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

Autor: Karaceper MD; School of Epidemiology and Public Health, Faculty of Medicine, University of Ottawa, 600 Peter Morand Cr, Ottawa, ON, K1G 5Z3, Canada., Khangura SD; School of Epidemiology and Public Health, Faculty of Medicine, University of Ottawa, 600 Peter Morand Cr, Ottawa, ON, K1G 5Z3, Canada., Wilson K; School of Epidemiology and Public Health, Faculty of Medicine, University of Ottawa, 600 Peter Morand Cr, Ottawa, ON, K1G 5Z3, Canada.; Ottawa Hospital Research Institute, University of Ottawa, Ottawa, Canada.; Department of Medicine, Faculty of Medicine, University of Ottawa, Ottawa, Canada., Coyle D; School of Epidemiology and Public Health, Faculty of Medicine, University of Ottawa, 600 Peter Morand Cr, Ottawa, ON, K1G 5Z3, Canada., Brownell M; Manitoba Centre for Health Policy, Department of Community Health Sciences, Max Rady College of Medicine, Rady Faculty of Health Sciences, University of Manitoba, Winnipeg, Canada., Davies C; Newborn Screening Ontario, Children's Hospital of Eastern Ontario, Ottawa, Canada., Dodds L; Departments of Obstetrics & Gynecology and Pediatrics, Dalhousie University, Halifax, Canada., Feigenbaum A; Department of Pediatrics, Division of Clinical & Metabolic Genetics, The Hospital for Sick Children and University of Toronto, Toronto, Canada., Fell DB; School of Epidemiology and Public Health, Faculty of Medicine, University of Ottawa, 600 Peter Morand Cr, Ottawa, ON, K1G 5Z3, Canada.; Children's Hospital of Eastern Ontario Research Institute, Ottawa, Canada.; ICES, Toronto and Ottawa, Canada., Grosse SD; Centers for Disease Control and Prevention, National Center on Birth Defects and Developmental Disabilities, Atlanta, USA., Guttmann A; Child Health Evaluative Sciences, The Hospital for Sick Children, Toronto, Canada.; ICES, Toronto and Ottawa, Canada.; Department of Pediatrics, Division of Paediatric Medicine, The Hospital for Sick Children, University of Toronto, Toronto, Canada.; Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, Canada., Hawken S; School of Epidemiology and Public Health, Faculty of Medicine, University of Ottawa, 600 Peter Morand Cr, Ottawa, ON, K1G 5Z3, Canada.; Ottawa Hospital Research Institute, University of Ottawa, Ottawa, Canada.; ICES, Toronto and Ottawa, Canada., Hayeems RZ; Child Health Evaluative Sciences, The Hospital for Sick Children, Toronto, Canada.; Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, Canada., Kronick JB; Department of Pediatrics, Division of Clinical & Metabolic Genetics, The Hospital for Sick Children and University of Toronto, Toronto, Canada., Laberge AM; Medical Genetics, CHU Sainte-Justine and Department of Pediatrics, Université de Montréal, Montreal, Canada., Little J; School of Epidemiology and Public Health, Faculty of Medicine, University of Ottawa, 600 Peter Morand Cr, Ottawa, ON, K1G 5Z3, Canada., Mhanni A; Department of Paediatrics and Child Health, College of Medicine, Faculty of Health Sciences, University of Manitoba, Winnipeg, Canada., Mitchell JJ; Montreal Children's Hospital, McGill University, Montreal, Canada., Nakhla M; Montreal Children's Hospital, McGill University, Montreal, Canada., Potter M; Department of Pathology and Molecular Medicine, Faculty of Health Sciences, McMaster University, Hamilton, Canada.; Clinical Genetics Program, McMaster University Medical Centre, Hamilton Health Sciences, Hamilton, Canada., Prasad C; London Health Sciences Centre, Western University, London, Canada., Rockman-Greenberg C; Department of Paediatrics and Child Health, College of Medicine, Faculty of Health Sciences, University of Manitoba, Winnipeg, Canada., Sparkes R; Department of Paediatrics, Section of Clinical Genetics, Alberta Children's Hospital, Calgary, Canada., Stockler S; Children's & Women's Health Centre of British Columbia, Vancouver, Canada.; Biochemical Genetics Laboratory, Children's & Women's Health Centre of British Columbia, Vancouver, Canada., Ueda K; Children's & Women's Health Centre of British Columbia, Vancouver, Canada., Vallance H; Biochemical Genetics Laboratory, Children's & Women's Health Centre of British Columbia, Vancouver, Canada.; Department of Pathology, University of British Columbia, Vancouver, Canada., Wilson BJ; Division of Community Health and Humanities, Memorial University of Newfoundland, St. John's, Canada., Chakraborty P; Department of Pediatrics, Faculty of Medicine, University of Ottawa, Ottawa, Canada.; Newborn Screening Ontario, Children's Hospital of Eastern Ontario, Ottawa, Canada.; Children's Hospital of Eastern Ontario Research Institute, Ottawa, Canada., Potter BK; School of Epidemiology and Public Health, Faculty of Medicine, University of Ottawa, 600 Peter Morand Cr, Ottawa, ON, K1G 5Z3, Canada. bpotter@uottawa.ca.; Children's Hospital of Eastern Ontario Research Institute, Ottawa, Canada. bpotter@uottawa.ca.; ICES, Toronto and Ottawa, Canada. bpotter@uottawa.ca.
Jazyk: angličtina
Zdroj: Orphanet journal of rare diseases [Orphanet J Rare Dis] 2019 Mar 22; Vol. 14 (1), pp. 70. Date of Electronic Publication: 2019 Mar 22.
DOI: 10.1186/s13023-019-1001-0
Abstrakt: 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.
Databáze: MEDLINE
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