Marec-Berard Perrine, Segura-Ferlay Céline, Tabone Marie-Dominique, Pacquement Helene, Lervat Cyril, Gentet Jean-Claude, Schmitt Claudine, Gaspar Nathalie, Brugières Laurence
Institute for Paediatric Haematology and Oncology, Leon Bérard Cancer Centre, University of Lyon, 1 Place Joseph Renaut, 69008 Lyon, France.
Unit of Biostatistics, Leon Bérard Cancer Centre, University of Lyon, 69008 Lyon, France.
Sarcoma. 2014;2014:475067. doi: 10.1155/2014/475067. Epub 2014 Feb 4.
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.
引言。骨肉瘤复发预后较差，5年生存率低于25%。我们描述了法国儿科肿瘤学会（SFCE）对45例复发性骨肉瘤患儿使用大剂量（HD）噻替派和自体干细胞移植（ASCT）的经验。
患者与方法。1992年至2004年间，53例患者在6个中心接受了HD噻替派（900mg/m²）治疗，随后进行ASCT。8例患者被排除在分析之外，我们对其余45例患者的临床、放射学和解剖病理学模式进行了回顾性研究。
结果。16名女孩和29名男孩（中位年龄15.9岁）在转移性疾病初次进展（2例）、首次复发（26例）以及第二次或第三次复发（17例）后接受了HD噻替派治疗。我们报告了12例放射学部分缓解以及31例组织学完全缓解中的9例。32例患者出现进一步复发，13例在手术切除残余病灶后持续处于完全缓解状态。3年总生存率为40%，3年无进展生存率为24%。复发延迟（从诊断起±2年）是一个预后因素（P = 0.011）。未发生急性严重不良事件。
结论。对于复发性骨肉瘤患者，使用HD噻替派和ASCT是可行的。一项关于复发性骨肉瘤在标准挽救化疗与大剂量噻替派联合干细胞救援之间的随机研究正在进行。
