Ann Liver Transplant.  2022 May;2(1):95-101. 10.52604/alt.22.0007.

Pancreaticoduodenectomy for recurrence of intraductal papillary neoplasm of bile duct at seven years after curative resection

Affiliations
  • 1Department of Surgery, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea

Abstract

Intraductal papillary neoplasm of bile duct (IPNB) is a rare variant of bile duct tumor with a wide range of histology from low-grade dysplasia to invasive carcinoma. A high-grade IPNB can be regarded as a malignant lesion because of potential tumor recurrence after curative resection. We herein report a case of IPNB recurrence at seven years after curative resection of IPNB with a high-grade dysplasia. A 63-yearold male patient underwent right anterior sectionectomy for IPNB with a high-grade dysplasia after close observation for four years. Postoperatively, recurrent IPNB lesion in the distal bile duct was diagnosed at postoperative seven years. Because the mass was localized without metastasis, pylorus-preserving pancreaticoduodenectomy was performed for the recurrent IPNB. The patient recovered uneventfully without specific complications. He has been doing well for one year without any evidence of recurrence. The recurrent IPNB lesion was a low-grade dysplasia, which was slightly different from the initial IPNB. Immunohistochemical status of MUC1/MUC2 stain was identical, indicating that the IPNB at the distal common bile duct was a recurrent lesion of the initial intrahepatic IPNB. The present case indicates that long-term follow-up is necessary for patients after undergoing curative resection for IPNB of low- or high-grade dysplasia, although the risk of tumor recurrence is not high. Aggressive surgical treatment for recurrent IPNB lesion is a viable option to achieve long-term survival after prudent consideration of postoperative recurrence pattern.

Keyword

Intraductal papillary neoplasm of bile duct; Recurrence; Dysplasia; Hepatectomy; Pancreaticoduodenectomy

Figure

  • Figure 1 Radiologic findings at initial symptom presentation. Biliary dynamic computed tomography (A) and magnetic resonance imaging with cholangiography (B, C) showed mild diffuse biliary dilatation and focal dilatation of a segment VIII branch (arrow). Endoscopic retrograde cholangiopancreatography (D) showed mild dilatation of the common hepatic duct and intrahepatic ducts without noticeable stricture.

  • Figure 2 Follow-up radiologic findings after four years. Liver dynamic computed tomography (A, B) showed slight worsening of the segment VIII duct dilatation with suspicious intraductal soft tissue lesions (arrow) and a separate 3 cm-sized mass in the right hepatic dome (arrowhead). Magnetic resonance imaging with cholangiography (C, D) showed significantly increased extent of the intraductal papillary lesion (arrow).

  • Figure 3 Microphotographs of the resected specimen. Intraductal papillary mass was diagnosed as intraductal papillary neoplasm (A: H&E stain, ×50), showing high-grade dysplasia (B: H&E stain, ×200). Immunohistochemical stain showed focal positivity for MUC2 (C: ×400).

  • Figure 4 Gross photographs of the resected specimen after right anterior sectionectomy showing intraductal papillary lesions (arrow) and liver abscess (arrowhead).

  • Figure 5 Follow-up liver dynamic computed tomography. Images taken at one month after hepatectomy (A) showed only mild intrahepatic duct dilatation, which was worsened after seven years (B).

  • Figure 6 Preoperative imaging studies taken at seven years and eight months after hepatectomy. A 2 cm-sized highly attenuating mass in the distal common bile duct (arrows) was identified on computed tomography (A) and magnetic resonance imaging with cholangiography (B, C). Fluorodeoxyglucose-positron emission tomography showed focal hypermetabolic uptake (arrow) of the intraductal mass (D).

  • Figure 7 Gross photographs of the resected specimen after pylorus-preserving pancreaticoduodenectomy. Intraductal papillary mass was identified (arrows) at the distal common bile duct (A) with preservation of the proximal resection margin (B).

  • Figure 8 Follow-up liver computed tomography taken at one year after pancreaticoduodenectomy. There was no interval change of mild intrahepatic duct dilatation (A). There was no abnormal finding at the pancreaticoduodenectomy site (B).


Reference

1. Nakanuma Y, Sato Y, Harada K, Sasaki M, Xu J, Ikeda H. 2010; Pathological classification of intrahepatic cholangiocarcinoma based on a new concept. World J Hepatol. 2:419–427. DOI: 10.4254/wjh.v2.i12.419. PMID: 21191517. PMCID: PMC3010511.
2. Jang KT, Hong SM, Lee KT, Lee JG, Choi SH, Heo JS, et al. 2008; Intraductal papillary neoplasm of the bile duct associated with Clonorchis sinensis infection. Virchows Arch. 453:589–598. DOI: 10.1007/s00428-008-0682-x. PMID: 18855009.
3. Itatsu K, Zen Y, Ohira S, Ishikawa A, Sato Y, Harada K, et al. 2007; Immunohistochemical analysis of the progression of flat and papillary preneoplastic lesions in intrahepatic cholangiocarcinogenesis in hepatolithiasis. Liver Int. 27:1174–1184. DOI: 10.1111/j.1478-3231.2007.01577.x. PMID: 17919228.
4. Zen Y, Fujii T, Itatsu K, Nakamura K, Minato H, Kasashima S, et al. 2006; Biliary papillary tumors share pathological features with intraductal papillary mucinous neoplasm of the pancreas. Hepatology. 44:1333–1343. DOI: 10.1002/hep.21387. PMID: 17058219.
5. Nakanuma Y. 2010; A novel approach to biliary tract pathology based on similarities to pancreatic counterparts: is the biliary tract an incomplete pancreas? Pathol Int. 60:419–429. DOI: 10.1111/j.1440-1827.2010.02543.x. PMID: 20518896.
6. Rocha FG, Lee H, Katabi N, DeMatteo RP, Fong Y, D'Angelica MI, et al. 2012; Intraductal papillary neoplasm of the bile duct: a biliary equivalent to intraductal papillary mucinous neoplasm of the pancreas? Hepatology. 56:1352–1360. DOI: 10.1002/hep.25786. PMID: 22504729.
7. WHO Classification of Tumours Editorial Board. 2019; WHO classification of tumours. Vol I. Digestive system tumours. 5th ed. Lyon:. IARC,. 279–282.
8. Youn JM, Hwang S, Ahn CS, Moon DB, Ha TY, Song GW, et al. DOI: 10.1007/s11605-022-05268-2. PMID: 35141839.
9. Wu SD, Lu CD, Lu CJ, Huang J, Zhou J. 2010; Mucin-producing intrahepatic biliary papillomatosis. Surg Today. 40:845–850. DOI: 10.1007/s00595-009-4132-4. PMID: 20740348.
10. Nakanuma Y, Zen Y, Harada K, Ikeda H, Sato Y, Uehara T, et al. 2010; Tumorigenesis and phenotypic characteristics of mucin-producing bile duct tumors: an immunohistochemical approach. J Hepatobiliary Pancreat Sci. 17:211–222. DOI: 10.1007/s00534-009-0158-7. PMID: 19680592.
11. Wan XS, Xu YY, Qian JY, Yang XB, Wang AQ, He L, et al. 2013; Intraductal papillary neoplasm of the bile duct. World J Gastroenterol. 19:8595–8604. DOI: 10.3748/wjg.v19.i46.8595. PMID: 24379576. PMCID: PMC3870504.
12. Park HJ, Kim SY, Kim HJ, Lee SS, Hong GS, Byun JH, et al. 2018; Intraductal papillary neoplasm of the bile duct: clinical, imaging, and pathologic features. AJR Am J Roentgenol. 211:67–75. DOI: 10.2214/AJR.17.19261. PMID: 29629808.
13. Yeh CN, Jan YY, Yeh TS, Hwang TL, Chen MF. 2004; Hepatic resection of the intraductal papillary type of peripheral cholangiocarcinoma. Ann Surg Oncol. 11:606–611. DOI: 10.1245/ASO.2004.04.028. PMID: 15172934.
14. Onoe S, Shimoyama Y, Ebata T, Yokoyama Y, Igami T, Sugawara G, et al. 2014; Prognostic delineation of papillary cholangiocarcinoma based on the invasive proportion: a single-institution study with 184 patients. Surgery. 155:280–291. DOI: 10.1016/j.surg.2013.08.011. PMID: 24287144.
15. Barton JG, Barrett DA, Maricevich MA, Schnelldorfer T, Wood CM, Smyrk TC, et al. 2009; Intraductal papillary mucinous neoplasm of the biliary tract: a real disease? HPB (Oxford). 11:684–691. DOI: 10.1111/j.1477-2574.2009.00122.x. PMID: 20495637. PMCID: PMC2799622.
16. Kim KM, Lee JK, Shin JU, Lee KH, Lee KT, Sung JY, et al. 2012; Clinicopathologic features of intraductal papillary neoplasm of the bile duct according to histologic subtype. Am J Gastroenterol. 107:118–125. DOI: 10.1038/ajg.2011.316. PMID: 21946282.
17. Jung G, Park KM, Lee SS, Yu E, Hong SM, Kim J. 2012; Long-term clinical outcome of the surgically resected intraductal papillary neoplasm of the bile duct. J Hepatol. 57:787–793. DOI: 10.1016/j.jhep.2012.05.008. PMID: 22634127.
18. Yeh TS, Tseng JH, Chiu CT, Liu NJ, Chen TC, Jan YY, et al. 2006; Cholangiographic spectrum of intraductal papillary mucinous neoplasm of the bile ducts. Ann Surg. 244:248–253. DOI: 10.1097/01.sla.0000217636.40050.54. PMID: 16858187. PMCID: PMC1602176.
Full Text Links
  • ALT
Actions
Cited
CITED
export Copy
Close
Share
  • Twitter
  • Facebook
Similar articles
Copyright © 2025 by Korean Association of Medical Journal Editors. All rights reserved.     E-mail: koreamed@kamje.or.kr