Skip to main content
Log in

Impact of microscopically margin-positive resection on survival in children with hepatoblastoma after hepatectomy: a retrospective cohort study

  • Original Article
  • Published:
International Journal of Clinical Oncology Aims and scope Submit manuscript

Abstract

Background

Impact of R1 (microscopically margin-positive) resection on survival of patients with hepatoblastoma (HB) remains debatable. This study aimed to compare the long-term outcomes of R0 (microscopically margin-negative) and R1 resection for HB in children after hepatectomy.

Methods

We retrospectively reviewed files of children with HB who underwent resection at our institution between September 1, 2005, and November 30, 2017. Survival analyses and prognostic factors were evaluated using Kaplan–Meier curves and Cox regression models.

Results

Of 259 patients, 218 (84.2%) underwent R0 and 41 (15.8%) R1 resection. After adjusting for confounding factors, R1 resection demonstrated non-significantly lower overall survival (OS: hazard ratio [HR] = 0.75; 95% CI 0.34–1.64) and shorter event-free survival (EFS: HR = 0.97; 95% CI 0.53–1.78) rates than R0 resection. However, stratified analysis showed significantly increased risk of poor OS and EFS in patients with metastasis and mixed epithelial/mesenchymal pathologic subtype in R1 compared with R0 resection (P values for interactions < 0.05). There was no significant difference between R0 resection with metastasis and R1 resection with metastasis in the incidence of local recurrence (P = 0.494); however, a significant difference in the incidence of local recurrence was seen between R0 and R1 resection for subgroups with mixed pathologic subtypes (P = 0.035).

Conclusions

With effective chemotherapy, microscopic margin status may not be associated with survival outcome in children with HB undergoing hepatectomy. However, stratified analysis showed that R1 resection might be associated with decreased survival in children with mixed epithelial/mesenchymal HB, compared with R0 resection, and not affect survival outcomes in those with an epithelial subtype and without metastasis.

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

Similar content being viewed by others

References

  1. Hung GY, Lin LY, Yu TY et al (2018) Hepatoblastoma incidence in Taiwan: a population-based study. J Chin Med Assoc 81:541–547

    Article  Google Scholar 

  2. Aronson DC, Meyers RL (2016) Malignant tumors of the liver in children. Sem Pedatr Surg 25:265–275

    Article  Google Scholar 

  3. Hishiki T, Matsunaga T, Sasaki F et al (2011) Outcome of hepatoblastomas treated using the Japanese Study Group for Pediatric Liver Tumor (JPLT) protocol-2: report from the JPLT. Pediatr Surg Int 27:1–8

    Article  Google Scholar 

  4. Haeberle B, Dv S (2012) Treatment of hepatoblastoma in the German cooperative pediatric liver tumor studies. Front Biosci 4:493–498

    Article  Google Scholar 

  5. Fahy AS, Shaikh F, Gerstle JT (2019) Multifocal hepatoblastoma: what is the risk of recurrent disease in the remnant liver? J Pediatr Surg. https://doi.org/10.1016/j.jpedsurg.2019.01.036

    Article  PubMed  Google Scholar 

  6. Yuan XJ, Wang HM, Jiang H et al (2016) Multidisciplinary effort in treating children with hepatoblastoma in China. Cancer Lett 375:39–46

    Article  CAS  Google Scholar 

  7. Fuchs J, Rydzynski J, Dietrich VS et al (2002) Pretreatment prognostic factors and treatment results in children with hepatoblastoma: a report from the German Cooperative Pediatric Liver Tumor Study HB 94. Cancer 95:172–182

    Article  Google Scholar 

  8. Pham TA, Gallo AM, Concepcion W et al (2015) Effect of liver transplant on long-term disease-free survival in children with hepatoblastoma and hepatocellular cancer. JAMA Surg 150:1150–1158

    Article  Google Scholar 

  9. Meyers RL, Tiao G, De Goyet JD et al (2014) Hepatoblastoma state of the art: pre-treatment extent of disease, surgical resection guidelines and the role of liver transplantation. Curr Opin Pediatr 26:29–36

    Article  CAS  Google Scholar 

  10. Towbin AJ, Meyers RL, Woodley H et al (2018) 2017 PRETEXT: radiologic staging system for primary hepatic malignancies of childhood revised for the Paediatric Hepatic International Tumour Trial (PHITT). Pediatr Radiol 48:1–19

    Google Scholar 

  11. Uchida H, Sakamoto S, Sasaki K et al (2018) Surgical treatment strategy for advanced hepatoblastoma: resection versus transplantation. Pediatr Blood Cancer 65:e27383

    Article  CAS  Google Scholar 

  12. Browne M, Sher D, Grant D et al (2008) Survival after liver transplantation for hepatoblastoma: a 2-center experience. J Pediatr Surg 43:1973–1981

    Article  Google Scholar 

  13. Faraj W, Dar F, Marangoni G et al (2008) Liver transplantation for hepatoblastoma. Liver Transpl 14:1614–1619

    Article  Google Scholar 

  14. Fuchs J, Cavdar S, Blumenstock G et al (2017) POST-TEXT III and IV hepatoblastoma: extended hepatic resection avoids liver transplantation in selected cases. Ann Surg 266:318–323

    Article  Google Scholar 

  15. Lautz TB, Ben-Ami T, Tantemsapya N et al (2011) Successful nontransplant resection of POST-TEXT III and IV hepatoblastoma. Cancer 117:1976–1983

    Article  Google Scholar 

  16. Tajiri T, Kimura O, Fumino S et al (2012) Surgical strategies for unresectable hepatoblastomas. J Pediatr Surg 47:2194–2198

    Article  Google Scholar 

  17. Roebuck DJ, Aronson D, Clapuyt P et al (2007) 2005 PRETEXT: a revised staging system for primary malignant liver tumours of childhood developed by the SIOPEL group. Pediatr Radiol 37:123–132

    Article  Google Scholar 

  18. József Z, Maibach R, Shafford E et al (2010) Successful treatment of childhood high-risk hepatoblastoma with dose-intensive multiagent chemotherapy and surgery: final results of the SIOPEL-3HR study. J Clin Oncol 28:2584–2590

    Article  CAS  Google Scholar 

  19. López-Terrada D, Alaggio R, de Dávila MT et al (2014) Towards an international pediatric liver tumor consensus classification: proceedings of the Los Angeles COG liver tumors symposium. Mod Pathol 27:472–491

    Article  Google Scholar 

  20. Qiao GL, Li L, Cheng W et al (2014) Predictors of survival after resection of children with hepatoblastoma: a single Asian center experience. Eur J Surg Oncol 40:1533–1539

    Article  Google Scholar 

  21. Aronson DC, Weeda VB, Maibach R et al (2019) Microscopically positive resection margin after hepatoblastoma resection: what is the impact on prognosis? A Childhood Liver Tumours Strategy Group (SIOPEL) report. Eur J Cancer 106:126–132

    Article  Google Scholar 

  22. Meyers RL, Maibach R, Hiyama E 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

    Article  Google Scholar 

  23. Czauderna P, Haeberle B, Hiyama E et al (2016) The Children’s Hepatic tumors International Collaboration (CHIC): Novel global rare tumor database yields new prognostic factors in hepatoblastoma and becomes a research model. Eur J Cancer 52:92–101

    Article  Google Scholar 

  24. Katzenstein HM, Furman WL, Malogolowkin MH 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

    Article  CAS  Google Scholar 

  25. Shanmugam N, Scott JX, Kumar V et al (2017) Multidisciplinary management of hepatoblastoma in children: experience from a developing country. Pediatr Blood Cancer 64:e26249

    Article  Google Scholar 

  26. Haas RJD, Wicherts DA, Flores E et al (2008) R1 resection by necessity for colorectal liver metastases is it still a contraindication to surgery? Ann Surg 248:626–637

    PubMed  Google Scholar 

  27. Sakamoto S, Kasahara M, Mizuta K et al (2014) Nationwide survey of the outcomes of living donor liver transplantation for hepatoblastoma in Japan. Liver Transpl 20:333–346

    Article  Google Scholar 

  28. Wang LL, Filippi RZ, Zurakowski D et al (2010) Effects of neoadjuvant chemotherapy on hepatoblastoma: a morphologic and immunohistochemical study. Am J Surg Pathol 34:287–299

    Article  Google Scholar 

  29. Venkatramani R, Wang L, Malvar J et al (2012) Tumor necrosis predicts survival following neo-adjuvant chemotherapy for hepatoblastoma. Pediatr Blood Cancer 59:493–498

    Article  Google Scholar 

  30. József Z, Laurence B, Penelope B et al (2013) Dose-dense cisplatin-based chemotherapy and surgery for children with high-risk hepatoblastoma (SIOPEL-4): a prospective, single-arm, feasibility study. Lancet Oncol 14:834–842

    Article  CAS  Google Scholar 

Download references

Funding

No funding was received.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Long Li.

Ethics declarations

Conflict of interest

The authors declare that they have no competing interests.

Additional information

Publisher's Note

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

Electronic supplementary material

Below is the link to the electronic supplementary material.

Supplementary file1 (PDF 62 kb)

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Ren, X., Li, H., Diao, M. et al. Impact of microscopically margin-positive resection on survival in children with hepatoblastoma after hepatectomy: a retrospective cohort study. Int J Clin Oncol 25, 765–773 (2020). https://doi.org/10.1007/s10147-019-01573-0

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s10147-019-01573-0

Keywords

Navigation