Malignant transformation of intraductal papillary neoplasm of the bile duct following non-standardized biliary-enteric drainage for hepatolithiasis: two case reports and literature review
- 1Department of Hepatobiliary Surgery, Hunan Provincial People’s Hospital/The First Affiliated Hospital of Hunan Normal University
- 2Department of Oncology, Hunan Provincial People’s Hospital/The First Affiliated Hospital of Hunan Normal University
Abstract
Background: Intraductal papillary neoplasm of the bile duct (IPNB) is a rare biliary epithelial neoplasm with malignant potential. It is characterized by intraductal papillary growth, variable mucin production, and a close association with chronic biliary inflammation.
Case presentation: We describe two patients with hepatolithiasis who developed IPNB after non-standardized biliary-enteric drainage without complete common bile duct transection. Both patients had long-term enterobiliary reflux and chronic cholangitis. Case 1 was diagnosed with intestinal-type IPNB with high-grade intraepithelial neoplasia and focal invasive mucinous adenocarcinoma and remained recurrence-free at 12 months despite a microscopically positive ductal margin (R1 resection). Case 2 had pancreatobiliary-type IPNB with moderately to poorly differentiated invasive adenocarcinoma and developed rapid postoperative peritoneal and distant metastases.
Conclusion: In these two patients with hepatolithiasis, non-standardized biliary-enteric drainage with a retained distal common bile duct and blind pouch was associated with long-term enterobiliary reflux, chronic cholangitis, and subsequent IPNB. These findings suggest a plausible contributing mechanism rather than an established causal relationship. When biliary-enteric reconstruction is otherwise indicated, surgical techniques that avoid a residual blind pouch and minimize enterobiliary reflux should be considered. Long-term surveillance may be warranted in patients with long-standing biliary inflammation after non-standardized drainage.
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BibTeX
@article{peng2026malignant,
title = {{Malignant transformation of intraductal papillary neoplasm of the bile duct following non-standardized biliary-enteric drainage for hepatolithiasis: two case reports and literature review}},
author = {Jinpeng Peng and Pingzhou Yang and Hao Huang and Chuang Peng and Zhiguo Tan},
journal = {Frontiers in Oncology},
year = {2026},
month = oct,
doi = {10.3389/fonc.2026.1980694},
url = {https://www.frontiersin.org/articles/10.3389/fonc.2026.1980694/full},
note = {CC BY 4.0},
}
RIS
TY - JOUR
TI - Malignant transformation of intraductal papillary neoplasm of the bile duct following non-standardized biliary-enteric drainage for hepatolithiasis: two case reports and literature review
AU - Jinpeng Peng
AU - Pingzhou Yang
AU - Hao Huang
AU - Chuang Peng
AU - Zhiguo Tan
PY - 2026
DA - 2026/10/26
JO - Frontiers in Oncology
DO - 10.3389/fonc.2026.1980694
UR - https://www.frontiersin.org/articles/10.3389/fonc.2026.1980694/full
AB - Background: Intraductal papillary neoplasm of the bile duct (IPNB) is a rare biliary epithelial neoplasm with malignant potential. It is characterized by intraductal papillary growth, variable mucin production, and a close association with chronic biliary inflammation. Case presentation: We describe two patients with hepatolithiasis who developed IPNB after non-standardized biliary-enteric drainage without complete common bile duct transection. Both patients had long-term enterobiliary reflux and chronic cholangitis. Case 1 was diagnosed with intestinal-type IPNB with high-grade intraepithelial neoplasia and focal invasive mucinous adenocarcinoma and remained recurrence-free at 12 months despite a microscopically positive ductal margin (R1 resection). Case 2 had pancreatobiliary-type IPNB with moderately to poorly differentiated invasive adenocarcinoma and developed rapid postoperative peritoneal and distant metastases. Conclusion: In these two patients with hepatolithiasis, non-standardized biliary-enteric drainage with a retained distal common bile duct and blind pouch was associated with long-term enterobiliary reflux, chronic cholangitis, and subsequent IPNB. These findings suggest a plausible contributing mechanism rather than an established causal relationship. When biliary-enteric reconstruction is otherwise indicated, surgical techniques that avoid a residual blind pouch and minimize enterobiliary reflux should be considered. Long-term surveillance may be warranted in patients with long-standing biliary inflammation after non-standardized drainage.
SN - 2234-943X
ER -