title: Health status utilities in pediatrics: a systematic review of acute lymphoblastic leukemia.
abstract: Measuring utilities and health-related quality of life (HRQL) in children is challenging due to their cognitive abilities and changing developmental stages.
  
  
  
  . To identify methodological issues on utility measurements in children, we performed a systematic review on utilities measured with a single instrument, the Health Utilities Index (HUI), in pediatric acute lymphoblastic leukemia (ALL). The secondary goal was to facilitate future cost-utility analyses without the need for time-consuming assessments. Data Sources. PubMed, Embase, Cochrane Library, CINAHL, and PsycINFO were searched from inception to June 2012. Studies had to report on utility scores in pediatric ALL, either on or after treatment, to be included.
  
  
  
  . Fifteen studies were included. Most studies had methodological shortcomings, which mainly concerned
  
  
  
  and definition and representativeness of the study group. Utility scores were dependent on treatment variables, and there generally was an improvement in HRQL as treatment or survivorship advanced. In general, proxy-respondents were less reliable for subjective phenomena than for observable conditions. HUI2 and HUI3 scores were not interchangeable. Limitations. Studies may have been missed because no validated search method for utility studies exists, due to language bias or the exclusion of non-peer-reviewed papers.
  
  
  
  . Most studies in this review were methodologically suboptimal. Future developments should focus on including developmentally appropriate items for the whole pediatric age group. Adding disease-specific domains may enhance the sensitivity and responsiveness of instruments. Efforts should be undertaken to elicit valuation of health states from older children and teenagers as much as possible. For now, it remains difficult to make valid and informed decisions on the financing of interventions until health state valuation in children has become more methodologically robust.
authors.affiliation: Department of Pediatrics, Division of Oncology-Hematology, VU University Medical Center Amsterdam, Amsterdam, the Netherlands (RRLVL, GJLK).
authors.forename: Raphaële R L
authors.initials: RR
authors.lastname: van Litsenburg
authors.affiliation: Department of Pediatrics, VU University Medical Center Amsterdam, Amsterdam, the Netherlands (AK, RJBJG)
authors.forename: Annemieke
authors.initials: A
authors.lastname: Kunst
authors.affiliation: Department of Pediatric Psychology & Social Work, University Medical Center Utrecht, Utrecht, the Netherlands (JH)
authors.forename: Jaap
authors.initials: J
authors.lastname: Huisman
authors.affiliation: VU University Library Amsterdam, Amsterdam, the Netherlands (JCFK)
authors.forename: Johannes C F
authors.initials: JC
authors.lastname: Ket
authors.affiliation: Department of Pediatrics, Division of Oncology-Hematology, VU University Medical Center Amsterdam, Amsterdam, the Netherlands (RRLVL, GJLK).
authors.forename: Gertjan J L
authors.initials: GJ
authors.lastname: Kaspers
authors.affiliation: Department of Pediatrics, VU University Medical Center Amsterdam, Amsterdam, the Netherlands (AK, RJBJG)
authors.forename: Reinoud J B J
authors.initials: RJ
authors.lastname: Gemke
mesh_terms: D002648:Child; D002675:Child, Preschool; D006304:Health Status; D006801:Humans; D010372:Pediatrics; D019057:Quality-Adjusted Life Years
keywords: acute lymphoblastic leukemia; child; health status; health utilities index; health-related quality of life; pediatric; utility score