Skip to main content
SpringerLink
Log in

A case of congenital biliary dilatation without pancreaticobiliary maljunction, so-called Type Ib according to Todani’s classification

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

Abstract

Congenital biliary dilatation (CBD) is a congenital malformation of focal dilatation of the extrahepatic bile ducts, including the common bile duct, and is often associated with pancreaticobiliary maljunction (PBM). In this article, we report a CBD case that presented with focal dilation of the common bile duct without PBM (Todani’s classification type Ib). The patient was a 32-year-old man who visited a doctor with a chief complaint of abdominal distension. Computed tomography revealed cystic dilatation of the common bile duct, and the patient was referred to our institution. Magnetic resonance cholangiopancreatography showed cystic dilatation of the common bile duct with a maximum diameter of 7 cm; however, evaluating the presence of PBM was challenging. Endoscopic ultrasonography showed small gallstones and debris in the dilated common bile duct and no thickening of the gallbladder wall. Endoscopic retrograde cholangiopancreatography revealed no PBM or markedly elevated bile amylase levels. Based on these findings, the patient was diagnosed with Todani Type Ib CBD. Since this patient did not have pancreatobiliary reflux, it was unclear whether the risk of developing biliary tract cancer was high, and since the treatment was highly invasive, the decision was to follow up without surgical treatment.

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

Access this article

Log in via an institution

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

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5

Similar content being viewed by others

References

  1. Alonso-Lej F, Rever WB Jr, Pessagno DJ. Congenital choledochal cysts, with a report of 2 and an analysis of 94 cases. Int Abstr Surg. 1959;108:1–30.

    CAS  PubMed  Google Scholar 

  2. Todani T, Watanabe Y, Narusue M, et al. Congenital bile duct cysts: classification, operative procedures, and review of thirty-seven cases including cancer arising from choledochal cyst. Am J Surg. 1977;134:263–9.

    Article  CAS  PubMed  Google Scholar 

  3. Todani T. Congenital choledochal dilatation: classification, clinical features, and long-term results. J Hep Bil Pancr Surg. 1997;4:276–82.

    Article  Google Scholar 

  4. Hamada Y, Ando H, Kamisawa T, et al. Diagnostic criteria for congenital biliary dilatation 2015. J Hepatobiliary Pancreat Sci. 2016;23:342–6.

    Article  PubMed  Google Scholar 

  5. Takauji S, Obara M, Koizumi K, et al. Case with Todani’s type Ib congenital cystic dilatation of the common bile duct. Nihon Shokakibyo Gakkai Zasshi. 2008;105:391–6 ((in Japanese)).

    PubMed  Google Scholar 

  6. Tashiro S, Imaizumi T, Ohkawa H, et al. Pancreaticobiliary maljunction: retrospective and nationwide survey in Japan. J Hepatobiliary Pancreat Surg. 2003;10:345–51.

    Article  PubMed  Google Scholar 

  7. Morine Y, Shimada M, Takamatsu H, et al. Clinical features of pancreaticobiliary maljunction: update analysis of 2nd Japan-nationwide survey. Hepatobiliary Pancreat Sci. 2013;20:472–80.

    Article  Google Scholar 

  8. Jesudason SR, Jesudason MR, Mukha RP, et al. Management of adult choledochal cysts–a 15-year experience. HPB (Oxford). 2006;8:299–305.

    Article  PubMed  Google Scholar 

  9. Morine Y, Mori H, Utsunomiya T, et al. Epidemiology and clinical features of pancreaticobiliary maljunction. 2011;25:133-40 (in Japanese with English abstract).

  10. Irie H, Honda H, Jimi M, et al. Value of MR cholangiopancreatography in evaluating choledochal cysts. AJR Am J Roentgenol. 1998;171:1381–5.

    Article  CAS  PubMed  Google Scholar 

  11. Park DH, Kim MH, Lee SK, et al. Can MRCP replace the diagnostic role of ERCP for patients with choledochal cysts? Gastrointest Endosc. 2005;62:360–6.

    Article  PubMed  Google Scholar 

  12. Suzuki M, Shimizu T, Kudo T, et al. Usefulness of nonbreath-hold 1-shot magnetic resonance cholangiopancreatography for the evaluation of choledochal cyst in children. J Pediatr Gastroenterol Nutr. 2006;42:539–44.

    Article  PubMed  Google Scholar 

  13. Huang CT, Lee HC, Chen WT, et al. Usefulness of magnetic resonance cholangiopancreatography in pancreatobiliary abnormalities in pediatric patients. Pediatr Neonatol. 2011;52:332–6.

    Article  PubMed  Google Scholar 

  14. Fumino S, Ono S, Kimura O, et al. Diagnostic impact of computed tomography cholangiography and magnetic resonance cholangiopancreatography on pancreaticobiliary maljunction. J Pediatr Surg. 2011;46:1373–8.

    Article  PubMed  Google Scholar 

  15. Itokawa F, Kamisawa T, Nakano T, et al. Exploring the length of the common channel of the pancreaticobiliary maljunction on magnetic resonance cholangiopancreatography. J Hepatobiliary Pancreat Sci. 2015;22:68–73.

    Article  PubMed  Google Scholar 

  16. Huang SG, Guo WL, Wang J, et al. Factors interfering with delineation on MRCP of pancreaticobiliary maljunction in paediatric patients. PLoS ONE. 2016;11: e0154178.

    Article  PubMed  PubMed Central  Google Scholar 

  17. Kamisawa T, Ando H, Suyama M, et al. Japanese clinical practice guidelines for pancreaticobiliary maljunction. J Gastroenterol. 2012;47:731–59.

    Article  PubMed  Google Scholar 

  18. Kamisawa T, Takuma K, Anjiki H, et al. Pancreaticobiliary maljunction. Clin Gastroenterol Hepatol. 2009;7:S84–8.

    Article  PubMed  Google Scholar 

  19. Fujii H, Yang Y, Tang R, et al. Epithelial cell proliferation activity of the biliary ductal system with congenital biliary malformations. Hepatobiliary Pancreat Surg. 1999;6:294–302.

    Article  CAS  Google Scholar 

  20. Hanada K, Itoh M, Fujii K, et al. Pathology and cellular kinetics of the gallbladder with anomalous junction of the pancreaticobiliary duct. Am J Gastroenterol. 1996;91:1007–11.

    CAS  PubMed  Google Scholar 

  21. Tanno S, Obara T, Fujii T, et al. Proliferative potential and K-ras mutation in epithelial hyperplasia of the gallbladder in patients with anomalous pancreaticobiliary ductal union. Cancer. 1998;83:267–75.

    Article  CAS  PubMed  Google Scholar 

  22. Sugai M, Ishido K, Endoh M, et al. Sonographic demonstration of the wall thickness of the gallbladder in pediatric patients with pancreatico-biliary maljunction. J Hepatobiliary Pancreat Sci. 2010;17:345–8.

    Article  PubMed  Google Scholar 

  23. Tsunoda T, Furui J, Yamada M, et al. Caroli’s disease associated with hepatolithiasis: a case report and review of the Japanese literature. Gastroenterol Jpn. 1991;26:74–9.

    Article  CAS  PubMed  Google Scholar 

  24. Millwala F, Segev DL, Thuluvath PJ. Caroli’s disease and outcomes after liver transplantation. Liver Transpl. 2008;14:11–7.

    Article  PubMed  Google Scholar 

  25. Mabrut JY, Kianmanesh R, Nuzzo G, et al. Surgical management of congenital intrahepatic bile duct dilatation, Caroli’s disease, and syndrome: long-term results of the French Association of Surgery Multicenter Study. Ann Surg. 2013;258:713–21.

    Article  PubMed  Google Scholar 

  26. Malhi H, Gores GJ. Cholangiocarcinoma: modern advances in understanding a deadly old disease. J Hepatol. 2006;45:856–67.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  27. Ishibashi H, Shimada M, Kamisawa T, et al. Japanese clinical practice guidelines for congenital biliary dilatation. J Hepatobiliary Pancreat Sci. 2017;24:1–16.

    Article  PubMed  Google Scholar 

Download references

Acknowledgements

The authors would like to thank Editage (www.editage.jp) for the English language editing.

Author information

Authors and Affiliations

  1. Department of Gastroenterology, Graduate School of Biomedical & Health Sciences, Hiroshima University, 1-2-3 Kasumi, Minami-Ku, Hiroshima, 734-8551, Japan

    Yusuke Kiyoshita, Yasutaka Ishii, Masahiro Serikawa, Shinya Nakamura, Juri Ikemoto, Yosuke Tamura, Sayaka Miyamoto, Kazuki Nakamura, Masaru Furukawa & Shiro Oka

Authors
  1. Yusuke Kiyoshita
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Yasutaka Ishii
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Masahiro Serikawa
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Shinya Nakamura
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Juri Ikemoto
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Yosuke Tamura
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Sayaka Miyamoto
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. Kazuki Nakamura
    View author publications

    You can also search for this author in PubMed Google Scholar

  9. Masaru Furukawa
    View author publications

    You can also search for this author in PubMed Google Scholar

  10. Shiro Oka
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Yasutaka Ishii.

Ethics declarations

Conflict of interest

The authors declare no conflicts of interest.

Human/animal rights

All procedures were performed per the ethical standards of the 1964 Declaration of Helsinki and its amendments.

Informed consent

The patient presented in this study provided informed consent.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Kiyoshita, Y., Ishii, Y., Serikawa, M. et al. A case of congenital biliary dilatation without pancreaticobiliary maljunction, so-called Type Ib according to Todani’s classification. Clin J Gastroenterol 17, 177–182 (2024). https://doi.org/10.1007/s12328-023-01873-z

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12328-023-01873-z

Keywords

Search

Navigation