Li Jiayi,Li Zonghan,Fang Yiwei,et al.Development of a preoperative imaging-based predictive model for nephron-sparing surgery feasibility in pediatric bilateral Wilms tumor and comparative evaluation of multiscore systems[J].Journal of Clinical Pediatric Surgery,2025,(10):916-922.[doi:10.3760/cma.j.cn101785-202501058-003]
基于术前影像构建儿童双侧肾母细胞瘤保留肾单位手术可行性预测模型及多评分系统的对比研究
- Title:
- Development of a preoperative imaging-based predictive model for nephron-sparing surgery feasibility in pediatric bilateral Wilms tumor and comparative evaluation of multiscore systems
- Keywords:
- Wilms Tumor; Nephron-sparing Surgery; Prediction Model; Evaluation Study; Comparative Study
- 摘要:
- 目的 本研究旨在基于术前影像资料构建儿童双侧肾母细胞瘤(bilateral Wilms tumor,BWT)保留肾单位手术(nephron-sparing surgery,NSS)可行性的临床预测模型,并与现有成人肾脏肿瘤术前评分系统进行对比。方法 回顾性分析2008—2024年在首都医科大学附属北京儿童医院接受手术治疗的BWT患儿临床及影像资料。共纳入61例患儿121个肾单位,采取保留肾单位手术99个,为NSS组;非保留肾单位手术22个,为非NSS组。提取患儿术前影像学特征,采用单因素及多因素Logistic回归分析确定与NSS可行性相关的因素,构建预测模型,通过受试者操作特征曲线下面积(area under curve,AUC)、校准曲线和决策曲线分析评估模型性能,并对比新模型与R.E.N.A.L、PADUA、SPARE评分系统的预测效能。结果 单因素和多因素Logistic回归分析显示,肿瘤最大长径(OR=0.651,95%CI:0.476~0.891,P=0.007)与集合系统的关系(OR=0.037,95%CI:0.006~0.220,P=0.001)是NSS的独立影响因素。基于肿瘤大小(size,S)与集合系统的关系(relation with collecting system,R)及残余肾实质比例(remaining renal parenchyma,R)构建的RSR(relation-size-remaining renal parenchyma)模型,其AUC为0.957(95%CI:0.924~0.989)。与成人评分系统相比,RSR模型的AUC显著高于R.E.N.A.L(0.778)、PADUA(0.828)和SPARE(0.805),且临床收益曲线显示RSR模型具有更高的临床净收益。结论 RSR模型是目前国内外首个针对BWT患儿NSS可行性的预测模型,具有针对性强、评价指标简化及预测准确性高的优势,可为BWT患儿NSS手术适应证的制定提供参考。
- Abstract:
- Objective To construct a clinical prediction model for the feasibility of nephron-sparing surgery (NSS) in children with bilateral Wilms tumor (BWT) based upon preoperative imaging data and compare it with existing adult renal tumor preoperative scoring systems. Methods A retrospective analysis was conducted for the clinical and imaging data of surgical BWT children from 2008 to 2024.A total of 121 renal units from 61 children were included (99 in NSS group and 22 in non-NSS group).Preoperative imaging features were extracted and univariate and multivariate Logistic regression analyses performed for identifying the factors associated with the feasibility of NSS.A prediction model was constructed and evaluated using area under receiver operating characteristic curve (AUC),calibration curve and decision curve.The model was compared with R.E.N.A.L,PADUA and SPARE scoring systems. Results Multivariate Logistic regression analysis identified tumor maximum diameter (OR=0.651,95%CI:0.476-0.891,P=0.007) and relationship with collecting system (OR=0.037,95%CI:0.006-0.220,P=0.001) as independent predictors of NSS feasibility.RSR (relation-size-remaining renal parenchyma) model,based upon tumor size (S),relationship with collecting system (R) and remaining renal parenchyma (R),achieved an AUC of 0.957 (95%CI: 0.924-0.989),demonstrating good predictive capability and clinical applicability.As compared with adult scoring systems,RSR model had demonstrated significantly higher AUC values than R.E.N.A.L (0.778),PADUA (0.828) and SPARE (0.805).And decision curve analysis revealed greater clinical net benefit. Conclusions As the first prediction model for NSS feasibility in BWT children,RSR model offers strong targeting,simplified evaluation criteria and high predictive accuracy.It may serve as a reference for defining the indications for NSS in BWT children.
参考文献/References:
[1] 中国抗癌协会小儿肿瘤专业委员会,中华医学会小儿外科分会肿瘤学组,中国研究型医院学会儿童肿瘤专业委员会.中国儿童双侧肾母细胞瘤诊疗专家共识[J].中华小儿外科杂志,2024,45(11):961-977.DOI:10.3760/cma.j.cn421158-20240703-00306. Chinese Children Cancer Group (CCCG),Chinese Anti-Cancer Association,Group of Surgical Oncology,Chinese Association of Pediatric Surgeons,Committee of Pediatric Oncology,Chinese Research Hospital Association.Expert consensus on diagnosing and treating bilateral Wilms tumor in Chinese children[J].Chin J Pediatr Surg,2024,45(11):961-977.DOI:10.3760/cma.j.cn421158-20240703-00306.
[2] van den Heuvel-Eibrink MM,Hol JA,Pritchard-Jones K,et al.Position paper: rationale for the treatment of Wilms tumour in the UMBRELLA SIOP-RTSG 2016 protocol[J].Nat Rev Urol,2017,14(12):743-752.DOI:10.1038/nrurol.2017.163.
[3] Ficarra V,Novara G,Secco S,et al.Preoperative aspects and dimensions used for an anatomical (PADUA) classification of renal tumours in patients who are candidates for nephron-sparing surgery[J].Eur Urol,2009,56(5):786-793.DOI:10.1016/j.eururo.2009.07.040.
[4] Kutikov A,Uzzo RG.The R.E.N.A.L.nephrometry score: a comprehensive standardized system for quantitating renal tumor size,location and depth[J].J Urol,2009,182(3):844-853.DOI:10.1016/j.juro.2009.05.035.
[5] Ehrlich P,Chi YY,Chintagumpala MM,et al.Results of the first prospective multi-institutional treatment study in children with bilateral Wilms tumor (AREN0534): a report from the children’s oncology group[J].Ann Surg,2017,266(3):470-478.DOI:10.1097/SLA.0000000000002356.
[6] Ficarra V,Porpiglia F,Crestani A,et al.The simplified PADUA REnal (SPARE) nephrometry system: a novel classification of parenchymal renal tumours suitable for partial nephrectomy[J].BJU Int,2019,124(4):621-628.DOI:10.1111/bju.14772.
[7] 中华医学会小儿外科学分会泌尿外科学组.儿童肾母细胞瘤诊疗专家共识[J].中华小儿外科杂志,2020,41(7):585-590.DOI:10.3760/cma.j.cn421158-20190805-00479. Group of Urological Surgery,Branch of Pediatric Surgery,Chinese Medical Association.Expert consensus on pediatric nephroblastoma[J].Chin J Pediatr Surg,2020,41(7):585-590.DOI:10.3760/cma.j.cn421158-20190805-00479.
[8] Bednarek OL,Seemann N,Brzezinski J,et al.Outcomes according to treatment using an established protocol in patients with bilateral Wilms’ tumor: a national Canadian population-based study[J].J Pediatr Surg,2023,58(5):1014-1017.DOI: 10.1016/j.jpedsurg.2023.01.016.
[9] Chen SY,Lee WGH,Laifman E,et al.A single center experience with bilateral Wilms tumor[J].Am Surg,2023,89(10):4101-4104.DOI:10.1177/00031348231175446.
[10] Long CJ,Mittal S,Kolon TF.Expanding the use of nephron-sparing surgery for Wilms tumor[J].J Natl Compr Canc Netw,2022,20(5):540-546.DOI: 10.6004/jnccn.2022.7099.
[11] Sudour H,Audry G,Schleimacher G,et al.Bilateral Wilms tumors (WT) treated with the SIOP 93 protocol in France: epidemiological survey and patient outcome[J].Pediatr Blood Cancer,2012,59(1):57-61.DOI:10.1002/pbc.24059.
[12] Kieran K,Davidoff AM.Nephron-sparing surgery for bilateral Wi-lms tumor[J].Pediatr Surg Int,2015,31(3):229-236.DOI:10.1007/s00383-015-3668-1.
[13] Kieran K,Williams MA,McGregor LM,et al.Repeat nephron-sp-aring surgery for children with bilateral Wilms tumor[J].J Pediatr Surg,2014,49(1):149-153.DOI:10.1016/j.jpedsurg.2013.09.048.
[14] Sutthatarn P,Gomez Quevedo O,Gleason J,et al.Management of intravascular thrombus in cases of bilateral Wilms tumor or horseshoe kidney[J].J Pediatr Surg,2022,57(9):166-173.DOI:10.1016/j.jpedsurg.2021.07.025.
[15] Veccia A,Antonelli A,Uzzo RG,et al.Predictive value of neph-rometry scores in nephron-sparing surgery: a systematic review and Meta-analysis[J].Eur Urol Focus,2020,6(3):490-504.DOI:10.1016/j.euf.2019.11.004.
[16] Mrad C,Chamouni A,Guerra X,et al.Nephrometry scoring systems for nephron sparing surgery in children[J].J Pediatr Hematol Oncol,2024,46(1):1-7.DOI:10.1097/MPH.0000000000002781.
[17] Mittal S,Long C,El-Ali A,et al.Utilizing RENAL nephrometry in pediatric patients undergoing nephron-sparing surgery for renal tumors: a single-institutional cohort[J].J Pediatr Urol,2023,19(5):641.e1-641.e6.DOI:10.1016/j.jpurol.2023.03.041.
[18] Gao PF,Jiang H,Wu CL,et al.RENAL nephrometry scoring system in bilateral Wilms tumor: predictive application[J].Pediatr Surg Int,2023,39(1):230.DOI: 10.1007/s00383-023-05500-3.
[19] Tornberg SV,Kilpel?inen TP,J?rvinen P,et al.Renal tumor invasion depth and diameter are the two most accurate anatomical features regarding the choice of radical versus partial nephrectomy[J].Scand J Surg,2018,107(1):54-61.DOI: 10.1177/1457496917731186.
[20] Godzinski J,Graf N,Audry G.Current concepts in surgery for Wilms tumor-the risk and function-adapted strategy[J].Eur J Pediatr Surg,2014,24(6):457-460.DOI:10.1055/s-0034-1396425.
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备注/Memo
收稿日期:2025-1-20。
基金项目:北京市科委、中关村管委会"医药创新品种及平台培育"专项资助(Z231100004823034)
通讯作者:宋宏程,Email:songhch1975@126.com;李振武,Email:omally@126.com