Adv Pediatr Surg.  2023 Dec;29(2):72-77. 10.13029/aps.2023.29.2.72.

Biliary Intraepithelial Neoplasia (BilIN) Diagnosed From Choledochal Cyst in 7-Year-Old Girl With Underlying Anomalous Pancreaticobiliary Ductal Union (APBDU)

• Gang S
• Lee Yi
• Ha S
• Kwon H
• Park J
• Namgoong JM

• KMID: 2551546
• DOI: http://doi.org/10.13029/aps.2023.29.2.72

Abstract

A choledochal cyst (CC) is an abnormal dilatation of the bile duct that is known to be a precursor of cholangiocarcinoma (CCA). Biliary intraepithelial neoplasia (BilIN) is a premalignant lesion that is considered to be found during the carcinogenesis of CCA. While BilINs are frequently identified in adult populations during pathology, there has been no report of them in the pediatric population. We first report a case of BilIN incidentally diagnosed from a CC in a 7-year-old child. This case suggests that BilIN should not be overlooked in children, especially those with risk factors such as anomalous pancreaticobiliary ductal union. Also, it supports the importance of early and complete resection of CC and its impact on preventing neoplastic changes in the biliary system.

Keyword

Choledochal cyst; Cholangiocarcinoma; Anomalous pancreaticobiliary ductal union; Carcinogenesis; Malignancy