Welcome to visit Zhongnan Medical Journal Press Series journal website!

Construction and validation of a prognostic prediction model for osteosarcoma in children based on the SEER database

Published on Jul. 25, 2025Total Views: 78 timesTotal Downloads: 34 timesDownloadMobile

Author: ZHANG Lei 1 LI Yuwan 2 WU Yindan 3 XU Zhi 4 SHAO Jinfu 5

Affiliation: 1. Department of Pediatrics, Zhangjiagang Fifth People's Hospital, Zhangjiagang 215600, Jiangsu Province, China 2. Department of Orthopedics, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou 310009, China 3. Department of Pediatric Surgery, Zhangjiagang First People's Hospital, Zhangjiagang 215600, Jiangsu Province, China 4. Department of Orthopedics, Zhangjiagang Fifth People's Hospital, Zhangjiagang 215600, Jiangsu Province, China 5. Department of Pediatric Medicine, Zhangjiagang First People's Hospital, Zhangjiagang 215600, Jiangsu Province, China

Keywords: Children Osteosarcoma Prognosis Predictive model Nomogram SEER database

DOI: 10.12173/j.issn.1004-5511.202409090

Reference: Zhang L, Li YW, Wu YD, Xu Z, Shao JF. Construction and validation of a prognostic prediction model for osteosarcoma in children based on the SEER database[J]. Yixue Xinzhi Zazhi, 2025, 35(7): 800-809. DOI: 10.12173/j.issn.1004-5511.202409090. [Article in Chinese]

  • Abstract
  • Full-text
  • References
Abstract

Objective  To construct and validate a nomogram model for predicting the survival prognosis of pediatric osteosarcoma patients.

Methods  Data were collected from pediatric patients diagnosed with osteosarcoma based on the U.S. SEER database between 2000 and 2021, aged less than 14 years. Patients were randomly divided into a training set and a validation set in a 7 ∶ 3 ratio. Univariate and multivariate Cox proportional hazards models were used to identify factors affecting survival, then the prediction model was constructed and nomogram was plotted. The nomogram was validated using the concordance index (C-index), the receiver operating characteristic curve and the area under the curve (AUC), calibration curves, and decision curve analysis. Patients were divided into high-risk and low-risk groups according to the risk scores which were calculated based on the nomogram model. Subgroup survival analyses were performed using Kaplan- Meier survival curves.

Results  A total of 726 pediatric osteosarcoma patients were included, with 508 in the training set and 218 in the validation set. Multivariate Cox regression analysis identified tumor size [HR=1.002, 95%CI (1.001, 1.004)], lymph node metastasis [HR=3.341, 95%CI (2.368, 4.712)], and surgical approach (radical resection) [HR=0.382, 95%CI (0.219, 0.665)]; amputation [HR=0.471, 95%CI (0.255, 0.870)]) as independent prognostic factors. The C-index of the nomogram models constructed based on the above factors in the training and validation sets were 0.715 and 0.690, respectively. The AUC values for predicting 1-year, 3-year, and 5-year tumor specific survival rates were 0.833 and 0.810 (1-year), 0.751 and 0.718 (3-year), and 0.708 and 0.657 (5- year) in the training and validation sets, respectively. The calibration curve showed that the predicted values were highly consistent with the observed values. The clinical decision curve showed that the model had demonstrated a certain clinical net benefit in predicting 1-year, 3-year, and 5-year tumor specific survival rates within a certain prediction probability threshold range. Kaplan-Meier analysis showed that the survival rate of the high-risk group was significantly lower than that of the low-risk group (P<0.05).

Conclusion  This study successfully developed a nomogram model for predicting tumor-specific survival rates in pediatric osteosarcoma patients based on key prognostic factors including tumor size, lymph node metastasis status, and type of surgery, demonstrating good predictive performance and potential clinical utility.

Full-text
Please download the PDF version to read the full text: download
References

1.Papakonstantinou E, Athanasiadou KI, Markozannes G, et  al. Prognostic factors in high-grade pediatric osteosarcoma among children and young adults: Greek nationwide registry for childhood hematological malignancies and solid tumors (NARECHEM- ST) data along with a systematic review and Meta- analysis[J]. Cancer Epidemiol, 2024, 90: 102551. DOI: 10.1016/j.canep.2024.102551.

2.Ward E, DeSantis C, Robbins A, et al. Childhood and adolescent cancer statistics, 2014[J]. CA Cancer J Clin, 2014, 64(2): 83-103. DOI: 10.3322/caac.21219.

3.单正丽, 郭豫, 彭雪华. 儿童常见原发性恶性骨肿瘤影像学表现[J]. 中华放射学杂志, 2023, 57(10): 1142-1146. [Shan ZL, Guo Y, Peng XH. Imaging findings of common primary malignant bone tumors in children[J]. Chinese Journal of Radiology, 2023, 57(10): 1142-1146.] DOI: 10.3760/cma.j.cn112149-20230628-00445.

4.Nagarajan R, Weigel BJ, Thompson RC, et al. Osteosarcoma in the first decade of life[J]. Med Pediatr Oncol, 2003, 41(5): 480-483. DOI: 10.1002/mpo.10403.

5.Meyers PA, Schwartz CL, Krailo M, et al. Osteosarcoma: a randomized, prospective trial of the addition of ifosfamide and/or muramyl tripeptide to cisplatin, doxorubicin, and high-dose methotrexate[J]. J Clin Oncol, 2005, 23(9): 2004-2011. DOI: 10.1200/JCO.2005.06.031.

6.Boam T, Rogoyski BG, Jawaid W, et al. Do children with osteosarcoma benefit from pulmonary metastasectomy? a systematic review of published studies and "real world" outcomes[J]. Ann Surg, 2024, 280(2): 235-240. DOI: 10.1097/SLA.0000000000006239.

7.Smrke A, Anderson PM, Gulia A, et al. Future directions in the treatment of osteosarcoma[J]. Cells, 2021, 10(1): 172. DOI: 10.3390/cells10010172.

8.Kager L, Zoubek A, Pötschger U, et al. Primary metastatic osteosarcoma: presentation and outcome of patients treated on neoadjuvant cooperative osteosarcoma study group protocols[J]. J Clin Oncol, 2003, 21(10): 2011-2018. DOI: 10.1200/JCO.2003.08.132.

9.Pastorino U, Palmerini E, Porcu L, et al. Lung metastasectomy for osteosarcoma in children, adolescents, and young adults: proof of permanent cure[J]. Tumori, 2023, 109(1): 79-85. DOI: 10.1177/03008916211053048.

10.Yang QK, Lai QY, Wang Y, et al. Establishment and validation of prognostic nomograms to predict overall survival and cancer-specific survival for patients with osteosarcoma[J]. Neoplasma, 2021, 68(2): 434-446. DOI: 10.4149/neo_2020_200617N639.

11.Lu S, Wang Y, Liu G, et al. Construction and validation of nomogram to predict distant metastasis in osteosarcoma: a retrospective study[J]. J Orthop Surg Res, 2021, 16(1): 231. DOI: 10.1186/s13018-021-02376-8.

12.苏小涵, 谭巧, 曾姣, 等. 基于SEER数据库构建和验证男性乳腺癌预后模型[J]. 医学新知, 2024, 34(6): 611-621. [Su XH, Tan Q, Zeng J, et al. Construction and validation of a prognostic model for male breast cancer based on the SEER database[J]. Yixue Xinzhi Zazhi, 2024, 34(6): 611-621.] DOI: 10.12173/j.issn.1004-5511.202401099.

13.刘建, 师金, 田国. 基于SEER数据库的年轻肺腺癌晚期患者预后预测及风险分层的模型构建[J]. 解放军医学杂志, 2024, 49(8): 889-896. [Liu J, Shi J, Tian G. Construction of prognosis prediction and risk stratification model for young patients with advanced lung adenocarcinoma based on SEER database[J]. Medical Journal of Chinese People's Liberation Army, 2024, 49(8): 889-896.] DOI: 10.11855/j.issn.0577-7402.1133.2024.0327.

14.Cronin KA, Ries LA, Edwards BK. The surveillance, epidemiology, and end results (SEER) program of the national cancer institute[J]. Cancer, 2014, 120 Suppl 23: 3755-3757. DOI: 10.1002/cncr.29049.

15.Vickers AJ, Cronin AM, Elkin EB, et al. Extensions to decision curve analysis, a novel method for evaluating diagnostic tests, prediction models and molecular markers[J]. BMC Med Inform Decis Mak, 2008, 8: 53. DOI: 10.1186/1472-6947-8-53.

16.Luetke A, Meyers PA, Lewis I, et al. Osteosarcoma treatment-where do we stand? A state of the art review[J]. Cancer Treat Rev, 2014, 40(4): 523-532. DOI: 10.1016/j.ctrv.2013.11.006.

17.薛晓川, 周阳云, 魏兰懿, 等. 中药联合化疗药物治疗骨肉瘤的研究进展[J]. 中成药, 2024, 46(6): 1948-1954. [Xue XC, Zhou YY, Wei LY, et al. Research progress on the combination of traditional Chinese medicine and chemotherapy drugs for the treatment of osteosarcoma[J]. Chinese Traditional Patent Medicine, 2024, 46(6): 1948-1954.] DOI: 10.3969/i.issn.1001-1528.2024.06.028.

18.景义生, 谢兴文, 李宁, 等. 缓释化疗系统在骨肉瘤治疗中的研究进展[J]. 肿瘤, 2023, 43(12): 984-992. [Jing YS, Xie  XW, Li N, et al. Research progress of slow-release chemotherapy system for osteosarcoma[J]. Tumor, 2023, 43(12): 984-992.] DOI: 10.3781/j.issn.1000-7431.2023.2303-0118.

19.Kim MS, Lee SY, Lee TR, et al. Prognostic nomogram for predicting the 5-year probability of developing metastasis after neo-adjuvant chemotherapy and definitive surgery for AJCC stage II extremity osteosarcoma[J]. Ann Oncol, 2009, 20(5): 955-960. DOI: 10.1093/annonc/mdn723.

20.蔡猛, 流小舟, 樊根涛, 等. 20~40岁四肢骨肉瘤患者预后分析[J]. 医学研究生学报, 2021, 34(6): 604-608. [Cai M, Liu XZ, Fan GT, et al. Prognostic analysis of extremity osteosarcoma in patients aged 20-40 years[J]. Journal of Medical Research & Combat Trauma Care, 2021, 34(6): 604-608.] DOI: 10.16571/j.cnki.1008-8199.2021.06.008.

21.徐志, 陈运动, 孙玉洁, 等. 基于SEER数据库美国脊柱骨肉瘤患者数据: 治疗结果及预后预测模型的建立与验证 [J]. 中国组织工程研究, 2025, 29(30): 6583-6590. [Xu Z, Chen  YD, Sun YJ, et al. Data of spinal osteosarcoma patients in united states based on SEER database: construction and validation of a prediction model for treatment outcomes and prognosis[J]. Chinese Journal of Tissue Engineering Research, 2025, 29(30): 6583-6590.] DOI: 10.12307/2025.904.

22.Berner K, Bruland ØS. Prognostic impact of proximal versus distal localization in extremity long bone osteosarcomas[J]. Anticancer Res. 2019, 39(5): 2459-2466. DOI: 10.21873/anticanres.13365.

23.Oberlin O, Deley MC, Bui BN, et al. Prognostic factors in localized Ewing's tumours and peripheral neuroectodermal tumours: the third study of the French society of paediatric oncology (EW88 study)[J]. Br J Cancer, 2001, 85(11): 1646-1654. DOI: 10.1054/bjoc.2001.2150.