Rubio-San-Simón Alba, Wilson William, Sironi Giovanna, le Deley Marie-Cécile, Palmerini Emanuela, Gaspar Nathalie, Piperno-Neumann Sophie, Hecker-Nolting Stefanie, Strauss Sandra, van Ewijk Roelof, Meazza Cristina
Pediatric Oncology and Hematology Department, Hospital Infantil Universitario Niño Jesús, Madrid, Spain.
Cancer Research UK and UCL Cancer Trials Centre, London, UK.
Br J Cancer. 2025 Oct;133(7):1020-1028. doi: 10.1038/s41416-025-03118-x. Epub 2025 Jul 29.
The outcome of patients with osteosarcoma after relapse is very poor, with a 5-year overall survival (OS) below 30%. Prognostic factors in this setting remain poorly defined, limiting treatment decisions. This study identifies key clinical and biological prognostic factors to guide future trials.
A systematic review and meta-analysis included studies published between 1976 and 2022 with ≥35 patients. Searches were performed in MEDLINE/PUBMED, EMBASE, and Cochrane. Study quality was assessed using QUIPS tool. Studies reporting any clinical outcomes in the post-relapse setting were included.
Nineteen studies involving 3245 patients were analysed. Nine prognostic factors were identified: relapse-free interval, site, number and size of lesions, resectability, sex, age, alkaline phosphatase levels and response to chemotherapy. Meta-analysis confirmed bilateral lung metastases worsened OS (HR 1.68, 95% CI 1.42-1.99). A relapse-free interval >24 months and complete surgical resection were consistently associated with better outcomes. Results on chemotherapy use were inconsistent. Substantial heterogeneity and low methodological quality were noted across studies.
Key prognostic factors should address clinical trial design. Stratification by resectability, number and site of lesions and relapse-free interval is essential to evaluate treatment efficacy. Standardised protocols are needed to improve outcomes and provide tailored strategies for relapsed osteosarcoma.
骨肉瘤患者复发后的预后非常差，5年总生存率低于30%。这种情况下的预后因素仍未明确界定，限制了治疗决策。本研究确定了关键的临床和生物学预后因素，以指导未来的试验。
一项系统评价和荟萃分析纳入了1976年至2022年间发表的、患者≥35例的研究。检索在MEDLINE/PUBMED、EMBASE和Cochrane中进行。使用QUIPS工具评估研究质量。纳入报告复发后任何临床结局的研究。
分析了19项涉及3245例患者的研究。确定了9个预后因素：无复发生存期、部位、病变数量和大小、可切除性、性别、年龄、碱性磷酸酶水平和对化疗的反应。荟萃分析证实双侧肺转移会使总生存率恶化（HR 1.68，95%CI 1.42-1.99）。无复发生存期>24个月和完全手术切除始终与更好的结局相关。关于化疗使用的结果不一致。各研究存在大量异质性和较低的方法学质量。
关键预后因素应纳入临床试验设计。根据可切除性、病变数量和部位以及无复发生存期进行分层对于评估治疗效果至关重要。需要标准化方案来改善结局，并为复发骨肉瘤提供个性化策略。
