ABSTRACT
BACKGROUND: The purpose of this study was to assess the construct validity of the Aberrant Behaviour Checklist-utility index (ABC-UI) by examining the relationship between healthcare resource utilisation by patients with fragile X syndrome (FXS) as well as burden experienced by their caregivers. METHOD: In 2011, a total of 350 US caregivers of individuals with FXS completed a questionnaire that captured information on FXS-related burden as well as the ABC-Community. Using the ABC-UI, a condition-specific outcome measure derived from the ABC-Community, five utility index categories were created: very low (0.00-0.33); low (0.34-0.66); moderate (0.67-0.77); high (0.78-0.89); and very high (0.90-1.00). Multivariable regression models examined the association between the utility value and nine burden-related outcomes. RESULTS: Approximately 2% of individuals with FXS were in the very low utility index category, 31% low, 27% moderate, 38% high and 3% very high. The median utility value was 0.74. Women with FXS and adults 18 years and older had higher values. Regression results indicate that individuals with higher utility values were more likely to have fewer specialist visits, use fewer prescription medications, need fewer hours of unpaid caregiving, inflict fewer caregiver injuries and have caregivers with fewer mental health provider visits. CONCLUSIONS: The ABC-UI appears to function well as condition-specific outcome measure, and as an indicator of health-related quality-of-life and economic burden in individuals with FXS. Among patients with FXS in the US and their caregivers, significant differences in health care resource utilisation and burden exist across health state utility categories.
Subject(s)
Checklist/standards , Cost of Illness , Fragile X Syndrome , Patient Acceptance of Health Care/statistics & numerical data , Problem Behavior , Registries/statistics & numerical data , Adolescent , Adult , Caregivers , Child , Child, Preschool , Family , Female , Fragile X Syndrome/economics , Fragile X Syndrome/physiopathology , Fragile X Syndrome/therapy , Humans , Male , Reproducibility of Results , Young AdultSubject(s)
Government Agencies/organization & administration , Health Benefit Plans, Employee/organization & administration , Utilization Review/organization & administration , Contract Services , Health Benefit Plans, Employee/economics , Health Benefit Plans, Employee/standards , Military Medicine/economics , United States , United States Department of Veterans Affairs/organization & administrationABSTRACT
Family Resource Centers/Youth Services Centers were mandated by law as part of the Kentucky Education Reform Act in response to changing needs of children and their families. These school-based centers provide services to maintain basic necessities for children and their families, enhancing the children's health and their ability to learn. This article describes how centers are developed, funded, staffed, governed, and evaluated as well as what services they provide.
