The impact of imperfect screening tools on measuring the prevalence of epilepsy and headaches in Burkina Faso

Background: Epilepsy and progressively worsening severe chronic headaches (WSCH) are the two most common clinical manifestations of neurocysticercosis, a form of cysticercosis. Most community-based studies in sub-Saharan Africa (SSA) use a two-step approach (questionnaire and confirmation) to estimate the prevalence of these neurological disorders and neurocysticercosis. Few validate the questionnaire in the field or account for the imperfect nature of the screening questionnaire and the fact that only those who screen positive have the opportunity to be confirmed. This study aims to obtain community-based validity estimates of a screening questionnaire, and to assess the impact of verification bias and misclassification error on prevalence estimates of epilepsy and WSCH. Methodology/Principal findings: Baseline screening questionnaire followed by neurological examination data from a cluster randomized controlled trial collected between February 2011 and January 2012 were used. Bayesian latent-class models were applied to obtain verification bias adjusted validity estimates for the screening questionnaire. These models were also used to compare the adjusted prevalence estimates of epilepsy and WSCH to those directly obtained from the data (i.e. unadjusted prevalence estimates). Different priors were used and their corresponding posterior inference was compared for both WSCH and epilepsy. Screening data were available for 4768 individuals. For epilepsy, posterior estimates for the sensitivity varied with the priors used but remained robust for the specificity, with the highest estimates at 66.1% (95%BCI: 56.4%;75.3%) for sensitivity and 88.9% (88.0%;89.8%) for specificity. For WSCH, the sensitivity and specificity estimates remained robust, with the highest at 59.6% (49.7%;69.1%) and 88.6% (87.6%;89.6%), respectively. The unadjusted prevalence estimates were consistently lower than the adjusted prevalence estimates for both epilepsy and WSCH. Conclusions/Significance: This study demonstrates that in some settings, the prevalence of epilepsy and WSCH can be considerably underestimated when using the two-step approach. We provide an analytic solution to obtain more valid prevalence estimates of these neurological disorders, although more community-based validity studies are needed to reduce the uncertainty of the estimates. Valid estimates of these two neurological disorders are essential to obtain accurate burden values for neglected tropical diseases such as neurocysticercosis that manifest as epilepsy or WSCH. Trial registration: ClinicalTrials.gov NCT03095339. Author summary: Epilepsy and progressively worsening severe chronic headaches are the two most common clinical manifestations of neurocysticercosis, a form of cysticercosis. To understand where the prevalence of these neurological disorders is highest for targeted infection control, valid prevalence estimates are needed. Most neuroepidemiological studies conducted in low-resource settings use a two-step approach to identify cases (screening questionnaire followed by physician examination among screened positives) to obtain prevalence and burden estimates. We found that this most commonly-used two-step approach in community-based studies leads to an underestimation of the prevalence in our study. Our paper provides an analytic solution to reduce errors in estimating the prevalence of neurological disorders in community-based studies when using the two-step approach. Our proposed approach provides more valid estimates of the prevalence of these neurological disorders and could be used to better reflect the consequences that neglected tropical diseases manifesting as epilepsy or headaches have on people’s health and disabilities.