Mucin-Secreting Bile Duct Adenoma – Clinicopathological Resemblance to Intraductal Papillary Mucinous Tumor of the Pancreas

Abstract

We describe a case of mucin-secreting biliary adenoma associated with obstructive jaundice due to mucin production. Duodenal endoscopy revealed mucin secretion through an enlarged papilla of Vater. On the cholangiogram, large filling defects caused by mucin were observed in the dilated bile duct. Granular and/or papillary mucosa was noted from the hilar to the common bile duct by using percutaneous transhepatic cholangioscopy. Biopsy specimens taken from the papillary mucosa revealed slightly papillary projection without any evidence of malignancy. This lesion was diagnosed as mucin-secreting superficial adenoma and pancreatoduodenectomy was performed according to the malignant behavior. Using a histologic examination of the specimen, pure adenoma was documented. Since the surgical margin was positive, radiation was added for the residual adenoma in the intrahepatic bile ducts. The patient has been well without any evidence of recurrence for 21 months since her operation. It is only recently that the term ‘intraductal papillary mucinous tumor (IPMT)’, which is accepted as a distinct disease entity of pancreas, has begun to be used for mucin-secreting bile duct tumor. This case also seemed to clinicopathologically resemble an IPMT of the pancreas.

Biliary papillary tumors share pathological features with intraductal papillary mucinous neoplasm of the pancreas

“…28 Similarly, Adsay et al reported that two types of tumor progression could occur in IPMN-P: the MUC1-positive pathway to tubular adenocarcinoma and the MUC2-positive pathway to mucinous carcinoma. 29 There have been already several reports revealing close similarities between clinical, pathological and radiological characteristics of biliary papillary tumors and IPMN-P. 12,14,[30][31][32][33][34] This study, using a considerable number of biliary papillary tumors and IPMN-P, also showed sufficient evidence that biliary papilloma(tosis) and papillary-CC with or without mucus hypersecretion belong to a single tumor entity of intraductal papillary neoplasm of the bile duct, as a biliary counterpart of IPMN-P. Intraductal papillary neoplasm of the bile duct could manifest clinicopathologically as papilloma, papillomatosis and papillary adenocarcinoma (papillary-CC) (in situ and invasive). However, the distinction of papillary-CC in situ from biliary papilloma(tosis) is still controversial.…”

Carcinoma in situ arising in a tubulovillous adenoma of the distal common bile duct: A case report

“…Villous adenoma with mild dysplasia (n = 1), malignant foci (n = 4) M (n = 2) (mean) Chae, 1999 [12] M (n = 1) 77 Painless jaundice and pruritus Local excision Villous adenoma with malignant foci Inagaki, 1999 [13] M (n = 1) 73 Epigastric pain and jaundice Whipple Papilloma Chang, 2001 [14] M (n = 1) 51 Febrile jaundice, abdominal mass Operation refused Papilloma with focal dysplasia Oshikiri, 2002 [15] F (n = 1) 69 Jaundice Whipple Papilloma Ariche, 2002 [16] F (n = 1) 77 Abdominal pain Local excision Villous adenoma with adenocarcinoma Aggarwal, 2003 [17] M (n = 1) 55 Abdominal pain Whipple Adenoma with moderate dysplasia Jao, 2003 [18] M (n = [19] M (n = [20] M (n = 1) 74 Painless jaundice and pruritus Whipple Papilloma Katsinelos, 2006 [21] M (n = 1) 58 Painful jaundice Whipple Villous adenoma with atypia Xu, 2008 [22] rare. Adenomas arising from the CBD are summarized in Table 1.…”

“…Our patient did not exhibit any clinical signs of familial adenomatous polyposis [1] or Gardner's syndrome [2] . We also reviewed the literature about the common bile duct adenoma found in the English literature [3][4][5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22] (Table 1).…”

Bile tract adenomyoma: A case report

“…Therefore, according to the clinical presentation, biochemical, radiographic [13] , and endoscopic investigations, and intraoperative frozen section may not be able to confir m whether the tumor is benign or malignant, pancreaticoduodenectomy was sometimes necessary [2,[14][15][16] .…”