ectopic pancreas and insulinoma are rare. A 21-year-old male patient had suffered from hypoglycemia episodes for 3 years and was diagnosed to have insulinoma based on biochemical and endocrinological evaluations. Various localization approaches revealed a distinct tumor outside the pancreas. With intraoperative endoscopy, the tumor in duodenohepatic ligament was identified and successfully resected. Pathologic evaluation revealed an ectopic insulinoma with ectopic pancreas. Therefore, when the biochemically confirmed insulinoma could not be well-definitely localized, the possibility of ectopic insulinoma should be suspected. In addition, radiography examinations and operation exploration should extend to the field where ectopic pancreas usually presents.