Asano F, Matsuyama R, Kumamoto T, Shinkai M, Morioka D, Shinoda S, Endo I. Clinical characteristics of and risk factors for hepatolithiasis developed after surgery for congenital biliary dilatation. World J Gastrointest Surg 2026; 18(1): 113350 [DOI: 10.4240/wjgs.v18.i1.113350]
Corresponding Author of This Article
Fumio Asano, MD, Department of Gastroenterological Surgery, Yokohama City University Graduate School of Medicine, 3-9 Fukuura, Kanazawa Ward, Yokohama 236-0004, Kanagawa, Japan. t136001f@yokohama-cu.ac.jp
Research Domain of This Article
Gastroenterology & Hepatology
Article-Type of This Article
Retrospective Study
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This article is an open-access article which 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: http://creativecommons.org/licenses/by-nc/4.0/
Jan 27, 2026 (publication date) through Jan 28, 2026
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Publication Name
World Journal of Gastrointestinal Surgery
ISSN
1948-9366
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Baishideng Publishing Group Inc, 7041 Koll Center Parkway, Suite 160, Pleasanton, CA 94566, USA
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Asano F, Matsuyama R, Kumamoto T, Shinkai M, Morioka D, Shinoda S, Endo I. Clinical characteristics of and risk factors for hepatolithiasis developed after surgery for congenital biliary dilatation. World J Gastrointest Surg 2026; 18(1): 113350 [DOI: 10.4240/wjgs.v18.i1.113350]
Fumio Asano, Ryusei Matsuyama, Takafumi Kumamoto, Daisuke Morioka, Itaru Endo, Department of Gastroenterological Surgery, Yokohama City University Graduate School of Medicine, Yokohama 236-0004, Kanagawa, Japan
Masato Shinkai, Department of Surgery, Kanagawa Children’s Medical Center, Yokohama 232-8555, Kanagawa, Japan
Satoru Shinoda, Department of Biostatistics, Yokohama City University School of Medicine, Yokohama 236-0004, Kanagawa, Japan
Author contributions: Asano F, Matsuyama R, and Morioka D wrote the manuscript; Asano F, Matsuyama R and Endo I participated in the conception and design of the study; Asano F, Kumamoto T, and Shinkai M were involved in data acquisition; Asano F and Shinoda S were involved in the statistical analysis of the data; all the authors participated in the interpretation of the data, and all the authors were responsible for the decision to submit the manuscript for publication.
Institutional review board statement: This investigation was approved by Institutional Ethics Committee of Yokohama City University, No. F241000006 (same in the two university hospitals) and Institutional Ethics Committee of Kanagawa Children’s Medical Center, No. C119-2024-92.
Informed consent statement: The need for patient consent was waived because of the retrospective nature of the study.
Conflict-of-interest statement: The authors declare that there are no conflicts of interest for this article.
Data sharing statement: No additional data are available.
Open Access: This article 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 NonCommercial (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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Fumio Asano, MD, Department of Gastroenterological Surgery, Yokohama City University Graduate School of Medicine, 3-9 Fukuura, Kanazawa Ward, Yokohama 236-0004, Kanagawa, Japan. t136001f@yokohama-cu.ac.jp
Received: August 29, 2025 Revised: September 17, 2025 Accepted: November 10, 2025 Published online: January 27, 2026 Processing time: 145 Days and 10.2 Hours
Abstract
BACKGROUND
Pancreaticobiliary maljunction (PBM) is a congenital disease in which the pancreatic and bile ducts fuse outside the duodenal wall. Congenital biliary dilatation (CBD) involves PBM and dilatation of the extrahepatic bile duct. The lack of Oddi sphincter action at the confluence results in the retrograde flow of pancreatic juice into the bile duct, placing patients with CBD at high risk of biliary carcinoma. The standard treatment for CBD is complete extrahepatic bile duct resection (EHBR). Hepatolithiasis (HL), a late complication following CBD surgery, has a deleterious clinical impact; further research is necessary to elucidate its risk factors.
AIM
To clarify the clinical impact of and risk factors for HL after CBD surgery.
METHODS
A retrospective study was conducted with 223 CBD patients who underwent EHBR across three tertiary hospitals to investigate postoperative complications. An exploratory analysis was performed to identify factors associated with HL development. Risk factors were subsequently identified using least absolute shrinkage and selection operator (LASSO) analysis.
RESULTS
HL was observed in 15/223 (6.7%) patients. Two of those patients developed liver failure owing to biliary cirrhosis; one died, and the other received liver transplantation. Two patients required major hepatectomy. The majority of the remaining patients required repeated enteroscopic and/or percutaneous lithotomy procedures. LASSO analysis revealed older age at surgery as an independent risk factor for HL; the time-dependent receiver operating characteristic analysis at 6 years after surgery revealed a cutoff age of 31 years.
CONCLUSION
HL following CBD surgery has a markedly deleterious clinical impact. Advanced age at the time of CBD surgery was identified as an independent risk factor for HL.
Core Tip: A total of 223 patients with congenital biliary dilatation (CBD) who underwent extrahepatic bile duct resection were retrospectively examined to clarify the clinical impact of hepatolithiasis (HL) that developed after CBD surgery. Among these 223 patients, 15 (6.7%) developed HL. Among these 15 patients, 2 developed biliary cirrhosis: (1) One died of liver failure; and (2) The other required liver transplantation. Two additional patients required major hepatectomy. Most of the remaining 11 patients underwent repeated hospitalization for lithotomy. Statistical analysis revealed that older age at the time of CBD surgery was an independent risk factor for the development of HL.
