Health services use among children diagnosed with medium-chain acyl-CoA dehydrogenase deficiency through newborn screening: a cohort study in Ontario, Canada

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Date
2019-03-22Author
Karaceper, Maria D
Khangura, Sara D
Wilson, Kumanan
Coyle, Doug
Brownell, Marni
Davies, Christine
Dodds, Linda
Feigenbaum, Annette
Fell, Deshayne B
Grosse, Scott D
Guttmann, Astrid
Hawken, Steven
Hayeems, Robin Z
Kronick, Jonathan B
Laberge, Anne-Marie
Little, Julian
Mhanni, Aizeddin
Mitchell, John J
Nakhla, Meranda
Potter, Murray
Prasad, Chitra
Rockman-Greenberg, Cheryl
Sparkes, Rebecca
Stockler, Sylvia
Ueda, Keiko
Vallance, Hilary
Wilson, Brenda J
Chakraborty, Pranesh
Potter, Beth K
Metadata
Show full item recordAbstract
Abstract
Background
We describe early health services utilization for children diagnosed with medium-chain acyl-CoA dehydrogenase (MCAD) deficiency through newborn screening in Ontario, Canada, relative to a screen negative comparison cohort.
Methods
Eligible children were identified via newborn screening between April 1, 2006 and March 31, 2010. Age-stratified rates of physician encounters, emergency department (ED) visits and inpatient hospitalizations to March 31, 2012 were compared using incidence rate ratios (IRR) and incidence rate differences (IRD). We used negative binomial regression to adjust IRRs for sex, gestational age, birth weight, socioeconomic status and rural/urban residence.
Results
Throughout the first few years of life, children with MCAD deficiency (n = 40) experienced statistically significantly higher rates of physician encounters, ED visits, and hospital stays compared with the screen negative cohort. The highest rates of ED visits and hospitalizations in the MCAD deficiency cohort occurred from 6 months to 2 years of age (ED use: 2.1–2.5 visits per child per year; hospitalization: 0.5–0.6 visits per child per year), after which rates gradually declined.
Conclusions
This study confirms that young children with MCAD deficiency use health services more frequently than the general population throughout the first few years of life. Rates of service use in this population gradually diminish after 24 months of age.