Health utilities and parental quality of life effects for three rare conditions tested in newborns View Full Text


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Article Info

DATE

2019-12

AUTHORS

Norma-Jean Simon, John Richardson, Ayesha Ahmad, Angela Rose, Eve Wittenberg, Brittany D’Cruz, Lisa A. Prosser

ABSTRACT

BACKGROUND: Measurement of health utilities is required for economic evaluations. Few studies have evaluated health utilities for rare conditions; even fewer have incorporated disutility that may be experienced by caregivers. This study aimed to (1) estimate health utilities for three rare conditions currently recommended for newborn screening at the state or federal level, and (2) estimate the disutility, or spillover, experienced by parents of patients diagnosed with a rare, heritable disorder. METHODS: A stated-preference survey using a time trade-off approach elicited health utilities for Krabbe disease, phenylketonuria, and Pompe disease at varying stages (mild, moderate, severe) and onset of disease symptoms (infancy, childhood, and adulthood). We recruited respondents from a nationally representative community sample (n = 862). Respondents valued disease specific health states in three consecutive question frames: (1) adult health state (> = 18 years of age), (2) child health state (< 18 years of age), and (3) as a parent of a child with a condition (parent spillover state). Corresponding mean utilities were calculated for plausible disease states in adulthood and childhood. Mean disutility was estimated for parental spillover. Predictors of utilities were evaluated using a negative binomial regression model. RESULTS: More severe conditions and infant health states received lower estimated utility and greater estimated disutility among parents. Conditions with the lowest estimated health utilities were severe infantile Pompe disease (0.40, CI: 0.34-0.46) and infantile Krabbe disease (0.37, CI: 0.32-0.43). Disutility was evident for all conditions evaluated (range: 0.07-0.19). CONCLUSIONS: Rare childhood conditions are associated with substantial estimated losses in quality of life. Evidence of disutility among parents further warrants the inclusion of spillover effects in cost-effectiveness analyses. Continued research is needed to assess and measure the effects of childhood disease from a family perspective. More... »

PAGES

4

References to SciGraph publications

  • 2007-09. Measuring Health Preferences for Use in Cost-Utility and Cost-Benefit Analyses of Interventions in Children in PHARMACOECONOMICS
  • 2005-02. The prevalence of limited health literacy in JOURNAL OF GENERAL INTERNAL MEDICINE
  • 2013-06. Disutility of Illness for Caregivers and Families: A Systematic Review of the Literature in PHARMACOECONOMICS
  • 2014-02. Decision-making process for conditions nominated to the Recommended Uniform Screening Panel: statement of the US Department of Health and Human Services Secretary’s Advisory Committee on Heritable Disorders in Newborns and Children in GENETICS IN MEDICINE
  • 2010-03. The Well-Being of Parental Caregivers of Children with Activity Limitations in MATERNAL AND CHILD HEALTH JOURNAL
  • 2008-12. Evaluation of quality of life and description of the sociodemographic state in adolescent and young adult patients with phenylketonuria (PKU) in HEALTH AND QUALITY OF LIFE OUTCOMES
  • 2014-12. Cost-effectiveness of enzyme replacement therapy with alglucosidase alfa in classic-infantile patients with Pompe disease in ORPHANET JOURNAL OF RARE DISEASES
  • 2015-04. Comparison of EQ-5D and SF-6D utilities in Pompe disease in QUALITY OF LIFE RESEARCH
  • 2003-09. Whose quality of life? A commentary exploring discrepancies between health state evaluations of patients and the general public in QUALITY OF LIFE RESEARCH
  • 2010-09. Weighing the evidence for newborn screening for early-infantile Krabbe disease in GENETICS IN MEDICINE
  • 2011-10. Burden of illness of Pompe disease in patients only receiving supportive care in JOURNAL OF INHERITED METABOLIC DISEASE
  • 2006-08. The CarerQol instrument: A new instrument to measure care-related quality of life of informal caregivers for use in economic evaluations in QUALITY OF LIFE RESEARCH
  • 2012-06. Assessing health-related quality-of-life changes in informal caregivers: an evaluation in parents of children with major congenital anomalies in QUALITY OF LIFE RESEARCH
  • 2007-02. The course of life and quality of life of early and continuously treated Dutch patients with phenylketonuria in JOURNAL OF INHERITED METABOLIC DISEASE
  • Identifiers

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    http://scigraph.springernature.com/pub.10.1186/s41687-019-0093-6

    DOI

    http://dx.doi.org/10.1186/s41687-019-0093-6

    DIMENSIONS

    https://app.dimensions.ai/details/publication/pub.1111610116

    PUBMED

    https://www.ncbi.nlm.nih.gov/pubmed/30671727


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        "description": "BACKGROUND: Measurement of health utilities is required for economic evaluations. Few studies have evaluated health utilities for rare conditions; even fewer have incorporated disutility that may be experienced by caregivers. This study aimed to (1) estimate health utilities for three rare conditions currently recommended for newborn screening at the state or federal level, and (2) estimate the disutility, or spillover, experienced by parents of patients diagnosed with a rare, heritable disorder.\nMETHODS: A stated-preference survey using a time trade-off approach elicited health utilities for Krabbe disease, phenylketonuria, and Pompe disease at varying stages (mild, moderate, severe) and onset of disease symptoms (infancy, childhood, and adulthood). We recruited respondents from a nationally representative community sample (n\u00a0=\u2009862). Respondents valued disease specific health states in three consecutive question frames: (1) adult health state (>\u2009=\u200918\u2009years of age), (2) child health state (<\u200918\u2009years of age), and (3) as a parent of a child with a condition (parent spillover state). Corresponding mean utilities were calculated for plausible disease states in adulthood and childhood. Mean disutility was estimated for parental spillover. Predictors of utilities were evaluated using a negative binomial regression model.\nRESULTS: More severe conditions and infant health states received lower estimated utility and greater estimated disutility among parents. Conditions with the lowest estimated health utilities were severe infantile Pompe disease (0.40, CI: 0.34-0.46) and infantile Krabbe disease (0.37, CI: 0.32-0.43). Disutility was evident for all conditions evaluated (range: 0.07-0.19).\nCONCLUSIONS: Rare childhood conditions are associated with substantial estimated losses in quality of life. Evidence of disutility among parents further warrants the inclusion of spillover effects in cost-effectiveness analyses. Continued research is needed to assess and measure the effects of childhood disease from a family perspective.", 
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