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Volume 2014, Article ID 475067, 7 pages
Clinical Study

High Dose Thiotepa in Patients with Relapsed or Refractory Osteosarcomas: Experience of the SFCE Group

1Institute for Paediatric Haematology and Oncology, Leon Bérard Cancer Centre, University of Lyon, 1 Place Joseph Renaut, 69008 Lyon, France
2Unit of Biostatistics, Leon Bérard Cancer Centre, University of Lyon, 69008 Lyon, France
3Department of Paediatric Haematology and Oncology, Hôpital Armand Trousseau, 75012 Paris, France
4Department of Paediatrics, Curie Institute, 75248 Paris, France
5Department of Paediatrics, Children’s Hospital, 59037 Lilles, France
6Department of Paediatrics, La Timone Hospital, 13385 Marseille, France
7Department of Paediatrics, Children’s Hospital, 54000 Nancy, France
8Department of Paediatrics, Gustave Roussy Institute, 94805 Villejuif, France

Received 12 November 2013; Accepted 15 December 2013; Published 4 February 2014

Academic Editor: Carola A. S. Arndt

Copyright © 2014 Perrine Marec-Berard et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.


Introduction. Osteosarcoma relapse has a poor prognosis, with less than 25% survival at 5 years. We describe the experience of the French Society of Paediatric Oncology (SFCE) with high dose (HD) thiotepa and autologous stem cell transplantation (ASCT) in 45 children with relapsed osteosarcoma. Patients and Methods. Between 1992 and 2004, 53 patients received HD thiotepa (900 mg/m2) followed by ASCT in 6 centres. Eight patients were excluded from analysis, and we retrospectively reviewed the clinical radiological and anatomopathological patterns of the 45 remaining patients. Results. Sixteen girls and 29 boys (median age, 15.9 years) received HD thiotepa after initial progression of metastatic disease (2), first relapse (26), and second or third relapse (17). We report 12 radiological partial responses and 9 of 31 histological complete responses. Thirty-two patients experienced further relapses, and 13 continued in complete remission after surgical resection of the residual disease. Three-year overall survival was 40%, and 3-year progression-free survival was 24%. Delay of relapse (+/− 2 years from diagnosis) was a prognostic factor ( ). No acute toxic serious adverse event occurred. Conclusion. The use of HD thiotepa and ASCT is feasible in patients with relapsed osteosarcoma. A randomized study for recurrent osteosarcoma between standard salvage chemotherapy and high dose thiotepa with stem cell rescue is ongoing.