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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.author Karaceper, Maria D
dc.contributor.author Chakraborty, Pranesh
dc.contributor.author Coyle, Doug
dc.contributor.author Wilson, Kumanan
dc.contributor.author Kronick, Jonathan B
dc.contributor.author Hawken, Steven
dc.contributor.author Davies, Christine
dc.contributor.author Brownell, Marni
dc.contributor.author Dodds, Linda
dc.contributor.author Feigenbaum, Annette
dc.contributor.author Fell, Deshayne B
dc.contributor.author Grosse, Scott D
dc.contributor.author Guttmann, Astrid
dc.contributor.author Laberge, Anne-Marie
dc.contributor.author Mhanni, Aizeddin
dc.contributor.author Miller, Fiona A
dc.contributor.author Mitchell, John J
dc.contributor.author Nakhla, Meranda
dc.contributor.author Prasad, Chitra
dc.contributor.author Rockman-Greenberg, Cheryl
dc.contributor.author Sparkes, Rebecca
dc.contributor.author Wilson, Brenda J
dc.contributor.author Potter, Beth K
dc.date.accessioned 2016-02-04T15:00:19Z
dc.date.available 2016-02-04T15:00:19Z
dc.date.issued 2016-02-03
dc.identifier.citation Orphanet Journal of Rare Diseases. 2016 Feb 03;11(1):12
dc.identifier.uri http://dx.doi.org/10.1186/s13023-016-0391-5
dc.identifier.uri http://hdl.handle.net/1993/31127
dc.description.abstract Abstract 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.title The health system impact of false positive newborn screening results for medium-chain acyl-CoA dehydrogenase deficiency: a cohort study
dc.type Journal Article
dc.language.rfc3066 en
dc.rights.holder Karaceper et al.
dc.date.updated 2016-02-03T17:04:20Z


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