• Hyung Jun Kwon, Sang Geol Kim, and Jinyoung Park.
• Department of Surgery, School of Medicine, Kyungpook National University, Kyungpook National University Hospital, Daegu, South Korea.
• Medicine (Baltimore). 2023 Jun 16; 102 (24): e34080e34080.

RationaleAdenomyoma is a rare reactive, hamartomatous benign tumor-like lesion. Although adenomyoma can occur anywhere in the gastrointestinal tract, including the gallbladder, stomach, duodenum, and jejunum, it is very rarely observed in the extrahepatic bile duct and ampulla of Vater (AOV). The preoperative accurate diagnosis of adenomyoma of the Vaterian system, including the AOV and common bile duct, is significant to appropriate patient management. However, discriminating between benign and malignancy is highly challenging. Patients are frequently mistaken as having periampullary malignancy, thereby leading to unnecessary extensive surgical resection with a high risk of complications.Patient ConcernsA 47-year-old woman visited a local hospital owing to epigastric and right upper-quadrant abdominal pain for 2 days.DiagnosesAbdominal ultrasonography performed in the local hospital revealed suspicious of a distal common bile duct malignancy. She was transferred to our hospital for further evaluation and management.InterventionsAfter consulting with the patient, a multidisciplinary team, including a gastroenterologist, finally decided to perform surgery under the impression of an ampullary malignancy, and pylorus-preserving pancreatoduodenectomy was performed without any complications. She was histopathologically diagnosed with an adenomyoma of the AOV.OutcomesAt the 5-year follow-up, she was well and did not develop further symptoms or complications.LessonsAlthough adenomyoma is very rare, it should be included in the differential diagnosis of mass-like lesions of the AOV to avoid unnecessary surgeries.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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