Parental Tolerance of False-positive Newborn Screening Results

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.