title: 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 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.
  
  
  METHODS
  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.
  
  
  RESULTS
  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.
  
  
  CONCLUSIONS
  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.
authors.affiliation: Seattle Children's Hospital, Seattle, Washington.|University of Washington, Seattle, Washington.
authors.forename: Jamie
authors.initials: J
authors.lastname: Stokke
authors.affiliation: Hospital for Sick Children, Toronto, Ontario, Canada.
authors.forename: Lillian
authors.initials: L
authors.lastname: Sung
authors.affiliation: Hospital for Sick Children, Toronto, Ontario, Canada.|University of Toronto, Toronto, Ontario, Canada.
authors.forename: Abha
authors.initials: A
authors.lastname: Gupta
authors.affiliation: Seattle Children's Hospital, Seattle, Washington.|University of Washington, Seattle, Washington.
authors.forename: Antoinette
authors.initials: A
authors.lastname: Lindberg
authors.affiliation: Seattle Children's Hospital, Seattle, Washington.|University of Washington, Seattle, Washington.|Fred Hutchinson Cancer Research Center, Seattle, Washington.|Treuman Katz Center for Pediatric Bioethics, Seattle, Washington.
authors.forename: Abby R
authors.initials: AR
authors.lastname: Rosenberg
mesh_terms: D000293:Adolescent; D000367:Age Factors; D000671:Amputation, Surgical / Q000523:psychology; D001859:Bone Neoplasms / Q000523:psychology* / Q000601:surgery; D002648:Child; D002675:Child, Preschool; D003430:Cross-Sectional Studies; D006233:Persons with Disabilities / Q000523:psychology; D005260:Female; D006801:Humans; D023821:Limb Salvage / Q000523:psychology; D008297:Male; D011446:Prospective Studies; D011788:Quality of Life; D020127:Recovery of Function; D012509:Sarcoma / Q000523:psychology / Q000601:surgery; D057566:Self Report; D011795:Surveys and Questionnaires; D017741:Survivors / Q000523:psychology*; D055815:Young Adult
keywords: Ewing Sarcoma; bone cancer; late effects; osteosarcoma; patient-reported outcomes; pediatric cancer; quality of life; sarcoma; survivorship
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