A case of hepatic intraductal papillary neoplasm of the bile duct
Article Sidebar
Main Article Content
Abstract
Introduction: Intraductal Papillary Neoplasm of the Bile ducts (IPNB) is a rare entity characterized by exophytic growth of the bile ducts.
Case presentation: In this report, we present a 57-year-old male with no prior medical history consulted for upper right abdominal pain, jaundice and pruritus. Abdominal Ultrasound (US) and magnetic resonance imaging/Bili-magnetic resonance were performed. They revealed that the circumferential parietal thickening of the common hepatic duct had extended approximately 4 cm with moderate dilatation of the left intrahepatic bile ducts. Computed tomography showed no evidence of distant metastasis.
Biopsy revealed a high-grade intraductal papillary neoplasm. After 40 days, the patient had left hepatectomy with resection of the main bile duct and the gallbladder.
Macroscopic examination of the surgical specimen showed a dilatation duct at the hilum with thickening of their walls.
The histopathology report revealed multiple intra-hepatic papillary neoplasms with high-grade dysplasia with an invasive carcinoma component in the left hepatic duct without extending to the biliary wall, classified as pT1N0.
Conclusion: This premalignant lesion has the potential to transform into invasive carcinoma if not properly diagnosed.
Our case illustrates how early identification can lead to potential surgical resection.
Article Details
Copyright (c) 2023 Beltaifa D, et al.
This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
The Archives of Case Reports is committed in making it easier for people to share and build upon the work of others while maintaining consistency with the rules of copyright. In order to use the Open Access paradigm to the maximum extent in true terms as free of charge online access along with usage right, we grant usage rights through the use of specific Creative Commons license.
License: Copyright © 2017 - 2025 | 
Open Access by Archives of Case Reports is licensed under a Creative Commons Attribution 4.0 International License. Based on a work at Heighten Science Publications Inc.
With this license, the authors are allowed that after publishing with the journal, they can share their research by posting a free draft copy of their article to any repository or website.
Compliance 'CC BY' license helps in:
| Permission to read and download | ✓ |
| Permission to display in a repository | ✓ |
| Permission to translate | ✓ |
| Commercial uses of manuscript | ✓ |
'CC' stands for Creative Commons license. 'BY' symbolizes that users have provided attribution to the creator that the published manuscripts can be used or shared. This license allows for redistribution, commercial and non-commercial, as long as it is passed along unchanged and in whole, with credit to the author.
Please take in notification that Creative Commons user licenses are non-revocable. We recommend authors to check if their funding body requires a specific license.
Guido M, Sarcognato S, Sacchi D, Ludwig K. The Anatomy and Histology of the Liver and Biliary Tract. In: D’Antiga L, editor. Pediatric Hepatology and Liver Transplantation. Cham: Springer International Publishing; 2019; 41–55. https://doi.org/10.1007/978-3-319-96400-3_3
Ohtsuka M, Shimizu H, Kato A, Yoshitomi H, Furukawa K, Tsuyuguchi T, Sakai Y, Yokosuka O, Miyazaki M. Intraductal papillary neoplasms of the bile duct. Int J Hepatol. 2014;2014:459091. doi: 10.1155/2014/459091. Epub 2014 May 18. PMID: 24949206; PMCID: PMC4052179.
Uemura S, Higuchi R, Yazawa T, Izumo W, Matsunaga Y, Shiihara M, Ota T, Furukawa T, Yamamoto M. Prognostic Factors for Surgically Resected Intraductal Papillary Neoplasm of the Bile Duct: A Retrospective Cohort Study. Ann Surg Oncol. 2021 Feb;28(2):826-834. doi: 10.1245/s10434-020-08835-6. Epub 2020 Jul 10. PMID: 32651697.
Matono R, Ninomiya M, Morita K, Tomino T, Oshiro Y, Yokota T, Nishizaki T. Branch-type intraductal papillary neoplasm of the bile duct treated with laparoscopic anatomical resection: a case report. Surg Case Rep. 2020 May 15;6(1):103. doi: 10.1186/s40792-020-00864-3. PMID: 32415464; PMCID: PMC7229076.
Pattarapuntakul T, Ovartlarnporn B, Sottisuporn J. Mucinous cystic neoplasm of the liver with extrahepatic growth presenting with ascending cholangitis diagnosed by endoscopic ultrasound features: a case report. J Med Case Rep. 2018 Feb 15;12(1):33. doi: 10.1186/s13256-017-1560-4. PMID: 29444709; PMCID: PMC5813420.
Yi D, Zhao LJ, Ding XB, Wang TW, Liu SY. Clinical characteristics of intrahepatic biliary papilloma: A case report. World J Clin Cases. 2021 May 6;9(13):3185-3193. doi: 10.12998/wjcc.v9.i13.3185. PMID: 33969107; PMCID: PMC8080758.
Arnaoutakis DJ, Kim Y, Pulitano C, Zaydfudim V, Squires MH, Kooby D, Groeschl R, Alexandrescu S, Bauer TW, Bloomston M, Soares K, Marques H, Gamblin TC, Popescu I, Adams R, Nagorney D, Barroso E, Maithel SK, Crawford M, Sandroussi C, Marsh W, Pawlik TM. Management of biliary cystic tumors: a multi-institutional analysis of a rare liver tumor. Ann Surg. 2015 Feb;261(2):361-7. doi: 10.1097/SLA.0000000000000543. PMID: 24509187; PMCID: PMC4655107.
Ying S, Ying M, Liang W, Wang Z, Wang Q, Chen F, Xiao W. Morphological classification of intraductal papillary neoplasm of the bile duct. Eur Radiol. 2018 Apr;28(4):1568-1578. doi: 10.1007/s00330-017-5123-2. Epub 2017 Nov 14. PMID: 29138880.
Nakanuma Y, Kakuda Y, Uesaka K. Characterization of Intraductal Papillary Neoplasm of the Bile Duct with Respect to the Histopathologic Similarities to Pancreatic Intraductal Papillary Mucinous Neoplasm. Gut Liver. 2019 Nov; 13(6):617–27. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6860037/
Park HJ, Kim SY, Kim HJ, Lee SS, Hong GS, Byun JH, Hong SM, Lee MG. Intraductal Papillary Neoplasm of the Bile Duct: Clinical, Imaging, and Pathologic Features. AJR Am J Roentgenol. 2018 Jul;211(1):67-75. doi: 10.2214/AJR.17.19261. Epub 2018 Apr 9. PMID: 29629808.
Luvira V, Somsap K, Pugkhem A, Eurboonyanun C, Luvira V, Bhudhisawasdi V. Morphological Classification of Intraductal Papillary Neoplasm of the Bile Duct with Survival Correlation. Asian Pac J Cancer Prev APJCP. 2017; 18(1):207–13. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5563102/
Schlitter AM, Born D, Bettstetter M, Specht K, Kim-Fuchs C, Riener MO, Jeliazkova P, Sipos B, Siveke JT, Terris B, Zen Y, Schuster T, Höfler H, Perren A, Klöppel G, Esposito I. Intraductal papillary neoplasms of the bile duct: stepwise progression to carcinoma involves common molecular pathways. Mod Pathol. 2014 Jan;27(1):73-86. doi: 10.1038/modpathol.2013.112. Epub 2013 Jul 5. PMID: 23828315.
Kim KM, Lee JK, Shin JU, Lee KH, Lee KT, Sung JY, Jang KT, Heo JS, Choi SH, Choi DW, Lim JH. Clinicopathologic features of intraductal papillary neoplasm of the bile duct according to histologic subtype. Am J Gastroenterol. 2012 Jan;107(1):118-25. doi: 10.1038/ajg.2011.316. Epub 2011 Sep 27. PMID: 21946282.
Onoe S, Shimoyama Y, Ebata T, Yokoyama Y, Igami T, Sugawara G. Prognostic delineation of papillary cholangiocarcinoma based on the invasive proportion: a single-institution study with 184 patients. Surgery. 2014 Feb; 155(2):280–91.
Nakanishi Y, Kondo S, Hirano S, Ambo Y, Tanaka E, Morikawa T. Recurrence of mucosal carcinoma of the bile duct, with superficial flat spread, 12 years after operation. J Hepatobiliary Pancreat Surg. 2006; 13(4):355–8. https://onlinelibrary.wiley.com/doi/abs/10.1007/s00534-006-1092-6