title: High Dose Thiotepa in Patients with Relapsed or Refractory Osteosarcomas: Experience of the SFCE Group.
abstract: 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/m(2)) 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 (P = 0.011). 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.
authors.affiliation: Institute for Paediatric Haematology and Oncology, Leon Bérard Cancer Centre, University of Lyon, 1 Place Joseph Renaut, 69008 Lyon, France.
authors.forename: Perrine
authors.identifier: 0000-0001-5315-5864
authors.initials: P
authors.lastname: Marec-Berard
authors.affiliation: Unit of Biostatistics, Leon Bérard Cancer Centre, University of Lyon, 69008 Lyon, France.
authors.forename: Céline
authors.initials: C
authors.lastname: Segura-Ferlay
authors.affiliation: Department of Paediatric Haematology and Oncology, Hôpital Armand Trousseau, 75012 Paris, France.
authors.forename: Marie-Dominique
authors.initials: MD
authors.lastname: Tabone
authors.affiliation: Department of Paediatrics, Curie Institute, 75248 Paris, France.
authors.forename: Helene
authors.initials: H
authors.lastname: Pacquement
authors.affiliation: Department of Paediatrics, Children's Hospital, 59037 Lilles, France.
authors.forename: Cyril
authors.initials: C
authors.lastname: Lervat
authors.affiliation: Department of Paediatrics, La Timone Hospital, 13385 Marseille, France.
authors.forename: Jean-Claude
authors.initials: JC
authors.lastname: Gentet
authors.affiliation: Department of Paediatrics, Children's Hospital, 54000 Nancy, France.
authors.forename: Claudine
authors.initials: C
authors.lastname: Schmitt
authors.affiliation: Department of Paediatrics, Gustave Roussy Institute, 94805 Villejuif, France.
authors.forename: Nathalie
authors.initials: N
authors.lastname: Gaspar
authors.affiliation: Department of Paediatrics, Gustave Roussy Institute, 94805 Villejuif, France.
authors.forename: Laurence
authors.initials: L
authors.lastname: Brugières