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Rob van Hest Capture-recapture Methods in Surveillance - RePub ...

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Underreport<strong>in</strong>g of Legionnaires’ disease <strong>in</strong> the Netherlands<br />

notification but the notified and ascerta<strong>in</strong>ed <strong>in</strong>cidence rates <strong>in</strong> region South were higher<br />

than <strong>in</strong> the rest of the Netherlands (P < 0.0001).<br />

<strong>Capture</strong>-<strong>recapture</strong> analysis<br />

Internal validity analysis by two-source capture-<strong>recapture</strong> analysis on Notification and<br />

Hospital and on Laboratory and Hospital both estimate 865 Legionnaires’ disease patients<br />

through Chapman’s Nearly Unbiased Estimator. The considerable lower capture<strong>recapture</strong><br />

estimate obta<strong>in</strong>ed with Notification and Laboratory (523 Legionnaires’ disease<br />

patients) <strong>in</strong>dicates a larger positive association between this pair than between the other<br />

pairs, result<strong>in</strong>g <strong>in</strong> an estimate more biased downwards.<br />

The best-fitt<strong>in</strong>g three-source log-l<strong>in</strong>ear capture-<strong>recapture</strong> model was the<br />

saturated model, i.e. the model <strong>in</strong>clud<strong>in</strong>g all two-variable associations and assum<strong>in</strong>g<br />

absent three-way <strong>in</strong>teraction, which yielded an estimate of 1253 Legionnaires’ disease<br />

patients (95% confidence <strong>in</strong>terval (CI) 1019-1715). Estimated under-notification was<br />

70.2%. To acknowledge the geographical differences capture-<strong>recapture</strong> analysis stratified<br />

by region was performed. For all regions apart from region East a more parsimonious<br />

model, conta<strong>in</strong><strong>in</strong>g only one two-way <strong>in</strong>teraction (between Notification and Laboratory),<br />

was selected as best-fitt<strong>in</strong>g model, with totals of 78, 327 and 277 Legionnaires’ disease<br />

patients and <strong>in</strong>cidence rates of 2.33, 2.75 and 3.56 per 100 000 <strong>in</strong>habitants for region<br />

North, West and South respectively. For region East a saturated model was selected that<br />

estimated an unexpectedly high number of 650 Legionnaires’ disease patients with a wide<br />

95%CI of 283-2382 patients.<br />

As an alternative to the stratified capture-<strong>recapture</strong> analysis we specified a logl<strong>in</strong>ear<br />

covariate (region) capture-<strong>recapture</strong> model. The covariate model that served as a<br />

start<strong>in</strong>g po<strong>in</strong>t conta<strong>in</strong>ed, apart from the ma<strong>in</strong> effects for Region and the three registers,<br />

the Region-Notification, Region-Laboratory, Region-Hospital, Notification-Laboratory,<br />

Notification-Hospital, Laboratory-Hospital two-variable terms. In this model we allow for<br />

regional differences <strong>in</strong> the number of cases <strong>in</strong> the three registers, but not for <strong>in</strong>teraction<br />

with other effects per stratum, as the association between the registers is assumed equal<br />

across regions. This model fits the data reasonably well (G 2 = 22.1; df = 9; P = 0.009) and<br />

estimates 932 Legionnaires’ disease patients with a narrower CI of 851-1106, reduc<strong>in</strong>g<br />

statistical uncerta<strong>in</strong>ty. Inspection of the misfit for <strong>in</strong>dividual cells showed a large adjusted<br />

residual for Legionnaires’ disease patients only known to Laboratory <strong>in</strong> region East. After<br />

<strong>in</strong>clud<strong>in</strong>g a separate parameter for this s<strong>in</strong>gle cell we obta<strong>in</strong> a good fitt<strong>in</strong>g model (G 2 =<br />

5.7; df = 8; P = 0.686). The estimated number of Legionnaires’ disease patients was 886<br />

(95%CI 827-1022), similar to the two <strong>in</strong>ternal validity estimates with least assumed<br />

<strong>in</strong>terdependence.<br />

The estimated register-specific coverage rate of Notification, Laboratory and<br />

Hospital was 42.1% (373/886), 29.5% (261/886) and 74.9% (663/886) respectively. The<br />

estimated under-notification was 57.9%. The estimated average annual <strong>in</strong>cidence rate of<br />

Legionnaires’ disease was 2.77/100 000.<br />

71

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