Abstract
A 59-year-old male with history of sigmoid colon cancer had a high serum-CEA level and was referred for the evaluation of metastatic liver disease. Ultrasonography and computerized tomography showed two tumours in the liver. Macroscopically, these were in segment 4 (S4) and 2 (S2). Histologically, the tumour in S4 showed a number of bile ductules with variable amounts of stroma, an appearance compatible with bile duct adenoma (BDA). There were markedly atypical ductules of various sizes, the epithelium of which had coarsely granular/hyperchromatic large nuclei, in some areas of the lesion. These atypical ductules showed invasive growth into the liver parenchyma. Some cystically dilated ductules with bile plugs resembling bile duct hamartoma (BDH) were also seen. The other tumour in S2, was a metastatic adenocarcinoma from sigmoid colon and showed strongly positive staining for CEA. Since the lesion in S4 of our case is solitary and most of histological features are similar to those of BDA with markedly atypical bile ductules, we consider that this may be the first case of cholangiocarcinoma associated with BDA with focal area of BDH. It is possible that the adenoma-carcinoma sequence occurs in biliary tumours.
This is a preview of subscription content, log in via an institution to check access.
Similar content being viewed by others
References
Allaire GS, Rabin L, Ishak KG, Sesternhenn IA (1988) Bile duct adenoma. A study of.152 cases. Am J Surg Pathol 12:708–715
Burns CD, Kuhns JG, Wieman TJ (1990) Cholangiocarcinoma in association with multiple biliary microhamartomas. Arch Pathol Lab Med 114:1287–1289
Dekker A, Ten Kate FJW, Terpstra OT (1990) Cholangiocarcinoma associated with multiple bileduct hamartomas of the liver. Dig Dis Sci 34:952–958
Edmondson HA (1958) Tumors of the liver and intrahepatic bile ducts. Atlas of tumor pathology. 1st ser, fasc 25, 18–24. Armed Forces Institute of Pathology, Washington, DC, pp 179–190
Foucar E, Kaplan LR, Gold JH, Kiang DT, Sibley RK, Bosl G (1979) Well-differentiated peripheral cholangiocarcinoma with an unusual clinical course. Gastroenterology 77:347–353
Govindarajan S, Peters RL (1984) The bile duct adenoma. A lesion distinct from Meyenburg complex. Arch Pathol Lab Med 108:922–924
Honda N, Cobb C, Lechago J (1986) Bile duct carcinoma associated with multiple von Meyenburg complexes in the liver. Hum Pathol 17:1287–1290
Hsu SM, Raine L, Fanger H (1981) The use of avidin-biotinperoxidase complex (ABC) in immunoperoxidase technique: a comparison between ABC and unlabelled antibody (PAP) procedure. J Histochem Cytochem 29:577–580
Imray CHE, Newbold KM, Davis A, Lavelle-Jones M, Neoptolemos JP (1992) Induction of cholangiocarcinoma in the golden Syrian hamster using methylazoxymethyl acetate. Eur J Surg Oncol 18:373–378
Ishak KG, Rabin L (1975) Benign tumours of the liver. Med Clin North Am 59:995–1013
Mathews MB, Bernstein RM, Franza BR, Garrels JI (1984) Identity of the proliferating cell nuclear antigen and cyclin. Nature 303:374–376
Matsuno Y, Hirohashi S, Furuya S, Sakamoto M, Mukai K, Shimosato Y (1990) Heterogeneity of proliferative activity in nodule-in-nodule lesions of small hepatocellular carcinoma. Jpn J Cancer Res 81:1137–1140
Miyachi K, Fritzler MJ, Tan EM (1978) Autoantibodies to a nuclear antigen in proliferation cells. J Immunol 121:2228–2234
Ohta T, Nagakawa T, Ueda N, Nakmura T, Akiyama T, Ueno K, Miyazaki I (1991) Mucosal dysplasia of the liver and the intraductal variant of peripheral cholangiocarcinoma in hepatholithiasis. Cancer 68:2217–2223
Ona FV, Dytoc JT (1991) Clonorchis-associated cholangiocarcinoma: a report of two cases with unusual manifestations. Gastroenterology 101:831–839
Peosi G, Zamboni G, Doglioni C, Rodella S, Bresaola E, Iacono C, Serio G, Iannucci A, Scarpa A (1992) Immunodetection of proliferating cell nuclear antigen assesses the growth fraction and predicts malignancy in endocrine tumors of the pancreas. Am J Surg Pathol 16:1215–1225
Srivatanakul P, Parkin M, Jiang Y-Z, Khlat M, Kao-Ian UT, Sontipong S, Wild C (1991a) The role of infection by Opisthorchis viverrini, hepatitis B virus and aflatoxin exposure in the etiology of liver cancer in Thailand. A correlation study. Cancer 68:2411–2417
Srivatanakul P, Ohshima H, Khlat M, Parkin M, Sukaryodhin S, Brouet I, Bartsch H (1991b) Opisthorchis viverrini infestation and endogenous nitrosamines as risk factors for cholangiocarcinoma in Thailand. Int J Cancer 48:821–825
Author information
Authors and Affiliations
Rights and permissions
About this article
Cite this article
Hasebe, T., Mukai, K., Sakamoto, M. et al. Cholangiocarcinoma arising in bile duct adenoma with focal area of bile duct hamartoma. Vichows Archiv A Pathol Anat 426, 209–213 (1995). https://doi.org/10.1007/BF00192644
Received:
Accepted:
Issue Date:
DOI: https://doi.org/10.1007/BF00192644