title: Health state preference scores of children with spina bifida and their caregivers.
abstract: Cost-effectiveness evaluations of interventions to prevent or treat spina bifida require quality of life information measured as preference scores. Preference scores of caregivers also may be relevant. This study tested whether the preference scores of children with spina bifida and their caregivers would decrease as disability in the child increased. Families of children aged 0-17 with spina bifida (N = 98) were identified using a birth defect surveillance system in the state of Arkansas. Primary caregivers of children with spina bifida identified other families with an unaffected child (N = 49). Preference scores for child health states were determined using the Health Utilities Index--Mark 2 (HUI2). Caregiver preference scores were determined using the Quality of Well-Being (QWB) scale. Children with spina bifida were categorized into three disability levels according to the location of the child's lesion. Mean preference scores declined for both affected children and the primary caregiver as disability in the child increased. In multivariate analysis, the preference score of the child was a significant and positive predictor of the primary caregiver's preference score. A more modest association was found for caregiver health preference scores by lesion location. The findings can inform cost-effectiveness evaluations of interventions to treat or prevent spina bifida.
authors.affiliation: Center for Birth Defects Research and Prevention, University of Arkansas for Medical Sciences and Arkansas Children's Hospital, Little Rock, AR 72202-3591, USA. tilfordmickj@uams.edu
authors.forename: John M
authors.initials: JM
authors.lastname: Tilford
authors.forename: Scott D
authors.initials: SD
authors.lastname: Grosse
authors.forename: James M
authors.initials: JM
authors.lastname: Robbins
authors.forename: Jeffrey M
authors.initials: JM
authors.lastname: Pyne
authors.forename: Mario A
authors.initials: MA
authors.lastname: Cleves
authors.forename: Charlotte A
authors.initials: CA
authors.lastname: Hobbs
mesh_terms: D000293:Adolescent; D000328:Adult; D001131:Arkansas; D017028:Caregivers / Q000523:psychology*; D002648:Child; D002675:Child, Preschool; D003362:Cost-Benefit Analysis; D019988:Children with Disabilities / Q000523:psychology*; D018450:Disease Progression; D005190:Family / Q000523:psychology*; D005260:Female; D006304:Health Status; D006801:Humans; D008297:Male; D017060:Patient Satisfaction / Q000706:statistics & numerical data*; D011788:Quality of Life; D019057:Quality-Adjusted Life Years; D016135:Spinal Dysraphism / Q000503:physiopathology* / Q000517:prevention & control / Q000628:therapy*