Comprehensive cost-utility analysis of newborn screening strategies

Research output: Contribution to journalArticle

98 Citations (Scopus)

Abstract

BACKGROUND. Inborn errors of metabolism are a significant cause of morbidity and death among children. Inconsistencies in how individual states arrive at screening strategies, however, lead to marked variations in testing between states. OBJECTIVE. To determine the cost-effectiveness of each component test of a multitest newborn screening program, including screening for phenylketonuria, congenital adrenal hyperplasia, congenital hypothyroidism, biotinidase deficiency, maple syrup urine disease, galactosemia, homocystinuria, and medium-chain acyl-CoA dehydrogenase deficiency. METHODS. A decision model was used, with cohort studies, government reports, secondary analyses, and other sources. Discounted costs, quality-adjusted life-years (QALYs), and incremental cost-effectiveness ratios were measured. RESULTS. All except 2 screening tests dominated the "no-test" strategy. The 2 exceptions were screening for congenital adrenal hyperplasia, which cost slightly more than $20 000 per QALY gained, and screening for galactosemia, which cost $94 000 per QALY gained. The screening test with the lowest expected cost was tandem mass spectrometry. The results found in our base-case analysis were stable across variations in nearly all variables. In instances in which changes in risks, sequelae, costs, or utilities did affect our results, the variation from base-case estimates was quite large. CONCLUSIONS. Newborn screening seems to be one of the rare health care interventions that is beneficial to patients and, in many cases, cost saving. Over the long term, funding comprehensive newborn screening programs is likely to save money for society.

Original languageEnglish
JournalPediatrics
Volume117
Issue number5
DOIs
StatePublished - May 2006

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Cost-Benefit Analysis
Newborn Infant
Costs and Cost Analysis
Quality-Adjusted Life Years
Galactosemias
Congenital Adrenal Hyperplasia
Biotinidase Deficiency
Maple Syrup Urine Disease
Homocystinuria
Congenital Hypothyroidism
Inborn Errors Metabolism
Phenylketonurias
Tandem Mass Spectrometry
Cause of Death
Cohort Studies
Morbidity
Delivery of Health Care

Keywords

  • Cost utility
  • Cost-effectiveness
  • Decision analysis
  • Newborn
  • Screening

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health

Cite this

Comprehensive cost-utility analysis of newborn screening strategies. / Carroll, Aaron; Downs, Stephen.

In: Pediatrics, Vol. 117, No. 5, 05.2006.

Research output: Contribution to journalArticle

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AB - BACKGROUND. Inborn errors of metabolism are a significant cause of morbidity and death among children. Inconsistencies in how individual states arrive at screening strategies, however, lead to marked variations in testing between states. OBJECTIVE. To determine the cost-effectiveness of each component test of a multitest newborn screening program, including screening for phenylketonuria, congenital adrenal hyperplasia, congenital hypothyroidism, biotinidase deficiency, maple syrup urine disease, galactosemia, homocystinuria, and medium-chain acyl-CoA dehydrogenase deficiency. METHODS. A decision model was used, with cohort studies, government reports, secondary analyses, and other sources. Discounted costs, quality-adjusted life-years (QALYs), and incremental cost-effectiveness ratios were measured. RESULTS. All except 2 screening tests dominated the "no-test" strategy. The 2 exceptions were screening for congenital adrenal hyperplasia, which cost slightly more than $20 000 per QALY gained, and screening for galactosemia, which cost $94 000 per QALY gained. The screening test with the lowest expected cost was tandem mass spectrometry. The results found in our base-case analysis were stable across variations in nearly all variables. In instances in which changes in risks, sequelae, costs, or utilities did affect our results, the variation from base-case estimates was quite large. CONCLUSIONS. Newborn screening seems to be one of the rare health care interventions that is beneficial to patients and, in many cases, cost saving. Over the long term, funding comprehensive newborn screening programs is likely to save money for society.

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