Skip to main content
Log in

Intraductal papillary neoplasm originating from an anomalous bile duct

  • Case Report
  • Published:
Clinical Journal of Gastroenterology Aims and scope Submit manuscript

Abstract

An 82-year-old woman who had been suffering from repeated obstructive jaundice for 7 years was referred to our hospital. Although endoscopic aspiration of the mucin in the common bile duct had been temporally effective, origin of the mucin production had not been detectable. The patient thus had been forced to be on long-term follow-up without curative resection. Endoscopic retrograde cholangioscopy on admission revealed massive mucin in the common bile duct. In addition, an anomalous bile duct located proximal to the gallbladder was identified. Since the lumen of the anomalous duct was irregular and the rest of biliary tree was completely free of suspicious lesions, the anomalous duct was judged to be the primary site. Surgical resection of the segment 4 and 5 of the liver combined with the extrahepatic biliary tract was performed. Pathological diagnosis was compatible to intraductal papillary neoplasm with high-grade intraepithelial dysplasia of the anomalous bile duct. The patient has been free from the disease for 6.5 years after resection. This is the first case of intraductal papillary neoplasm derived from an anomalous bile duct, which was resected after long-term conservative treatment. The present case suggested the slow growing character of natural history of the neoplasm.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3

Similar content being viewed by others

Abbreviations

IPNB:

Intraductal papillary neoplasm of the biliary duct

WHO:

World Health Organization

IPMN-P:

Intraductal papillary neoplasm of the pancreas

References

  1. Chen TC, Nakanuma Y, Zen Y, et al. Intraductal papillary neoplasms of the liver associated with heaptolithiasis. Hepatology. 2001;34:651–8.

    Article  CAS  PubMed  Google Scholar 

  2. Nakanuma Y, Curado M-P, Franceschi S, et al., editors. WHO classifyion of tumours of the digestive system. 4th ed. Lyon: WHO; 2010. p. 217–24.

    Google Scholar 

  3. Kubota K, Nakanuma Y, Kondo F, et al. Clinicopathological features and prognosis of mucin-producing bile duct tumor and mucinous cystic tumor of the liver: a multi-institutional study by the Japan Biliary Association. J Hepato-Biliary-Pancreat Sci. 2014;21:176–85.

    Article  Google Scholar 

  4. Gordon-Weeks AN, Jones K, Harriss E, et al. Systematic review and meta-analysis of current experience in treating IPNB: clinical and pathological correlates. Ann Surg. 2016;263(4):656–63.

    Article  PubMed  Google Scholar 

  5. Yeh T, Tseng J, Chiu C, et al. Cholangiographic spectrum of intraductal papillary mucinous neoplasm of the bile ducts. Ann Surg. 2006;244:248–53.

    Article  PubMed  PubMed Central  Google Scholar 

  6. Kim H, Kim M, Lee S, et al. Mucin-hypersecreting bile duct tumor characterized by a striking homology with an intraductal papillary mucinous tumor (IPMT) of the pancreas. Endoscopy. 2000;32:389–93.

    Article  CAS  PubMed  Google Scholar 

  7. Nakanuma Y, Zen Y, Harada K, et al. Tumorigenesis and phenotypic characteristics of mucin-producing bile duct tumors: an immunohistochemical approach. J Hepatobiliary Pancreat Sci. 2010;17:211–22.

    Article  PubMed  Google Scholar 

  8. Zen Y, Fujii T, Itatsu K, et al. Biliary papillary tumors share pathological features with intraductal papillary mucinous neoplasm of the pancreas. Hepatology. 2006;44:1333–43.

    Article  CAS  PubMed  Google Scholar 

  9. Rizzo R, Szucs R, Turner M. Congenital abnormalities of the pancreas and biliary tree in adults. Radiographics. 1995;15:49–68 (quiz 147-8).

    Article  CAS  PubMed  Google Scholar 

  10. Horaguchi J, Fujita N, Noda Y, et al. Choledochocele associated with superficial spreading cancer with cholesterolosis of the bile duct. J Gastroenterol. 2007;42:318–24.

    Article  PubMed  Google Scholar 

  11. Chirica M, Vullierme M, Sibert A, et al. Major hepatectomy for peripheral papillary cholangiocarcinoma with hilar extension in a patient with situs ambiguous. Gastroenterol Clin Biol. 2005;29:456–60.

    Article  PubMed  Google Scholar 

  12. Stanton R, Craig P, Jorgensen J, et al. Leakage via aberrant bile duct due to cholangiocarcinoma. HPB Surg. 1998;11:125–8.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  13. Schenk E, Findlay CJ. Intrahepatic duct system anomaly and hepatic duct carcinoma. Arch Pathol. 1963;75:453–8.

    CAS  PubMed  Google Scholar 

  14. Akiyama H, Kazitani T, Kuroda Y, et al. Bile duct carcinoma associated with an anomaly in the bile duct. Helv Chir Acta. 1979;45:813–6.

    CAS  PubMed  Google Scholar 

  15. Kagawa Y, Kashihara S, Kuramoto S, et al. Carcinoma arising in a congenitally dilated biliary tract. Report of a case and review of the literature. Gastroenterology. 1978;74:1286–94.

    CAS  PubMed  Google Scholar 

Download references

Acknowledgements

The authors thank Rumi Hino, Tetsuo Ushiku, and Masashi Fukayama at Department of Pathology, Graduate School of Medicine, The University of Tokyo, for their kind advice for histological diagnosis of the present case.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Norihiro Kokudo.

Ethics declarations

Conflict of interest:

All the authors declare that they have no conflict of interests.

Human/Animal Rights:

All procedures followed have been performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments.

Informed Consent:

Written informed consent was obtained from the patient for publication of this Case report and any accompanying images.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Maki, H., Aoki, T., Ishizawa, T. et al. Intraductal papillary neoplasm originating from an anomalous bile duct. Clin J Gastroenterol 10, 174–178 (2017). https://doi.org/10.1007/s12328-017-0721-8

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12328-017-0721-8

Keywords

Navigation