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.


METHODS
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).


METHODS
Telephone or in-person interviews were conducted from October 1, 2004, through January 31, 2006, for 2 populations in Massachusetts and Pennsylvania.


METHODS
Parents of children who had a false-positive newborn screening result (n = 91) and parents of children with normal screening results (n = 50).


METHODS
Telephone interviews.


METHODS
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.
Prosser
Lisa A
LA
Department 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
Ladapo
Joseph A
JA
Rusinak
Donna
D
Waisbren
Susan E
SE