Ruth Ann Marrie1, Julia O'Mahony2, Colleen Maxwell2, Vicki Ling2, E Ann Yeh2, Douglas L Arnold2, Amit Bar-Or2, Brenda Banwell2. 1. From the Departments of Internal Medicine and Community Health Sciences (R.A.M.), Max Rady College of Medicine, Rady Faculty of Health Sciences, University of Manitoba, Winnipeg; Institute of Health Policy, Management and Evaluation (J.O.), University of Toronto, the Hospital for Sick Children; Schools of Pharmacy and Public Health and Health Systems (C.M.), University of Waterloo; Institute for Clinical Evaluative Sciences (C.M., V.L.), Toronto; Department of Pediatrics (E.A.Y.), University of Toronto; Division of Neurology, Neurosciences and Mental Health (E.A.Y.), the Hospital for Sick Children, SickKids Research Institute, Toronto; Montreal Neurological Institute (D.L.A.), McGill University, Montreal, Canada; Center for Neuroinflammation and Experimental Therapeutics and Department of Neurology (A.B.-O.), Perelman School of Medicine (B.B.), University of Pennsylvania, Philadelphia; and Division of Child Neurology (B.B.), the Children's Hospital of Philadelphia, PA. rmarrie@hsc.mb.ca. 2. From the Departments of Internal Medicine and Community Health Sciences (R.A.M.), Max Rady College of Medicine, Rady Faculty of Health Sciences, University of Manitoba, Winnipeg; Institute of Health Policy, Management and Evaluation (J.O.), University of Toronto, the Hospital for Sick Children; Schools of Pharmacy and Public Health and Health Systems (C.M.), University of Waterloo; Institute for Clinical Evaluative Sciences (C.M., V.L.), Toronto; Department of Pediatrics (E.A.Y.), University of Toronto; Division of Neurology, Neurosciences and Mental Health (E.A.Y.), the Hospital for Sick Children, SickKids Research Institute, Toronto; Montreal Neurological Institute (D.L.A.), McGill University, Montreal, Canada; Center for Neuroinflammation and Experimental Therapeutics and Department of Neurology (A.B.-O.), Perelman School of Medicine (B.B.), University of Pennsylvania, Philadelphia; and Division of Child Neurology (B.B.), the Children's Hospital of Philadelphia, PA.
Abstract
OBJECTIVE: To validate a case definition of multiple sclerosis (MS) in the pediatric population using administrative (health claims) data, and to estimate the incidence and prevalence of MS in the pediatric population for Ontario, Canada. METHODS: We used population-based administrative data to identify persons aged ≤18 years with MS. We assessed the performance of multiple administrative case definitions using a clinical reference cohort including children with MS, children with monophasic demyelinating syndromes, and healthy children; we report sensitivity, specificity, positive predictive value (PPV), and negative predictive value (NPV). We applied 2 preferred case definitions to estimate the incidence and prevalence of MS from 2003 to 2014. RESULTS: The Canadian Chronic Disease Surveillance System definition of ≥1 hospitalization or ≥5 physician claims for MS within 2 years had a sensitivity of 81.1%, specificity of 100%, PPV of 100%, and NPV of 86%. The Marrie definition of ≥3 hospital or physician claims for MS ever had a sensitivity of 89.2%, specificity of 100%, PPV of 100%, and NPV of 91.5%. Depending on the administrative case definition used, in 2014, the annual age-standardized annual incidence of MS in the pediatric population ranged from 0.99 to 1.24 per 100,000 population, and the age-standardized prevalence ranged from 4.03 to 6.8 per 100,000 population. The prevalence of MS rose over time. CONCLUSION: Administrative data provide a feasible, valid means of estimating the incidence and prevalence of MS in the pediatric population. MS prevalence in the Ontario pediatric population is among the highest reported in pediatric populations worldwide.
OBJECTIVE: To validate a case definition of multiple sclerosis (MS) in the pediatric population using administrative (health claims) data, and to estimate the incidence and prevalence of MS in the pediatric population for Ontario, Canada. METHODS: We used population-based administrative data to identify persons aged ≤18 years with MS. We assessed the performance of multiple administrative case definitions using a clinical reference cohort including children with MS, children with monophasic demyelinating syndromes, and healthy children; we report sensitivity, specificity, positive predictive value (PPV), and negative predictive value (NPV). We applied 2 preferred case definitions to estimate the incidence and prevalence of MS from 2003 to 2014. RESULTS: The Canadian Chronic Disease Surveillance System definition of ≥1 hospitalization or ≥5 physician claims for MS within 2 years had a sensitivity of 81.1%, specificity of 100%, PPV of 100%, and NPV of 86%. The Marrie definition of ≥3 hospital or physician claims for MS ever had a sensitivity of 89.2%, specificity of 100%, PPV of 100%, and NPV of 91.5%. Depending on the administrative case definition used, in 2014, the annual age-standardized annual incidence of MS in the pediatric population ranged from 0.99 to 1.24 per 100,000 population, and the age-standardized prevalence ranged from 4.03 to 6.8 per 100,000 population. The prevalence of MS rose over time. CONCLUSION: Administrative data provide a feasible, valid means of estimating the incidence and prevalence of MS in the pediatric population. MS prevalence in the Ontario pediatric population is among the highest reported in pediatric populations worldwide.
Authors: Ruth Ann Marrie; Julia O'Mahony; Colleen J Maxwell; Vicki Ling; E Ann Yeh; Douglas L Arnold; Amit Bar-Or; Brenda Banwell Journal: PLoS One Date: 2019-06-11 Impact factor: 3.240
Authors: Aphra Luchesa Smith; Christina Benetou; Hayley Bullock; Adam Kuczynski; Sarah Rudebeck; Katie Hanson; Sarah Crichton; Kshitij Mankad; Ata Siddiqui; Susan Byrne; Ming Lim; Cheryl Hemingway Journal: Children (Basel) Date: 2020-11-09
Authors: Marco Kaufmann; Milo Alan Puhan; Anke Salmen; Christian P Kamm; Zina-Mary Manjaly; Pasquale Calabrese; Sven Schippling; Stefanie Müller; Jens Kuhle; Caroline Pot; Claudio Gobbi; Nina Steinemann; Viktor von Wyl Journal: Front Neurol Date: 2020-03-06 Impact factor: 4.003