Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Sep 28, 2019; 25(36): 5569-5577
Published online Sep 28, 2019. doi: 10.3748/wjg.v25.i36.5569
Long-term growth of intrahepatic papillary neoplasms: A case report
Takumu Hasebe, Koji Sawada, Hidemi Hayashi, Shunsuke Nakajima, Hiroyuki Takahashi, Masahiro Hagiwara, Koji Imai, Sayaka Yuzawa, Mikihiro Fujiya, Hiroyuki Furukawa, Toshikatsu Okumura
Takumu Hasebe, Koji Sawada, Hidemi Hayashi, Shunsuke Nakajima, Mikihiro Fujiya, Toshikatsu Okumura, Department of Medicine, Division of Gastroenterology and Hematology/Oncology, Asahikawa Medical University, Asahikawa 0788510, Hokkaido, Japan
Hiroyuki Takahashi, Masahiro Hagiwara, Koji Imai, Hiroyuki Furukawa, Department of Surgery, Division of Gastroenterological and General Surgery, Asahikawa Medical University, Asahikawa 0788510, Hokkaido, Japan
Sayaka Yuzawa, Department of Diagnostic Pathology, Asahikawa Medical University Hospital, Asahikawa 0788510, Hokkaido, Japan
Author contributions: Hasebe T, Sawada K, Hayashi H and Nakajima S performed the preoperative evaluation and diagnosis; Takahashi H, Hagiwara M, Imai K and Furukawa H performed surgical resection of the tumor; Yuzawa S pathologically diagnosed the tissue; Hasebe T, Sawada K, Fujiya M and Okumura T reviewed the literature and contributed to drafting the manuscript; and all authors issued final approval for the submitted version.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared according to the CARE Checklist (2016).
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: Takumu Hasebe, MD, PhD, Doctor, Department of Medicine, Division of Gastroenterology and Hematology/Oncology, Asahikawa Medical University, 2-1-1-1 Midorigaoka Higashi, Asahikawa 0788510, Hokkaido, Japan.
Telephone: +81-166-682462 Fax: +81-166-682469
Received: July 17, 2019
Peer-review started: July 17, 2019
First decision: August 18, 2019
Revised: August 29, 2019
Accepted: September 11, 2019
Article in press: September 11, 2019
Published online: September 28, 2019

Intraductal papillary neoplasm of the bile duct (IPNB) is a type of tumor that presents in the intra- or extrahepatic bile ducts. Cystic-type intrahepatic IPNB often mimics simple liver cysts, making the diagnosis difficult. Because the growth of IPNB is slow, careful follow-up and timely therapeutic intervention is recommended. There are few reports with a follow-up period longer than a decade; thus, we report the case of a patient with an IPNB that grew for over 13 years.


A 65-year-old man was diagnosed, 13 years prior with a cystic hepatic tumor with abnormal imaging findings. The targeted tumor biopsy results showed no malignancy. Biannual follow-up examinations were performed because of the potential for malignancy. The cystic lesions showed gradual enlargement over 11 years and a 4 mm papillary proliferation appeared on the cyst wall, which is compatible with IPNB. The tumor was observed for another 2 years because of the patient’s wishes. The imaging findings showed enlargement to 8 mm and a new 9 mm papillary proliferation of the cystic tumor. Contrast-enhanced ultrasonography showed hyperenhancement during the arterial phase in both cyst walls, indicating intraductal tumor progression in both tumors. Thus, liver segment 8 subsegmentectomy was performed. The pathological findings indicated that the tumors contained mucin, and high-grade atypia was observed in the papillary lesions, showing IPNB.


The development of IPNB should be monitored in patients with cystic lesions and ultrasonography are useful tool for the evaluation.

Keywords: Bile duct neoplasm, Mucin, Disease progression, Ultrasonography, Perfluorobutane, Case report

Core tip: Cystic lesions in the liver, such as intraductal papillary neoplasms of the bile duct, rarely yield malignant tumors; additionally, the development of a tumor over a decade is hardly ever observed. Here, we present the case of a patient with an intrahepatic intraductal papillary neoplasm of the bile duct that developed over 13 years from a cystic tumor of the liver. The tumor was diagnosed by contrast-enhanced ultrasonography with perfluorobutane and was completely surgically resected. Notably, the intracystic spread of the tumor was well represented by contrast-enhanced ultrasonography.