The health system impact of false positive newborn screening results for medium-chain acyl-CoA dehydrogenase deficiency: a cohort study

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dc.contributor.authorKaraceper, Maria D
dc.contributor.authorChakraborty, Pranesh
dc.contributor.authorCoyle, Doug
dc.contributor.authorWilson, Kumanan
dc.contributor.authorKronick, Jonathan B
dc.contributor.authorHawken, Steven
dc.contributor.authorDavies, Christine
dc.contributor.authorBrownell, Marni
dc.contributor.authorDodds, Linda
dc.contributor.authorFeigenbaum, Annette
dc.contributor.authorFell, Deshayne B
dc.contributor.authorGrosse, Scott D
dc.contributor.authorGuttmann, Astrid
dc.contributor.authorLaberge, Anne-Marie
dc.contributor.authorMhanni, Aizeddin
dc.contributor.authorMiller, Fiona A
dc.contributor.authorMitchell, John J
dc.contributor.authorNakhla, Meranda
dc.contributor.authorPrasad, Chitra
dc.contributor.authorRockman-Greenberg, Cheryl
dc.contributor.authorSparkes, Rebecca
dc.contributor.authorWilson, Brenda J
dc.contributor.authorPotter, Beth K
dc.date.accessioned2016-02-04T15:00:19Z
dc.date.available2016-02-04T15:00:19Z
dc.date.issued2016-02-03
dc.date.updated2016-02-03T17:04:20Z
dc.description.abstractAbstract Background There is no consensus in the literature regarding the impact of false positive newborn screening results on early health care utilization patterns. We evaluated the impact of false positive newborn screening results for medium-chain acyl-CoA dehydrogenase deficiency (MCADD) in a cohort of Ontario infants. Methods The cohort included all children who received newborn screening in Ontario between April 1, 2006 and March 31, 2010. Newborn screening and diagnostic confirmation results were linked to province-wide health care administrative datasets covering physician visits, emergency department visits, and inpatient hospitalizations, to determine health service utilization from April 1, 2006 through March 31, 2012. Incidence rate ratios (IRRs) were used to compare those with false positive results for MCADD to those with negative newborn screening results, stratified by age at service use. Results We identified 43 infants with a false positive newborn screening result for MCADD during the study period. These infants experienced significantly higher rates of physician visits (IRR: 1.42) and hospitalizations (IRR: 2.32) in the first year of life relative to a screen negative cohort in adjusted analyses. Differences in health services use were not observed after the first year of life. Conclusions The higher use of some health services among false positive infants during the first year of life may be explained by a psychosocial impact of false positive results on parental perceptions of infant health, and/or by differences in underlying health status. Understanding the impact of false positive newborn screening results can help to inform newborn screening programs in designing support and education for families. This is particularly important as additional disorders are added to expanded screening panels, yielding important clinical benefits for affected children but also a higher frequency of false positive findings.
dc.identifier.citationOrphanet Journal of Rare Diseases. 2016 Feb 03;11(1):12
dc.identifier.urihttp://dx.doi.org/10.1186/s13023-016-0391-5
dc.identifier.urihttp://hdl.handle.net/1993/31127
dc.language.rfc3066en
dc.rightsopen accessen_US
dc.rights.holderKaraceper et al.
dc.titleThe health system impact of false positive newborn screening results for medium-chain acyl-CoA dehydrogenase deficiency: a cohort study
dc.typeJournal Article
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