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,,():1149-1154.[doi:10.3760/cma.j.cn101785-202405033-008]
Application value of natal ultrasonographic combined with gamma-glutamyl transpeptidase testing in assessing the risk of choledochal cyst perforation
- 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.
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.
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