同时测试用于多种罕见疾病的被动和主动病例发现系统的可行性：三个美国州的经验。
Testing the Feasibility of a Passive and Active Case Ascertainment System for Multiple Rare Conditions Simultaneously: The Experience in Three US States.
机构信息
Institute on DisabilityUniversity of New HampshireDurham, NHUnited States.
Department of Epidemiology and BiostatisticsUniversity of South CarolinaColumbia, SCUnited States.
出版信息
JMIR Public Health Surveill. 2016 Aug 29;2(2):e151. doi: 10.2196/publichealth.5516.
BACKGROUND
Owing to their low prevalence, single rare conditions are difficult to monitor through current state passive and active case ascertainment systems. However, such monitoring is important because, as a group, rare conditions have great impact on the health of affected individuals and the well-being of their caregivers. A viable approach could be to conduct passive and active case ascertainment of several rare conditions simultaneously. This is a report about the feasibility of such an approach.
OBJECTIVE
To test the feasibility of a case ascertainment system with passive and active components aimed at monitoring 3 rare conditions simultaneously in 3 states of the United States (Colorado, Kansas, and South Carolina). The 3 conditions are spina bifida, muscular dystrophy, and fragile X syndrome.
METHODS
Teams from each state evaluated the possibility of using current or modified versions of their local passive and active case ascertainment systems and datasets to monitor the 3 conditions. Together, these teams established the case definitions and selected the variables and the abstraction tools for the active case ascertainment approach. After testing the ability of their local passive and active case ascertainment system to capture all 3 conditions, the next steps were to report the number of cases detected actively and passively for each condition, to list the local barriers against the combined passive and active case ascertainment system, and to describe the experiences in trying to overcome these barriers.
RESULTS
During the test period, the team from South Carolina was able to collect data on all 3 conditions simultaneously for all ages. The Colorado team was also able to collect data on all 3 conditions but, because of age restrictions in its passive and active case ascertainment system, it was able to report few cases of fragile X syndrome. The team from Kansas was able to collect data only on spina bifida. For all states, the implementation of an active component of the ascertainment system was problematic. The passive component appears viable with minor modifications.
CONCLUSIONS
Despite evident barriers, the joint passive and active case ascertainment of rare disorders using modified existing surveillance systems and datasets seems feasible, especially for systems that rely on passive case ascertainment.
背景
由于罕见疾病的患病率较低，通过当前的被动和主动病例确定系统难以监测单一的罕见疾病。然而，这种监测很重要，因为作为一个群体，罕见疾病对受影响个体的健康和护理人员的福祉有很大影响。一种可行的方法是同时对几种罕见疾病进行被动和主动病例确定。这是一份关于这种方法可行性的报告。
目的
测试一种具有被动和主动组件的病例确定系统的可行性，该系统旨在同时监测美国三个州（科罗拉多州、堪萨斯州和南卡罗来纳州）的三种罕见疾病：脊髓裂、肌肉营养不良症和脆性 X 综合征。
方法
每个州的团队评估了使用其当地的被动和主动病例确定系统及其数据集的当前或修改版本来监测三种疾病的可能性。这些团队共同确定了病例定义，并选择了主动病例确定方法的变量和抽象工具。在测试了其当地的被动和主动病例确定系统捕捉所有三种疾病的能力之后，下一步是报告每种疾病主动和被动发现的病例数量，列出阻碍联合被动和主动病例确定系统的当地障碍，并描述克服这些障碍的经验。
结果
在测试期间，南卡罗来纳州的团队能够同时为所有年龄段收集所有三种疾病的病例数据。科罗拉多州的团队也能够收集所有三种疾病的数据，但由于其被动和主动病例确定系统的年龄限制，它只能报告少数脆性 X 综合征病例。堪萨斯州的团队只能收集脊髓裂的数据。对于所有州，实施病例确定系统的主动组件都存在问题。经过轻微修改，被动组件似乎是可行的。
结论
尽管存在明显的障碍，但使用修改后的现有监测系统和数据集联合进行罕见疾病的被动和主动病例确定似乎是可行的，特别是对于依赖被动病例确定的系统。
Zotero 插件