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

Autor: Deshayne B. Fell, Scott D. Grosse, Kumanan Wilson, Beth K. Potter, Hilary Vallance, John J. Mitchell, Steven Hawken, Annette Feigenbaum, Robin Z. Hayeems, Cheryl Rockman-Greenberg, Chitra Prasad, Doug Coyle, Astrid Guttmann, Brenda Wilson, Keiko Ueda, Anne-Marie Laberge, Jonathan B. Kronick, Sylvia Stockler, Christine Davies, Sara D. Khangura, Meranda Nakhla, Rebecca Sparkes, Maria D. Karaceper, Aizeddin A. Mhanni, Murray A. Potter, Pranesh Chakraborty, Julian Little, Marni Brownell, Linda Dodds
Přispěvatelé: University of Manitoba
Rok vydání: 2018
Předmět:
0301 basic medicine
Male
Newborn screening
Pediatrics
lcsh:Medicine
medium-chain acyl-CoA dehydrogenase deficiency
Health service utilization
030105 genetics & heredity
Acyl-CoA Dehydrogenase
Inherited metabolic diseases
Cohort Studies
0302 clinical medicine
Residence Characteristics
Medicine
Birth Weight
Pharmacology (medical)
Genetics (clinical)
Ontario
education.field_of_study
Gestational age
General Medicine
3. Good health
Hospitalization
Child
Preschool
Cohort
Medium-chain acyl-CoA dehydrogenase deficiency
Female
Emergency Service
Hospital
Cohort study
medicine.medical_specialty
Birth weight
Population
Gestational Age
Health Services Misuse
Lipid Metabolism
Inborn Errors
inherited metabolic diseases
03 medical and health sciences
Neonatal Screening
Humans
education
business.industry
newborn screening
Research
lcsh:R
Infant
Newborn
Infant
Emergency department
Medium-Chain Acyl-CoA Dehydrogenase Deficiency
Socioeconomic Factors
business
health service utilization
030217 neurology & neurosurgery
Zdroj: Orphanet Journal of Rare Diseases
Orphanet Journal of Rare Diseases, Vol 14, Iss 1, Pp 1-10 (2019)
Paediatrics Publications
ISSN: 1750-1172
Popis: Copyright: The Authors in collaboration with the Canadian Inherited Metabolic Diseases Research Network (CIMDRN). 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. This study was funded through a Canadian Institutes of Health Research (CIHR) Emerging Team Grant (TR3–119195). Maria Karaceper received a graduate scholarship through a charitable donation to the Children’s Hospital of Eastern Ontario. Dr. Mitchell receives research support from the Dr. Eleanor Mackenzie Harpur Pediatric Endowment Fund.
Databáze: OpenAIRE
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