Skip to main content
Log in

Construction and validation of nomogram prognostic model for predicting survival in hepatoblastoma patients: a population-based study

  • Original Article
  • Published:
Updates in Surgery Aims and scope Submit manuscript

Abstract

For patients with hepatoblastoma (HB), current staging system is not accurate in predicting survival outcomes. The aim of this study was to develop two accurate survival prediction models to guide clinical decision making. A retrospective analysis of 424 HB patients was performed from 2004 to 2015 using the Surveillance, Epidemiology and End Results (SEER) database. Univariate and multivariate Cox regression analysis was used to screen for variables. The identified variables were used to build survival prediction model. The performance of the nomogram models was assessed based on the concordance index (C-index), calibration plot, and receiver operating characteristic (ROC) curve. The Cox regression analysis identified six variables affecting overall survival (OS) in HB patients, including race, tumor size, lymph node involvement, distant metastases, surgery and chemotherapy. And the Cox regression analysis identified five variables including race, lymph node involvement, distant metastases, surgery, and chemotherapy that affect cancer-specific survival (CCS) in HB patients. In the training cohort, the C-index of the nomogram in predicting the OS was 0.791 [95% confidence intervals (95% CI) 0.717–0.865], CSS was 0.805(95% CI 0.728–0.882). In the validation cohort, the C-index of the nomogram in predicting the OS was 0.712 (95% CI 0.511–0.913), the CSS was 0.751 (95% CI 0.566–0.936). In the training cohort, the area under the receiver operator characteristics curve (AUC) values of the nomogram in prediction of the 1-, 3-, and 5-year OS were 0.842 (95% CI 0.739–0.944), 0.759 (95% CI 0.670–0.849), and 0.770 (95% CI 0.686–0.852), respectively. In the validation cohort, the AUC values for prediction of the 1-, 3-, and 5-year OS were 0.920 (95% CI 0.806–1.034), 0.863 (95% CI 0.750–0.976), and 0.844 (95% CI 0.721–0.967), respectively. Two nomogram models were developed and validated in this study which provided accurate prediction of the OS and CSS in HB patients. The constructed models can be used for predicting survival outcomes and guide treatment for HB patients.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5
Fig. 6

Similar content being viewed by others

Data availability

Publicly available datasets were analyzed in this study. These data can be found here: https://seer.cancer.gov/data/.

References

  1. Xie L, Onysko J, Morrison H (2018) Childhood cancer incidence in Canada: demographic and geographic variation of temporal trends (1992–2010). Health Promot Chronic Dis Prev Can 38:79–115. https://doi.org/10.24095/hpcdp.38.3.01

    Article  PubMed  Google Scholar 

  2. Allan BJ, Parikh PP, Diaz S, Perez EA, Neville HL, Sola JE (2013) Predictors of survival and incidence of hepatoblastoma in the paediatric population. HPB (Oxford) 15:741–746. https://doi.org/10.1111/hpb.12112

    Article  PubMed  Google Scholar 

  3. Ranganathan S, Lopez-Terrada D, Alaggio R (2020) Hepatoblastoma and pediatric hepatocellular carcinoma: an update. Pediatr Dev Pathol 23:79–95. https://doi.org/10.1177/1093526619875228

    Article  PubMed  Google Scholar 

  4. Feng TC, Zai HY, Jiang W, Zhu Q, Jiang B, Yao L et al (2019) Survival and analysis of prognostic factors for hepatoblastoma: based on SEER database. Ann Transl Med 7:555. https://doi.org/10.21037/atm.2019.09.76

    Article  PubMed  PubMed Central  Google Scholar 

  5. Spector LG, Birch J (2012) The epidemiology of hepatoblastoma. Pediatr Blood Cancer 59:776–779. https://doi.org/10.1002/pbc.24215

    Article  PubMed  Google Scholar 

  6. Exelby PR, Filler RM, Grosfeld JL (1975) Liver tumors in children in the particular reference to hepatoblastoma and hepatocellular carcinoma: American Academy of Pediatrics Surgical Section Survey–1974. J Pediatr Surg 10:329–337. https://doi.org/10.1016/0022-3468(75)90095-0

    Article  CAS  PubMed  Google Scholar 

  7. Lake CM, Tiao GM, Bondoc AJ (2019) Surgical management of locally-advanced and metastatic hepatoblastoma. Semin Pediatr Surg 28:150856. https://doi.org/10.1016/j.sempedsurg.2019.150856

    Article  PubMed  Google Scholar 

  8. Venkatramani R, Stein JE, Sapra A, Genyk Y, Jhaveri V, Malogolowkin M et al (2015) Effect of neoadjuvant chemotherapy on resectability of stage III and IV hepatoblastoma. Br J Surg 102:108–113. https://doi.org/10.1002/bjs.9681

    Article  CAS  PubMed  Google Scholar 

  9. Venkatramani R, Furman WL, Fuchs J, Warmann SW, Malogolowkin MH (2012) Current and future management strategies for relapsed or progressive hepatoblastoma. Paediatr Drugs 14:221–232. https://doi.org/10.2165/11597740-000000000-00000

    Article  PubMed  Google Scholar 

  10. Czauderna P, Otte JB, Aronson DC, Gauthier F, Mackinlay G, Roebuck D et al (2005) Guidelines for surgical treatment of hepatoblastoma in the modern era–recommendations from the Childhood Liver Tumour Strategy Group of the International Society of Paediatric Oncology (SIOPEL). Eur J Cancer 41:1031–1036. https://doi.org/10.1016/j.ejca.2005.02.004

    Article  PubMed  Google Scholar 

  11. Meyers RL, Maibach R, Hiyama E, Häberle B, Krailo M, Rangaswami A et al (2017) Risk-stratified staging in paediatric hepatoblastoma: a unified analysis from the Children’s Hepatic tumors International Collaboration. Lancet Oncol 18:122–131. https://doi.org/10.1016/s1470-2045(16)30598-8

    Article  PubMed  Google Scholar 

  12. Aronson DC, Czauderna P, Maibach R, Perilongo G, Morland B (2014) The treatment of hepatoblastoma: Its evolution and the current status as per the SIOPEL trials. J Indian Assoc Pediatr Surg 19:201–207. https://doi.org/10.4103/0971-9261.142001

    Article  PubMed  PubMed Central  Google Scholar 

  13. Gloeckler Ries LA, Reichman ME, Lewis DR, Hankey BF, Edwards BK (2003) Cancer survival and incidence from the Surveillance, Epidemiology, and End Results (SEER) program. Oncologist 8:541–552. https://doi.org/10.1634/theoncologist.8-6-541

    Article  PubMed  Google Scholar 

  14. Collins GS, Reitsma JB, Altman DG, Moons KG (2015) Transparent reporting of a multivariable prediction model for individual prognosis or diagnosis (TRIPOD): the TRIPOD statement. BMJ 350:g7594. https://doi.org/10.1136/bmj.g7594

    Article  CAS  PubMed  Google Scholar 

  15. Kahla JA, Siegel DA, Dai S, Lupo PJ, Foster JH, Scheurer ME et al (2022) Incidence and 5-year survival of children and adolescents with hepatoblastoma in the United States. Pediatr Blood Cancer 69:e29763. https://doi.org/10.1002/pbc.29763

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  16. El Asmar A, El Rassi Z (2016) Hepatoblastoma in childhood, long term survival achieved: 2 case reports and literature review. Int J Surg Case Rep 21:55–58. https://doi.org/10.1016/j.ijscr.2016.02.019

    Article  PubMed  PubMed Central  Google Scholar 

  17. Brown J, Perilongo G, Shafford E, Keeling J, Pritchard J, Brock P et al (2000) Pretreatment prognostic factors for children with hepatoblastoma– results from the International Society of Paediatric Oncology (SIOP) study SIOPEL 1. Eur J Cancer 36:1418–1425. https://doi.org/10.1016/s0959-8049(00)00074-5

    Article  CAS  PubMed  Google Scholar 

  18. von Schweinitz D (2012) Hepatoblastoma: recent developments in research and treatment. Semin Pediatr Surg 21:21–30. https://doi.org/10.1053/j.sempedsurg.2011.10.011

    Article  Google Scholar 

  19. Perilongo G, Shafford E, Maibach R, Aronson D, Brugières L, Brock P et al (2004) Risk-adapted treatment for childhood hepatoblastoma final report of the second study of the International Society of Paediatric Oncology–SIOPEL 2. Eur J Cancer. https://doi.org/10.1016/j.ejca.2003.06.003

    Article  PubMed  Google Scholar 

  20. Yoon HM, Hwang J, Kim KW, Namgoong JM, Kim DY, Koh KN et al (2019) Prognostic factors for event-free survival in pediatric patients with hepatoblastoma based on the 2017 PRETEXT and CHIC-HS systems. Cancers (Basel). https://doi.org/10.3390/cancers11091387

    Article  PubMed  Google Scholar 

  21. Zhi T, Zhang WL, Zhang Y, Hu HM, Huang DS (2021) Clinical characteristics and prognostic factors of hepatoblastoma in 316 children aged under 3 years - a 14-year retrospective single-center study. BMC Pediatr 21:170. https://doi.org/10.1186/s12887-021-02630-2

    Article  PubMed  PubMed Central  Google Scholar 

  22. Katzenstein HM, Furman WL, Malogolowkin MH, Krailo MD, McCarville MB, Towbin AJ et al (2017) Upfront window vincristine/irinotecan treatment of high-risk hepatoblastoma: a report from the Children’s Oncology Group AHEP0731 study committee. Cancer 123:2360–2367. https://doi.org/10.1002/cncr.30591

    Article  CAS  PubMed  Google Scholar 

  23. Faraj W, Dar F, Marangoni G, Bartlett A, Melendez HV, Hadzic D et al (2008) Liver transplantation for hepatoblastoma. Liver Transpl 14:1614–1619. https://doi.org/10.1002/lt.21586

    Article  PubMed  Google Scholar 

  24. Ke M, Zhou Y, Yang CZ, Li L, Diao M (2022) Analysis of risk factors for angiolymphatic invasion and establishment of a predictive nomogram for hepatoblastomas. J Pediatr Surg 57:430–437. https://doi.org/10.1016/j.jpedsurg.2022.02.024

    Article  PubMed  Google Scholar 

  25. Shi SJ, Wang DL, Hu W, Peng F, Kang Q (2018) Ex vivo liver resection and autotransplantation with cardiopulmonary bypass for hepatoblastoma in children: a case report. Pediatr Transplant 22:e13268. https://doi.org/10.1111/petr.13268

    Article  PubMed  Google Scholar 

  26. Hung GY, Lin LY, Yu TY, Lee CY, Yen HJ, Horng JL (2018) Hepatoblastoma incidence in Taiwan: a population-based study. J Chin Med Assoc 81:541–547. https://doi.org/10.1016/j.jcma.2017.11.012

    Article  PubMed  Google Scholar 

  27. Ke M, Zhou Y, Chang-Zhen Y, Li L, Diao M (2022) A nomogram model to predict prognosis of patients with hepatoblastoma. Pediatr Blood Cancer. https://doi.org/10.1002/pbc.29932

    Article  PubMed  Google Scholar 

Download references

Acknowledgements

None.

Funding

This work was supported by the General Program of the National Natural Science Foundation of China (82070554 and 81770537); the Major Scientific and Technological Special Project for Public Health in Tianjin (21ZXGWSY00080); the Tianjin Medical University General Hospital Clinical Research Program (22ZYYLCCG06) to Daqing Sun. The funding bodies played no role in the study design, data collection, analysis, interpretation, or manuscript writing.

Author information

Authors and Affiliations

Authors

Contributions

DS and SW: conception and design. SW and SF: development of methodology. RL and WS: acquisition of data. ZG, YW and WS: analysis and interpretation of data. SW: writing, review, and/or revision of the manuscript. DS and SW: administrative, technical, or material support. DS: study supervision. All authors participated in the writing of the final manuscript and approved the final submission.

Corresponding author

Correspondence to Daqing Sun.

Ethics declarations

Conflict of interest

The authors declare no conflict of interest.

Human ethics statement

The study contained in this manuscript has no human participants, which waives the need for ethics approval and written informed consent from participants to take part in the study.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Supplementary Information

Below is the link to the electronic supplementary material.

Supplementary file1 (DOCX 18 KB)

Rights and permissions

Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Wang, S., Fu, S., Li, R. et al. Construction and validation of nomogram prognostic model for predicting survival in hepatoblastoma patients: a population-based study. Updates Surg (2024). https://doi.org/10.1007/s13304-024-01814-6

Download citation

  • Received:

  • Accepted:

  • Published:

  • DOI: https://doi.org/10.1007/s13304-024-01814-6

Keywords

Navigation