Parental tolerance of false-positive newborn screening results
Lisa A Prosser1, Joseph A Ladapo, Donna Rusinak
1Department of Ambulatory Care and Prevention, Harvard Medical School and Harvard Pilgrim Health Care, 133 Brookline Ave, Sixth Floor, Boston, MA 02215, USA. lisa_prosser@hphc.org
Insights
Parents show high tolerance for false-positive newborn screening results. This indicates that quality of life assessments for screening outcomes are crucial for evaluating expanded newborn screening programs for metabolic disorders.
Area of Science:
- Medical Genetics
- Public Health
- Health Economics
Background:
- Expanded newborn screening programs aim to detect metabolic disorders early.
- False-positive results can cause parental anxiety and impact perceived quality of life.
- Understanding parental preferences is vital for program evaluation.
Purpose of the Study:
- To measure parental tolerance for false-positive newborn screening results.
- To assess perceived quality of life associated with screening outcomes and health states for metabolic disorders.
- To inform cost-effectiveness analyses of expanded newborn screening.
Main Methods:
- Cross-sectional study involving parents of children with false-positive (n=91) and normal (n=50) newborn screening results.
- Telephone or in-person interviews conducted in Massachusetts and Pennsylvania.
- Quality of life measured using time trade-off and willingness-to-pay for various health states.
Main Results:
- Parents of children with false-positive results reported median quality of life values of 0 for time trade-off and willingness-to-pay.
- Parents of children with normal results reported median values of 1 week and $100, respectively (P = .07 and P < .001).
- Dietary treatments and developmental delay were rated as more burdensome than false-positive results or short-term hospitalization.
Conclusions:
- Parents demonstrate a high tolerance for false-positive newborn screening results.
- Parental preferences for screening outcomes and associated health states should be incorporated into economic evaluations.
- This research supports the inclusion of patient-reported outcomes in the assessment of newborn screening programs.
Objective:
To measure parental tolerance for a false-positive newborn screening result by assessing perceived quality of life for screening results and health states associated with expanded newborn screening programs for metabolic disorders.
Design:
Perceived quality of life was measured using time trade-off and willingness-to-pay questions for a false-positive newborn screening result and other conditions associated with metabolic disorders (ie, short-term hospitalization, dietary treatments, and developmental delay).
Setting:
Telephone or in-person interviews were conducted from October 1, 2004, through January 31, 2006, for 2 populations in Massachusetts and Pennsylvania.
Participants:
Parents of children who had a false-positive newborn screening result (n = 91) and parents of children with normal screening results (n = 50).
Intervention:
Telephone interviews.
Main Outcome Measures:
Time trade-off and willingness-to-pay amounts.
Results:
Median time trade-off and willingness-to-pay amounts for parents of children with false-positive screening results were both 0 compared with parents of children with normal screening results who had median values of 1 week (P = .07) and $100 (P < .001). For both populations, dietary treatments and developmental delay elicited higher time trade-off and willingness-to-pay amounts compared with ratings for experiencing a false-positive newborn screening result or short-term hospitalization because of an undiagnosed metabolic disorder.
Conclusions:
Parents have a high tolerance for false-positive newborn screening results. Preferences for test outcomes and other health states associated with screening for metabolic disorders should be included in cost-effectiveness and cost-benefit analyses of expanded newborn screening programs.
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