Combination of Adenomyoma and Adenomyomatous Hyperplasia of the Ampullary System: A First Case Report

Abstract

Adenomyoma and adenomyomatous hyperplasia are benign tumor-like lesions that rarely involve the major or minor duodenal papilla. We report the case of a 73-year-old patient who underwent a cephalic duodenopancreatectomy due to clinical and radiological evidence of underlying malignant neoplasm. The histopathology results revealed the unusual association of a major duodenal papilla adenomyoma and an adenomyomatous hyperplasia of the minor papilla. Because of their resemblance to pancreatic malignancy, the diagnosis of these lesions is particularly challenging. In most cases, it is established postoperatively, after histopathological examination of the surgical specimen.

Adenomyoma of the ampulla of Vater mimicking malignancy: A case report and literature review

“…[1] Women have a 3-fold higher prevalence of adenomyoma than men, with a median patient age of 67 years. [2] The exact mechanism of the histogenesis of adenomyoma in the Vaterian system remains unclear. The most widely accepted hypothesis is that these lesions may represent a form of an incomplete heterotopic pancreas.…”

“…Another hypothesis is that the dysfunction of the muscle fibers of the AOV due to chronic papillitis may lead to muscular hyperplasia or adenomyomatous hyperplasia. [2] Adenomyoma is histologically characterized by a dense proliferation of ducts and ductules with smooth muscle bundles and collagen fibers. Multiple lobules of the glands are mainly located in the muscular layers of the Vaterian system, thereby resulting in the hypertrophy of the sphincter of Oddi.…”

“…Adenomyoma is a rare reactive, hamartomatous benign tumorlike lesion. [1,2] Although it 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 AOV. [3][4][5] Hammarström et al reported that of 3131 ERCP cases, 4 patients were diagnosed with ampullary adenomyoma, and the incidence of adenomyoma of the AOV was 0.13%.…”

“…Adenomyoma is a rare reactive, hamartomatous benign tumorlike lesion. [1,2] Although it 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). [3][4][5] The preoperative accurate diagnosis of adenomyoma of the Vaterian system, including the AOV and common bile duct (CBD), is significant to appropriate patient management.…”

Differentiation of Adenomyoma from Localized Adenocarcinoma of the Ampulla of Vater Using Multidetector CT

“…However, it has been known to rarely occur in the ampulla of Vater (AOV) ( 2 ). Regarding the ampullary location, fewer than 60 cases of adenomyoma involving the major papilla have been reported ( 2 3 4 ). However, small adenomyomas (2–5 mm) are common and reported in 60% of the population in autopsies, with no relevant associated clinical history ( 2 ).…”