儿童多发性硬化症的发病率和患病率：加拿大安大略省的一项基于人群的研究。
Incidence and prevalence of MS in children: A population-based study in Ontario, Canada.
机构信息
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.
出版信息
Neurology. 2018 Oct 23;91(17):e1579-e1590. doi: 10.1212/WNL.0000000000006395. Epub 2018 Sep 26.
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.
目的
使用管理（健康索赔）数据验证儿童多发性硬化症（MS）的病例定义，并估计加拿大安大略省儿科人群中 MS 的发病率和患病率。
方法
我们使用基于人群的管理数据来识别年龄≤18 岁的 MS 患者。我们使用包括 MS 患儿、单相脱髓鞘综合征患儿和健康儿童在内的临床参考队列评估了多个管理病例定义的性能；我们报告了敏感性、特异性、阳性预测值（PPV）和阴性预测值（NPV）。我们应用了 2 种首选病例定义来估计 2003 年至 2014 年 MS 的发病率和患病率。
结果
加拿大慢性疾病监测系统定义的≥1 次住院或 2 年内≥5 次医生对 MS 的就诊，其敏感性为 81.1%，特异性为 100%，PPV 为 100%，NPV 为 86%。Marrie 定义的≥3 次住院或 MS 就诊，其敏感性为 89.2%，特异性为 100%，PPV 为 100%，NPV 为 91.5%。根据所使用的管理病例定义，2014 年儿科人群中 MS 的年龄标准化年发病率范围为 0.99 至 1.24 每 100,000 人，年龄标准化患病率范围为 4.03 至 6.8 每 100,000 人。MS 的患病率随着时间的推移而上升。
结论
管理数据为估计儿科人群中 MS 的发病率和患病率提供了一种可行、有效的方法。安大略省儿科人群中 MS 的患病率在全球儿科人群中属于较高水平。
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