Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastrointest Surg. Nov 27, 2019; 11(11): 414-421
Published online Nov 27, 2019. doi: 10.4240/wjgs.v11.i11.414
Pediatric intrahepatic bile duct adenoma - rare liver tumor: A case report
Anup Kumar Roy, Nrityen Nath Das
Anup Kumar Roy, Department of Surgery, Downtown Hospital, Guwahati, Assam 781006, India
Nrityen Nath Das, Hepato-biliary and Liver Transplant Center, Downtown Hospital, Guwahati, Assam 781006, India
Author contributions: Roy AK and Das NN contributed equally to this work in all aspects, from conception to writing of the manuscript.
Informed consent statement: Informed consent was obtained from the patient.
Conflict-of-interest statement: To the best of our knowledge, no conflict of interest exists.
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Open-Access: This is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See:
Corresponding author: Anup Kumar Roy, MBBS, Doctor, Department of Surgery, Downtown Hospital, G. S. Road, Guwahati, Assam 781006, India.
Telephone: +91-96-5096-0564
Received: July 4, 2019
Peer-review started: July 16, 2019
First decision: August 2, 2019
Revised: September 7, 2019
Accepted: September 25, 2019
Article in press: September 25, 2019
Published online: November 27, 2019

Intrahepatic bile duct adenoma (BDA) is one of the rarest of the rare benign tumors of the liver in the pediatric age group. It arises from the epithelial lining of intrahepatic bile ducts. The overall incidence of BDA is 1.3% of all primary benign liver tumors. Few case reports of this rare tumor occurring in adult population are present in the literature and to date, only one pediatric case has been reported worldwide.


An 18-month-old male child presented with chief complaints of mass per abdomen for 8 mo. Computerized tomography abdomen revealed a well-defined exophytic solid tumor arising from the posteroinferior margin of the right lobe of the liver with heterogenous enhancement and cystic changes, suggesting a suspicion of hepatoblastoma. Non-anatomical liver resection was done and a tumor of 10 cm × 9.5 cm was excised. Histopathology of the specimen was conclusive with the diagnosis of intrahepatic bile duct adenoma, which was further supported by immunohistochemistry panel testing. The post-operative period was uneventful. On follow-up, the child was doing well and had no post-operative complication.


Intrahepatic bile duct adenoma in children is very rare and to date only one case has been reported. This is the second pediatric case of intrahepatic bile duct adenoma in the world. Additionally this is the first ever case of such a large tumor presenting in a child.

Keywords: Bile duct adenoma, Peribiliary gland hamartoma, Benign liver tumor, Pediatric liver tumor, Case report

Core tip: Intrahepatic bile duct adenoma is a rare tumor arising from bile ductules in the liver. It is mostly seen as an incidental finding in adults but it is almost unheard of in children. Therefore it is one of the rarest tumors in children. Although it is a benign tumor, it has the potential to be malignant according to one study reported in adults. This case highlights the fact that it is the second case of this rare tumor occurring in the pediatric age group in the world. The progression into a large tumor in our case report necessitates the need for more information on this rare tumor.