PDF(Pubmed)
Abstract：
BACKGROUND: Biliary intraepithelial neoplasia (BilIN), a noninvasive precursor of cholangiocarcinoma, can manifest malignant transformation. Since cholangiocarcinoma (CCA) may progress due to chronic inflammation in the bile ducts and gallbladder, choledochal cysts are considered a precursor to CCA. However, BilIN has rarely been reported in children, to date.
METHODS: We reviewed medical records of patients (< 18 years of age, n = 329) who underwent choledochal cyst excision at Asan Medical Center from 2008 to 2022. BilIN was diagnosed in 15 patients. Subsequent analyses were performed of the demographics, surgical procedures, clinical course, and outcomes in these patients. Subgroup analysis and multivariate logistic regression test were performed to identify factors influencing BilIN occurrence.
RESULTS: The mean age of the patients included in our study was 40.1 ± 47.6 months. In 15 patients, BilIN of various grades was diagnosed. Todani type I was prevalent in 80% of the patients. The median age at surgery was 17 months. During a mean follow-up of 63.3 ± 94.0 months, no adverse events such as stone formation in the remnant intrapancreatic common bile duct and intrahepatic duct or cholangiocarcinoma were observed, indicating a favorable outcome until now.
CONCLUSIONS: The potential progression of choledochal cysts to BilIN in children was demonstrated. These results could underscore the importance of early and comprehensive excision of choledochal cysts, including resection margins for associated lesions and more thorough postoperative surveillance in patients with or at risk of BilIN.
METHODS: We reviewed medical records of patients (< 18 years of age, n = 329) who underwent choledochal cyst excision at Asan Medical Center from 2008 to 2022. BilIN was diagnosed in 15 patients. Subsequent analyses were performed of the demographics, surgical procedures, clinical course, and outcomes in these patients. Subgroup analysis and multivariate logistic regression test were performed to identify factors influencing BilIN occurrence.
RESULTS: The mean age of the patients included in our study was 40.1 ± 47.6 months. In 15 patients, BilIN of various grades was diagnosed. Todani type I was prevalent in 80% of the patients. The median age at surgery was 17 months. During a mean follow-up of 63.3 ± 94.0 months, no adverse events such as stone formation in the remnant intrapancreatic common bile duct and intrahepatic duct or cholangiocarcinoma were observed, indicating a favorable outcome until now.
CONCLUSIONS: The potential progression of choledochal cysts to BilIN in children was demonstrated. These results could underscore the importance of early and comprehensive excision of choledochal cysts, including resection margins for associated lesions and more thorough postoperative surveillance in patients with or at risk of BilIN.
摘要：
背景：胆道上皮内瘤变（BilIN），胆管癌的非侵入性前体，可以表现为恶性转化。由于胆管癌（CCA）可能由于胆管和胆囊的慢性炎症而进展，胆总管囊肿被认为是CCA的前兆。然而,Bilin在儿童中很少被报道，到目前为止。
方法：我们回顾了患者的医疗记录（<18岁，n=329），从2008年至2022年在Asan医学中心接受了胆总管囊肿切除术。在15例患者中诊断出BilIN。随后对人口统计学进行了分析，外科手术,临床课程，以及这些患者的结果。进行亚组分析和多因素logistic回归检验以确定影响BilIN发生的因素。
结果：纳入本研究的患者平均年龄为40.1±47.6个月。在15名患者中，诊断出各种等级的Bilin。TodaniI型在80％的患者中普遍存在。手术时的中位年龄为17个月。在平均63.3±94.0个月的随访中，未观察到不良事件，如残余胰内胆总管和肝内胆管结石或胆管癌，表明到目前为止是有利的结果。
结论：儿童胆总管囊肿可能进展为BilIN。这些结果可以强调早期和全面切除胆总管囊肿的重要性。包括相关病变的切除切缘，以及对患有BilIN或有BilIN风险的患者进行更彻底的术后监测。
方法：我们回顾了患者的医疗记录（<18岁，n=329），从2008年至2022年在Asan医学中心接受了胆总管囊肿切除术。在15例患者中诊断出BilIN。随后对人口统计学进行了分析，外科手术,临床课程，以及这些患者的结果。进行亚组分析和多因素logistic回归检验以确定影响BilIN发生的因素。
结果：纳入本研究的患者平均年龄为40.1±47.6个月。在15名患者中，诊断出各种等级的Bilin。TodaniI型在80％的患者中普遍存在。手术时的中位年龄为17个月。在平均63.3±94.0个月的随访中，未观察到不良事件，如残余胰内胆总管和肝内胆管结石或胆管癌，表明到目前为止是有利的结果。
结论：儿童胆总管囊肿可能进展为BilIN。这些结果可以强调早期和全面切除胆总管囊肿的重要性。包括相关病变的切除切缘，以及对患有BilIN或有BilIN风险的患者进行更彻底的术后监测。
