A capture-recapture model to estimate prevalence of children born in Scotland with developmental eye defects.
A capture-recapture model to estimate prevalence of children born in Scotland with developmental eye defects.
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DOI：
10.1080/14766650252962649
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发表时间：
2002-01-01
期刊：
影响因子：
--
通讯作者：
Jones, I
中科院分区：
文献类型：
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作者：
Campbell, H;Holmes, E;Jones, I
背景技术背景：小眼症、无眼症和缺损（MAC）是密切相关的结构发育性眼部缺陷，在英国媒体报道明显的病例集群后引起了公众的关注。进行了广泛的搜索，以确定潜在的情况下，诊断证实眼科审查。尽管作出了这一努力，但人们认识到，有些案件仍将无法查明。捕获-再捕获方法可以提高研究的出生患病率estimate.METHODS的准确性：所有出生的儿童与MAC在苏格兰（1981年至1996年）的发育性眼缺陷的登记。这从8个国家查明来源查明了188起案件。Hook和Regal描述的方法被用来确定每个来源的可能估计值。捕获-再捕获的对数线性方法被用来拟合一个模型，以确定由8个国家的来源的不同组合的儿童的数量，并在该模型中包括作为主effects.RESULTS：最大似然估计值计算表捕获/未捕获的一个源与任何其他来源。对于最完整的来源，苏格兰先天性异常登记处和其他七个合并的未观察到的病例估计数为78例（95% CI 53-115）。所有来源未观察到的病例数的两种不同对数线性模型计算的估计值分别为61（95% CI 43-86）和175（95% CI 106-287）。在一个地区进行外部检查，对一个独立的先天性异常登记册进行检查，已知该登记册具有很高的完整性，估计为120。该研究发现，根据研究期间的总活产率，苏格兰MAC的出生患病率为1.9/10000。捕获-再捕获分析表明，真实的出生患病率介于2.4/10000和3.5/10000之间。我们的经验表明，即使有多个确定来源和相当多的资源用于病例识别，仍会导致相当程度的先天性眼部异常的确定不足，并建议使用捕获-再捕获分析将是对先天性异常登记册的有益补充，用于需求评估、疾病负担估计和卫生服务规划。
BACKGROUND: Microphthalmia, anophthalmia and coloboma (MAC) are closely related structural developmental eye defects that have caused public concern in the UK following media reports of apparent clusters of cases. An extensive search was carried out to identify potential cases, and diagnoses were confirmed by ophthalmological review. Despite this effort, it was recognised that some cases would remain unidentified. Capture-recapture methods could improve the accuracy of the study's birth prevalence estimate.METHODS: A register of all children born with the developmental eye defects of MAC in Scotland (1981-96) was compiled. This identified 188 cases from eight national ascertainment sources. The methods described by Hook and Regal were used to determine possible estimates for each source. A log-linear method of capture-recapture was used to fit a model to the number of children identified by the different combinations of the eight national sources, and the individual sources were included in the model as main effects.RESULTS: Maximum-likelihood estimates were calculated for tables of captured/not captured by one source versus any other source. The estimated number of unobserved cases for the most complete source, the register of congenital anomalies for Scotland, and the seven others combined is 78 (95% CI 53-115). Estimates calculated from two different log-linear models of the numbers of cases unobserved by all sources were 61 (95% CI 43-86) and 175 (95% CI 106-287). An external check with an independent congenital-anomalies register in one region, which is known to have high completeness, gave an estimate of 120. The study found a birth prevalence of MAC in Scotland of 1.9/10000, based on total live births during the study period. The capture-recapture analysis suggests that the true birth prevalence lies between 2.4/10 000 and 3.5/10000.DISCUSSION: Our experience shows that, even with multiple sources of ascertainment and considerable resources directed at case identification, a considerable level of under-ascertainment of congenital eye-anomalies still results, and suggests that the use of capture-recapture analysis would be a useful addition to congenital anomalies registers for needs assessment, disease-burden estimates and health-service planning.