Advertisement

Pediatric Surgery International

, Volume 27, Issue 1, pp 1–8 | Cite as

Outcome of hepatoblastomas treated using the Japanese Study Group for Pediatric Liver Tumor (JPLT) protocol-2: report from the JPLT

  • Tomoro Hishiki
  • Tadashi Matsunaga
  • Fumiaki Sasaki
  • Michihiro Yano
  • Kohmei Ida
  • Hiroshi Horie
  • Satoshi Kondo
  • Ken-Ichiro Watanabe
  • Takaharu Oue
  • Tatsuro Tajiri
  • Arata Kamimatsuse
  • Naomi Ohnuma
  • Eiso Hiyama
Original Article

Abstract

Background

In the recent years, surgical resection with pre- and/or postoperative chemotherapy has markedly improved the survival rate of hepatoblastoma patients. We herein report the results of patients treated with the current protocol of the Japanese Study Group for Pediatric Liver Tumor, JPLT-2.

Methods

A total of 279 patients with malignant liver tumor were enrolled in JPLT-2. Data from 212 hepatoblastoma cases were analyzed. PRETEXT I patients were treated with primary resection followed by low doses of cisplatin–pirarubicin (tetrahydropyranyl-adriamycin). Otherwise, patients received preoperative cisplatin–pirarubicin (CITA), followed by surgery and postoperative chemotherapy. Ifosfamide, pirarubicin, etoposide, and carboplatin (ITEC) were given as a salvage treatment. High-dose chemotherapy with hematopoietic stem cell transplantation (SCT) was reserved for patients with metastatic diseases.

Results

The 5-year overall survival rate (OS) in non-metastatic cases was 100% for PRETEXT I, 87.1% for PRETEXT II, 89.7% for PRETEXT III, and 78.3% for PRETEXT IV. The 5-year OS in metastatic cases was 43.9%. The outcome in non-metastatic PRETEXT IV cases was markedly improved, while the results of metastatic tumors remained poor.

Conclusions

JPLT-2 protocol achieved satisfactory survival among children with non-metastatic hepatoblastoma. New approaches are needed for patients with metastatic diseases.

Keywords

Hepatoblastoma Prognostic factors PRETEXT Survival 

Notes

Acknowledgments

We would like to thank all the staff at institutes that participated in JPLT for enrolling their patients into the study. We also thank the following previous core members of JPLT, who participated in designing the initial JPLT-2 protocol in 1998; Drs. Yutaka Hayashi, Akira Hayashi, Kohei Hashizume, Hideo Mugishima.

Conflict of interest

The authors declare that they have no conflicts of interest.

References

  1. 1.
    Ortega JA, Douglass EC, Feusner JH et al (2000) Randomized comparison of cisplatin/vincristine/fluorouracil and cisplatin/continuous infusion doxorubicin for treatment of pediatric hepatoblastoma: a report from the Children’s Cancer Group and the Pediatric Oncology Group. J Clin Oncol 18:2665–2675PubMedGoogle Scholar
  2. 2.
    Fuchs J, Rydzynski J, von Shweinitz D et al (2002) Pretreatment prognostic factors and treatment results in children with hepatoblastoma. A report from the German Cooperative Pediatric Liver Tumour Study HB94. Cancer 95:172–182CrossRefPubMedGoogle Scholar
  3. 3.
    Perilongo G, Shafford E, Maibach R 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 40:411–421CrossRefPubMedGoogle Scholar
  4. 4.
    Sasaki F, Matsunaga T, Iwafuchi M et al (2002) Outcome of hepatoblastoma treated with the JPLT-1 (Japanese Study Group for Pediatric Liver Tumor) Protocol-1: a report from the Japanese Study Group for Pediatric Liver Tumor. J Pediatr Surg 37:851–856CrossRefPubMedGoogle Scholar
  5. 5.
    Katzenstein HM, London WB, Douglass EC et al (2002) Treatment of unresectable and metastatic hepatoblastoma: a Pediatric Oncology Group Phase II Study. J Clin Oncol 20:3438–3444CrossRefPubMedGoogle Scholar
  6. 6.
    Matsunaga T, Sasaki F, Ohira M et al (2003) Analysis of treatment outcome for children with recurrent or metastatic hepatoblastoma. Pediatr Surg Int 19:142–146PubMedGoogle Scholar
  7. 7.
    Malogolowkin MH, Katzenstein H, Krailo MD et al (2006) Intensified platinum therapy is an ineffective strategy for improving outcome in pediatric patients with advanced hepatoblastoma. J Clin Oncol 24:2879–2884CrossRefPubMedGoogle Scholar
  8. 8.
    Czauderna P, Otte JB, Aronson DC 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 41:1031–1036CrossRefPubMedGoogle Scholar
  9. 9.
    Tagge EP, Tagge DU, Reyes J et al (1992) Resection, including transplantation, for hepatoblastoma and hepatocellular carcinoma: impact on survival. J Pediatr Surg 27:292–297CrossRefPubMedGoogle Scholar
  10. 10.
    King DR, Ortega J, Campbell J et al (1991) The surgical management of children with incompletely resected hepatic cancer is facilitated by intensive chemotherapy. J Pediatr Surg 26:1074–1081CrossRefPubMedGoogle Scholar
  11. 11.
    Perilongo G, Maibach R, Shafford E et al (2009) Cisplatin versus cisplatin plus doxorubicin for standard-risk hepatoblastoma. N Engl J Med 361:1662–1670CrossRefPubMedGoogle Scholar
  12. 12.
    Brown J, Perilongo G, Shafford E et al (2000) Pretreatment prognostic factors for children with hepatoblatoma––results from the International Society of Paediatric Oncology (SIOP) Study SIOPEL 1. Eur J Cancer 36:1418–1425CrossRefPubMedGoogle Scholar
  13. 13.
    Aronson DC, Schnater JM, Staalman CR et al (2005) Predictive value of the pretreatment extent of disease system in hepatoblastoma: results from the International Society of Pediatric Oncology Liver Tumor Study Grou p SIOPEL-1 study. J Clin Oncol 23:1245–1252CrossRefPubMedGoogle Scholar
  14. 14.
    Meyers RL, Rowland JR, Krailo M et al (2009) Predictive power of pretreatment prognostic factors in children with hepatoblastoma: a report from the Children’s Oncology Group. Pediatr Blood Cancer 53:1016–1022CrossRefPubMedGoogle Scholar
  15. 15.
    Koh E, Ueda Y, Nakamura T, Kobayashi A, Katsuta S, Takahashi H (2002) Apoptosis in young rats with adriamycin-induced cardiomyopathy––comparison with pirarubicin, a new anthracycline derivative. Pediatr Res 51:256–259CrossRefPubMedGoogle Scholar
  16. 16.
    Zsíros J, 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 28:2584–2590CrossRefPubMedGoogle Scholar
  17. 17.
    Otte JB, Oritchars J, Aronson DC et al (2004) Liver transplantation for hepatoblastoma: results from the international society of pediatric oncology (SIOP) study SIOPEL-1 and review of the world experience. Pediatr Blood Cancer 42:74–83CrossRefPubMedGoogle Scholar
  18. 18.
    Otte JB, de Goyet JV, Reding R (2005) Liver transplantation for hepatoblastoma: indications and contraindications in the modern era. Pediatr Transplant 9:557–565CrossRefPubMedGoogle Scholar
  19. 19.
    Reyes JD, Carr B, Dvorchik I et al (2000) Liver transplantation and chemotherapy for hepatoblastoma and hepatocellular cancer in childhood and adolescence. J Pediatr 136:795–804CrossRefPubMedGoogle Scholar
  20. 20.
    Tanaka K, Uemoto S, Tokunaga Y et al (1993) Surgical techniques and innovations in living related liver transplantation. Ann Surg 217:82–91CrossRefPubMedGoogle Scholar
  21. 21.
    Kasahara M, Ueda M, Haga H et al (2005) Living-donor liver transplantation for hepatoblastoma. Am J Transplant 5:2229–2235CrossRefPubMedGoogle Scholar
  22. 22.
    Nishimura S, Sato T, Fujita N et al (2002) High-dose chemotherapy in children with metastatic hepatoblastoma. Pediatr Int 44:300–305CrossRefPubMedGoogle Scholar
  23. 23.
    Yoshinari M, Imaizumi M, Hayashi Y et al (1998) Peripheral blood stem cell transplantation for hepatoblastoma with microscopical residue: a therapeutic approach for incompletely resected tumor. Tohoku J Exp Med 184:247–254CrossRefPubMedGoogle Scholar
  24. 24.
    Kitahara S, Makuuchi M, Ishizone S et al (1995) Successful left trisegmentectomy for ruptured hepatoblastoma using intraoperative transarterial embolization. J Pediatr Surg 30:1709–1712CrossRefPubMedGoogle Scholar
  25. 25.
    Iida T, Senaga M, Takeuchi Y et al (2004) Successful resection of a ruptured hepatoblastoma prior to chemotherapy: report of a case. Surg Today 34:710–714CrossRefPubMedGoogle Scholar
  26. 26.
    Hara J, Osugi Y, Ohta H et al (1998) Double-conditioning regimens consisting of thiotepa, melphalan and busulfan with stem cell rescue for the treatment of pediatric tumors. Bone Marrow Transplant 22:7–12CrossRefPubMedGoogle Scholar
  27. 27.
    Suita S, Tajiri T, Takamatsu H et al (2004) Improved survival outcome for hepatoblastoma based on an optimal chemotherapeutic regimen––a report from the study group for pediatric solid malignant tumors in the Kyushu area. J Pediatr Surg 39:195–198CrossRefPubMedGoogle Scholar
  28. 28.
    von Schweinitz D, Hecker H, Harmas D et al (1995) Complete resection before development of drug resistance is essential for survival from advanced hepatoblastoma––a report from the German Cooperative Pediatric Liver Tumor Study HB-89. J Pediatr Surg 30:845–852CrossRefGoogle Scholar
  29. 29.
    Katzenstein HM, Rigsby C, Shaw PH et al (2002) Novel therapeutic approaches in the treatment of children with hepatoblastoma. J Pediatr Hematol Oncol 24:751–755CrossRefPubMedGoogle Scholar
  30. 30.
    Palmer RD, Williams DM (2003) Dramatic response of multiply relapsed hepatoblastoma to irinotecan (CPT-11). Med Pediatr Oncol 41:78–80CrossRefPubMedGoogle Scholar
  31. 31.
    Ijichi O, Ishikawa S, Shinkoda Y et al (2006) Response of heavily treated and relapsed hepatoblastoma in the transplanted liver to single-agent therapy with irinotecan. Pediatr Transplant 10(5):635–638CrossRefPubMedGoogle Scholar
  32. 32.
    Bomgaars LR, Bernstein M, Krailo M et al (2007) Phase II trial of irinotecan in children with refractory solid tumors: a Children’s Oncology Group Study. J Clin Oncol 25:4622–4627CrossRefPubMedGoogle Scholar
  33. 33.
    Qayed M, Powell C, Morgan ER et al (2010) Irinotecan as maintenance therapy in high-risk hepatoblastoma. Pediatr Blood Cancer 54:761–763PubMedGoogle Scholar

Copyright information

© Springer-Verlag 2010

Authors and Affiliations

  • Tomoro Hishiki
    • 1
  • Tadashi Matsunaga
    • 1
  • Fumiaki Sasaki
    • 2
  • Michihiro Yano
    • 3
  • Kohmei Ida
    • 4
  • Hiroshi Horie
    • 5
  • Satoshi Kondo
    • 6
  • Ken-Ichiro Watanabe
    • 7
  • Takaharu Oue
    • 8
  • Tatsuro Tajiri
    • 9
  • Arata Kamimatsuse
    • 10
  • Naomi Ohnuma
    • 1
  • Eiso Hiyama
    • 10
  1. 1.Department of Pediatric SurgeryChiba University Graduate School of MedicineChibaJapan
  2. 2.Department of Pediatric SurgeryHokkaido University Graduate School of MedicineSapporoJapan
  3. 3.Department of PediatricsAkita University School of MedicineAkitaJapan
  4. 4.Department of Pediatrics, Graduate School of MedicineUniversity of TokyoTokyoJapan
  5. 5.Division of PathologyChiba Children’s HospitalChibaJapan
  6. 6.Division of Pediatric Surgery and Transplant SurgeryNagoya City University Medical SchoolNagoyaJapan
  7. 7.Department of Pediatrics, Graduate School of MedicineKyoto UniversityKyotoJapan
  8. 8.Division of Pediatric Surgery, Department of SurgeryOsaka University Graduate School of MedicineOsakaJapan
  9. 9.Department of Pediatric Surgery, Graduate School of Medical SciencesKyushu UniversityFukuokaJapan
  10. 10.Department of Pediatric Surgery, Graduate School of Biomedical ScienceHiroshima UniversityHiroshimaJapan

Personalised recommendations