Li Siyuan,Diao Mei,Li Long.Application value of natal ultrasonographic combined with gamma-glutamyl transpeptidase testing in assessing the risk of choledochal cyst perforation[J].Journal of Clinical Pediatric Surgery,2024,(12):1149-1154.[doi:10.3760/cma.j.cn101785-202405033-008]
超声联合谷氨酰转肽酶检测对产前诊断的胆总管囊肿穿孔风险的预测价值研究
- Title:
- Application value of natal ultrasonographic combined with gamma-glutamyl transpeptidase testing in assessing the risk of choledochal cyst perforation
- 摘要:
- 目的 探讨妊娠期至手术前超声检查联合血清谷氨酰转肽酶(gamma-glutamyl transpeptidase,GGT)检测对产前诊断的胆总管囊肿(choledochal cyst,CDC)穿孔风险的预测价值。方法 收集北京大学首都儿科研究所教学医院普通外科2018年1月至2023年9月收治的435例产前诊断为CDC患儿胎儿期至手术前超声数据、术前肝功能指标。将435例CDC患儿按照3∶1的比例随机分为建模集(n=326)和验证集(n=109)。将建模集326例患儿按照术中诊断分为穿孔组和未穿孔组,比较两组各指标差异,通过Logistic回归分析建立诊断模型,采用霍斯默—莱梅肖(Hosmer-Lemeshow,HL)拟合优度检验和受试者操作特征(receiver operating characteristic curve,ROC)曲线评估模型对产前诊断CDC穿孔的诊断效能。在验证集中对模型进行灵敏度、特异度评价。结果 建模集326例患儿中,穿孔组43例,未穿孔组283例。手术时日龄、GGT、直接胆红素(direct bilirubin,DB)、总胆红素(total bilirubin,TB)、胎儿期至手术前囊肿体积总增长速率与产前诊断CDC穿孔存在关联性(P<0.05)。多因素Logistic回归分析结果提示,囊肿体积总增长速率和GGT是产前诊断CDC穿孔的独立危险因素(P<0.05),并基于以上指标建立诊断模型。ROC曲线分析显示,囊肿体积总增长速率的最佳截断值为每周1.00 cm3,ROC曲线下面积(area under ROC curve,AUC)为0.849,灵敏度、特异度分别为87.0%、73.4%;GGT的最佳截断值为150.85 IU/L,AUC为0.796,灵敏度、特异度分别为84.2%、61.4%;诊断模型的AUC为0.915,灵敏度、特异度提升至92.1%、82.6%。HL拟合优度检验提示P=0.805。验证集中模型的AUC为0.858,灵敏度、特异度分别为92.9%、73.1%。结论 胎儿期至手术前囊肿体积总增长速率和GGT水平有助于评估产前诊断CDC的穿孔风险,并预测发生穿孔的可能性。二者联合可在一定程度上改善预测结果的灵敏度和特异度,从而尽可能提高评估穿孔风险的效能。
- Abstract:
- Objective To explore the value of combining natal ultrasonographic and serum gamma-glutamyl transpeptidase (GGT) testing from pregnancy to pre-surgery in assessing the risk of choledochal cyst (CDC) perforation diagnosed prenatally. Methods Data were collected from 435 children diagnosed prenatally with CDC,including natal ultrasonographic data from pregnancy to per-surgery and pre-surgical liver function indicators.The 435 children were randomly divided into a modeling set (n=326) and a validation set (n=109) in a 3:1 ratio.The 326 children in the modeling set were further divided into perforation and non-perforated groups based on intraoperative diagnosis.The differences in various indicators between the two groups were compared,and a diagnostic model was established through logistic regression analysis.The model’s diagnostic performance for prenatal CDC perforation was evaluated using the Hosmer-Lemeshow (HL) test and the receiver operating characteristic (ROC) curve.The model was then evaluated in the validation set. Results Among the 326 children in the modeling set,43 were in the perforation group and 283 in the non-perforation group.Age at surgery,GGT,direct bilirubin (DB),total bilirubin (TB),and total growth rate of cyst volume from pregnancy to pre-surgery were correlated with prenatal CDC perforation (P<0.05).Multivariate logistic regression indicated that the total growth rate of cyst volume and GGT were independent risk factors for CDC perforation.A diagnostic model was established based on these indicators.ROC results showed that the optimal cut-off value for the total growth rate of cyst volume was 1.00 cm3/week,with an area under the ROC curve (AUC) of 0.849,sensitivity of 87.0%,and specificity of 73.4%.The optimal cut-off value for GGT was 150.85 IU/L,with an AUC of 0.796,sensitivity of 84.2%,and specificity of 61.4%.The model’s AUC was 0.915,with sensitivity and specificity improved to 92.1% and 82.6%,respectively.HL test indicated P=0.805.In the validation set,the model’s AUC was 0.858,with sensitivity and specificity of 92.9 % and 73.7%,respectively. Conclusions The total growth rate of cyst volume and GGT testing from pregnancy to pre-surgery can help assess the risk of CDC perforation diagnosed prenatally and predict the likelihood of perforation.Combined use of these two indicators can improve the sensitivity and specificity of prediction,enhancing the effectiveness of perforation risk assessment and has good application value.
参考文献/References:
[1] Yamaguchi M.Congenital choledochal cyst.Analysis of 1,433 patients in the Japanese literature[J].Am J Surg,1980,140(5):653-657.DOI:10.1016/0002-9610(80)90051-3.
[2] Ando H,Ito T,Watanabe Y,et al.Spontaneous perforation of choledochal cyst[J].J Am Coll Surg,1995,181(2):125-128.
[3] Stringer MD,Dhawan A,Davenport M,et al.Choledochal cysts:lessons from a 20 year experience[J].Arch Dis Child,1995,73(6):528-531.DOI:10.1136/adc.73.6.528.
[4] Ando K,Miyano T,Kohno S,et al.Spontaneous perforation of choledochal cyst:a study of 13 cases[J].Eur J Pediatr Surg,1998,8(1):23-25.DOI:10.1055/s-2008-1071113.
[5] Diao M,Li L,Cheng W.Timing of choledochal cyst perforation[J].Hepatology,2020,71(2):753-756.DOI:10.1002/hep.30902.
[6] Kim YJ,Kim SH,Yoo SY,et al.Comparison of clinical and radiologic findings between perforated and non-perforated choledochal cysts in children[J].Korean J Radiol,2022,23(2):271-279.DOI:10.3348/kjr.2021.0169.
[7] Lilly JR,Weintraub WH,Altman RP.Spontaneous perforation of the extrahepatic bile ducts and bile peritonitis in infancy[J].Surgery,1974,75(5):664-673.
[8] Goel P,Jain V,Manchanda V,et al.Spontaneous biliary perforations:an uncommon yet important entity in children[J].J Clin Diagn Res,2013,7(6):1201-1206.DOI:10.7860/JCDR/2013/5429.3076.
[9] Diao M,Li L,Cheng W.Single-incision laparoscopic hepaticojejunostomy for children with perforated choledochal cysts[J].Surg Endosc,2018,32(7):3402-3409.DOI:10.1007/s00464-018-6047-x.
[10] Zhang SH,Cai DT,Chen QJ,et al.Value of serum GGT level in the timing of diagnosis of choledochal cyst perforation[J].Front Pediatr,2022,10:921853.DOI:10.3389/fped.2022.921853.
[11] Bilal H,Irshad M,Shahzadi N,et al.Neonatal cholestasis:the changing etiological spectrum in Pakistani children[J].Cureus,2022,14(6):e25882.DOI:10.7759/cureus.25882.
[12] Chen JY,Tang Y,Wang ZG,et al.Clinical value of ultrasound in diagnosing pediatric choledochal cyst perforation[J].AJR Am J Roentgenol,2015,204(3):630-635.DOI:10.2214/AJR.14.12935.
[13] Xin Y,Wang XM,Wang Y,et al.Value of ultrasound in diagnosing perforation of congenital choledochal cysts in children[J].J Ultrasound Med,2021,40(10):2157-2163.DOI:10.1002/jum.15604.
[14] Yu P,Dong N,Pan YK,et al.Ultrasonography is useful in differentiating between cystic biliary atresia and choledochal cyst[J].Pediatr Surg Int,2021,37(6):731-736.DOI:10.1007/s00383-021-04886-2.
[15] 张雪华,陈文娟,杨芳,等.超声在先天性囊肿型胆道闭锁及胆总管囊肿的鉴别诊断探讨[J].中国超声医学杂志,2016,32(7):619-621.DOI:10.3969/j.issn.1002-0101.2016.07.013. Zhang XH,Chen WJ,Yang F,et al.The value of differential diagnosis of congenital cystic biliary atresia and choledochal cyst by ultrasound[J].Chin J Ultrasound Med,2016,32(7):619-621.DOI:10.3969/j.issn.1002-0101.2016.07.013.
[16] Chiang L,Chui CH,Low Y,et al.Perforation:a rare complication of choledochal cysts in children[J].Pediatr Surg Int,2011,27(8):823-827.DOI:10.1007/s00383-011-2882-8.
[17] Evans K,Marsden N,Desai A.Spontaneous perforation of the bile duct in infancy and childhood:a systematic review[J].J Pediatr Gastroenterol Nutr,2010,50(6):677-681.DOI:10.1097/MPG.0b013e3181d5eed3.
[18] Chang MY,Kim MJ,Han SJ,et al.Choledochal cyst rupture with an intrahepatic pseudocyst mimicking hepatic mesenchymal hamartoma in an infant[J].Clin Imaging,2015,39(5):914-916.DOI:10.1016/j.clinimag.2015.04.016.
[19] Sherwood W,Boyd P,Lakhoo K.Postnatal outcome of antenatally diagnosed intra-abdominal cysts[J].Pediatr Surg Int,2008,24(7):763-765.DOI:10.1007/s00383-008-2148-2.
[20] Thakkar HS,Bradshaw C,Impey L,et al.Post-natal outcomes of antenatally diagnosed intra-abdominal cysts:a 22-year single-institution series[J].Pediatr Surg Int,2015,31(2):187-190.DOI:10.1007/s00383-014-3635-2.
[21] Lee MJ,Kim MJ,Yoon CS.MR cholangiopancreatography findings in children with spontaneous bile duct perforation[J].Pediatr Radiol,2010,40(5):687-692.DOI:10.1007/s00247-009-1447-7.
[22] Yasufuku M,Hisamatsu C,Nozaki N,et al.A very low-birth-weight infant with spontaneous perforation of a choledochal cyst and adjacent pseudocyst formation[J].J Pediatr Surg,2012,47(7):E17-E19.DOI:10.1016/j.jpedsurg.2012.03.055.
[23] Davenport M,Basu R.Under pressure:choledochal malformation manometry[J].J Pediatr Surg,2005,40(2):331-335.DOI:10.1016/j.jpedsurg.2004.10.015.
[24] Kaneko K,Ando H,Seo T,et al.Proteomic analysis of protein plugs:causative agent of symptoms in patients with choledochal cyst[J].Dig Dis Sci,2007,52(8):1979-1986.DOI:10.1007/s10620-006-9398-4.
[25] Fukuzawa H,Urushihara N,Miyakoshi C,et al.Clinical features and risk factors of bile duct perforation associated with pediatric congenital biliary dilatation[J].Pediatr Surg Int,2018,34(10):1079-1086.DOI:10.1007/s00383-018-4321-6.
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备注/Memo
收稿日期:2024-5-15。
基金项目:北京市医院管理中心登峰人才培养计划(DFL20221101);小儿外科微创诊疗体系建设,中国医学科学院医学与健康科技创新工程(2021RU015)
通讯作者:李龙,Email:lilong23@126.com