Biliary papillomatosis – a rare cause of obstructive jaundice

Česká verzia

Authors: A. Buriánová ¹; J. Chalupa ²; O. Urban ³; J. Ürge ¹; V. Hořava Jr. ⁴; P. Vítek ²
Authors place of work: Interní oddělení, Nemocnice s poliklinikou Havířov, p. o. ¹; Beskydské gastrocentrum, Interní oddělení, Nemocnice ve Frýdku-Místku, p. o. ²; Centrum péče o zažívací trakt, Vítkovická nemocnice a. s., Ostrava ³; Oddělení patologie, Nemocnice ve Frýdku-Místku, p. o. ⁴
Published in the journal: Gastroent Hepatol 2015; 69(3): 204-210
Category: Digestive Endoscopy: Case Report
doi: https://doi.org/10.14735/amgh2015204

Summary

Biliary papillomatosis is a very rare disease, which is characterized by the presence of multiple papillomata involving the intrahepatic or extrahepatic biliary tract. Rarely it is also found in the pancreatic duct. Its symptoms include recurrent abdominal pain, dyspepsia and intermittent obstructive jaundice. Although it affects particularly older patients – men being affected twice as often as women – it has been also described in children. In addition to the risk of malignant reversal, the main risks for the patient include obstruction of the biliary or pancreatic duct and infectious complications. The diagnosis is based on a typical macroscopic appearance during cholangioscopy and microscopic evaluation of targeted samples. Treatment is individualized and includes mainly surgical procedures or drainage.

Case study:
We present two cases: An 80-year-old woman was hospitalized for recurrent biliary problems and obstructive jaundice. ERCP was performed with the introduction of the DB stent for stenosis between the common bile duct and common hepatic duct. The woman underwent a CT examination of the abdomen, endoscopic ultrasonography and cholangioscopy. The diagnosis of biliary papillomatosis was confirmed. Due to the patient’s age and overall condition, radical surgical treatment was contraindicated. Very soon, it was necessary to perform percutaneous transhepatic drainage. Two months after its introduction and one year after diagnosis, the woman died. The second patient, a 59-year-old woman was examined for asymptomatic dilatation of biliary duct on abdominal ultrasonography and elevated GGT level. She underwent endoscopic ultrasonography and ERCP with biopsy of the distal common bile duct. SpyGlass cholangioscopy was performed on the basis of histological examination (papillary adenoma) which revealed multiple papillomata of the common bile duct. She refused surgical treatment, and is now being observed in our gastroenterologic outpatient department.

Conclusion:
Biliary papillomatosis is among the unusual causes of biliary obstruction. Cholangiscopy with targeted sampling biopsy are the key methods of its diagnostic. This condition should always be considered in the context of differential diagnosis of benign biliary stenosis and cholangioscopy should be indicated soon. This condition with high malignant potential has a very poor prognosis; radical surgery is the only curative treatment. The increasing availability of cholangioscopy is likely to lead to a more frequent diagnosis of this disease.

Key words:
obstructive jaundice – cholangitis – abdominal pain – biliary tract neoplasms – cholestasis

The authors declare they have no potential conflicts of interest concerning drugs, products, or services used in the study.

The Editorial Board declares that the manuscript met the ICMJE „uniform requirements“ for biomedical papers.

Submitted:
9. 5. 2015

Accepted:
16. 5. 2015

Zdroje

1. Vassiliou I, Kairi-Vassilatou E, Marinis Aet al. Malignant potential of intrahepatic biliary papillomatosis: a case report and review of literature. World J Surg Oncol 2006; 4: 71.

2. Chappet V. Cancerepithelial primitif du canal choljdoque. Lyon Med 1894; 76: 145– 157.

3. Caroli J. Papillomas and papillomatoses of the common bile duct (in French). Rev Med Chir Mal Foie 1959; 34: 191– 230.

4. Yeung YP, AhChong K, Chung CK et al. Biliary paillomatosis: report of seven cases and review of English literature. J Hepatobiliary Pancreat Surg 2003; 10(8): 390– 395.

5. Chung DJ, Lee SK, Ha HK et al. Multiple biliary papillomatosis: comparison of MR cholangiography with endoscopic retrograde cholangiography. J Comput Assist Tomogr 2002; 26(6): 968– 974.

6. Lee SS, Kim MH, Lee SK et al. Clinicopathologic review of 58 patients with biliary papillomatosis. Cancer 2004; 100(4): 783– 793.

7. Zen Y, Fujii T, Itatsu K et al. Biliary papillary tumors share pathological features with intraductal papillary mucinous neoplasm of the pankreas. Hepatology 2006; 44(5): 1333– 1343.

8. Colombari R, Tsui WM. Biliary tumors of the liver. Semin Liver Dis 1995; 15(4): 402– 413.

9. Aoki S, Okayama Y, Kitajima Y et al. Intrahepatic biliary papilloma morphologically similar to biliary cystadenoma. J Gastroenterol Hepatol 2005; 20(2): 321– 324.

10. Mourra N, Hannoun L, Rousvoal G et al. Malignant intrahepatic biliary papillomatosis associated with viral C cirrhosis. Arch Pathol Lab Med 2002; 126(3): 369– 371.

11. Taguchi J, Yasunaga M, Kojiro M et al. Intrahepatic and extrahepatic biliary papillomatosis. Arch Pathol Lab Med 1993; 117(9): 944– 947.

12. Ma KF, Iu PP, Chau LF et al. Clinical and radiological features of biliary papillomatosis. Australas Radiol 2000; 44(2): 169– 173.

13. Levy AD, Murakata LA, Abbott RM et al. From the archives of the AFIP: benign tumors and tumorlike lesions of the gallbladder and extrahepatic bile ducts – radiologic/ patologic correlation. Armed Forces Institute of Pathology. Radiographics 2002; 22(2): 387– 413.

14. Hoang TV, Bluemke DA. Biliary papillomatosis: CT and MR findings. J Comput Assist Tomogr 1998; 22(4): 671– 672.

15. Kim YS, Myung SJ, Kim SY et al. Biliary papillomatosis: clinical, cholangiographic and cholangioscopic findings. Endoscopy 1998; 30(9): 763– 767.

16. Seo DW, Lee SK, Kim MH et al. Neoplasms of the biliary tree. In: Seo DW (ed). Cholangioscopy. Seoul, Korea: Koonja, Inc. 2002: 95– 98.

17. Chen TC, Nakanuma Y, Zen Y et al. Intraductal papillary neoplasia of the liver associated with hepatolithiasis. Hepatology 2001; 34(4 Pt 1): 651– 658.

18. Neumann RD, LiVolsi VA, Rosenthal NS et al. Adenocarcinoma in biliary papillomatosis. Gastroenterology 1976; 70 (5 Pt 1): 779– 782.

19. Holtkamp W, Reis HE. Papillomatosis of the bile ducts: papilloma-carcinoma sequence. Am J Gastroenterol 1994; 89(12): 2253– 2255.