Stokke Jamie, Sung Lillian, Gupta Abha, Lindberg Antoinette, Rosenberg Abby R
Seattle Children's Hospital, Seattle, Washington.
University of Washington, Seattle, Washington.
Pediatr Blood Cancer. 2015 Sep;62(9):1616-29. doi: 10.1002/pbc.25514. Epub 2015 Mar 27.
Pediatric, adolescent, and young adult survivors of bone sarcomas are at risk for poor quality of life (QOL). We conducted a systematic review and meta-analysis to summarize the literature describing QOL in this population and differences in QOL based on local control procedures.
Included studies described ≥5 patients <25 years old who had completed local control treatment for bone sarcoma, defined QOL as a main outcome, and measured it with a validated instrument. Data extraction and quality assessments were conducted with standardized tools. Meta-analyses compared QOL based on surgical procedure (limb-sparing vs. amputation) and were stratified by assessment type (objective physical function, clinician-assessed disability, patient-reported disability, and patient-reported QOL). Effect sizes were reported as the standard mean difference when multiple instruments were used within a comparison and weighted mean difference otherwise. All were weighted by inverse variance and modeled with random effects.
Twenty-two of 452 unique manuscripts were included in qualitative syntheses, eight of which were included in meta-analyses. Manuscripts were heterogeneous with respect to included patient populations (age, tumor type, time since treatment) and QOL instruments. Prospective studies suggested that QOL improves over time, and that female sex and older age at diagnosis are associated with poor QOL. Meta-analyses showed no differences in outcomes between patients who underwent limb-sparing versus amputation for local control.
QOL studies among children and AYAs with bone sarcoma are remarkably diverse, making it difficult to detect trends in patient outcomes. Future research should focus on standardized QOL instruments and interpretations.
骨肉瘤的儿童、青少年及年轻成人幸存者存在生活质量（QOL）较差的风险。我们进行了一项系统综述和荟萃分析，以总结描述该人群生活质量以及基于局部控制程序的生活质量差异的文献。
纳入的研究描述了≥5名年龄<25岁且已完成骨肉瘤局部控制治疗的患者，将生活质量定义为主要结局，并使用经过验证的工具进行测量。使用标准化工具进行数据提取和质量评估。荟萃分析比较了基于手术程序（保肢与截肢）的生活质量，并按评估类型（客观身体功能、临床医生评估的残疾、患者报告的残疾和患者报告的生活质量）进行分层。当在比较中使用多种工具时，效应量报告为标准平均差，否则报告为加权平均差。所有效应量均采用逆方差加权并采用随机效应模型。
452篇独特手稿中有22篇纳入定性综合分析，其中8篇纳入荟萃分析。手稿在纳入的患者群体（年龄、肿瘤类型、治疗后时间）和生活质量工具方面存在异质性。前瞻性研究表明，生活质量随时间改善，女性性别和诊断时年龄较大与生活质量较差相关。荟萃分析显示，接受保肢手术与截肢手术进行局部控制的患者在结局方面没有差异。
骨肉瘤儿童和青少年及年轻成人的生活质量研究差异显著，难以检测患者结局的趋势。未来研究应侧重于标准化的生活质量工具和解释。
