Systematic review and meta‐analysis of objective and subjective quality of life among pediatric, adolescent, and young adult bone tumor survivors

Abstract

Background Pediatric, adolescent and young adult (AYA) 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. Procedure 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 versus 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. Results Twenty-two of 452 unique manuscripts were included in qualitative syntheses, 8 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. Conclusion 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.

Effect of Temporal Changes in Therapeutic Exposure on Self-reported Health Status in Childhood Cancer Survivors

“…However, there was no improvement among osteosarcoma survivors, where the biggest change in treatment was replacement of amputation with limb-sparing surgery. Our findings are consistent with literature that indicates a dose-response association (7% (1-13%) per Gray) between CRT dose and risk for adverse events among childhood brain tumor survivors (40), and with meta-analyses that report no difference in long-term disability among childhood sarcoma survivors who require amputation compared to those who had limb-sparing surgery (41). …”

Quality of Life in Children With Advanced Cancer: A Report From the PediQUEST Study

“…We also had relatively little racial or ethnic diversity in our sample and could not assess potential cultural differences in HRQOL. Importantly, poor child HRQOL has been associated with other unexamined factors such as socioeconomic status and parent physical and emotional health (23, 40, 44, 47, 58, 59). …”

“…Three recent systematic reviews identified a wide array of variables associated with HRQOL in children with cancer (26, 36, 40). Factors consistently associated with poor HRQOL include concurrent cancer therapy (41–45), higher treatment-intensity (23, 46, 47), poor prognosis or history of relapse (23, 26), older age (25, 36, 40), cancer-type (where patients with sarcomas or brain tumors have poorer HRQOL) (36, 40, 45, 46), and female sex (23, 25, 40, 44).…”

“…Factors consistently associated with poor HRQOL include concurrent cancer therapy (41–45), higher treatment-intensity (23, 46, 47), poor prognosis or history of relapse (23, 26), older age (25, 36, 40), cancer-type (where patients with sarcomas or brain tumors have poorer HRQOL) (36, 40, 45, 46), and female sex (23, 25, 40, 44). While existing results may help identify patients at risk for poor HRQOL (23), immediately modifiable factors of HRQOL, such as symptoms, have been insufficiently described (26, 36).…”

Health-Related Quality of Life in Children with Cancer

“…Previous studies have found children who underwent either limb sparing or amputation reported no significant differences between either procedures in terms of HRQOL outcomes. 79 Our analysis of HRQOL in children with osteosarcoma found similar results, with no noted differences in HRQOL between children on the two surgical procedures. However, this outcome has been reported to change as children with osteosarcoma progress into survivorship, with evidence suggesting limb-sparing procedures offer higher HRQOL outcomes for survivors of lower-limb osteosarcoma in comparison to survivors with amputations.…”