Abstract

Various global health initiatives are currently advocating the elimination of schistosomiasis within the next decade. Schistosomiasis is a highly debilitating tropical infectious disease with severe burden of morbidity and thus operational research accurately evaluating diagnostics that quantify the epidemic status for guiding effective strategies is essential. Latent class models (LCMs) have been generally considered in epidemiology and in particular in recent schistosomiasis diagnostic studies as a flexible tool for evaluating diagnostics because assessing the true infection status (via a gold standard) is not possible. However, within the biostatistics literature, classical LCM have already been criticised for real-life problems under violation of the conditional independence (CI) assumption and when applied to a small number of diagnostics (i.e. most often 3-5 diagnostic tests). Solutions of relaxing the CI assumption and accounting for zero-inflation, as well as collecting partial gold standard information, have been proposed, offering the potential for more robust model estimates. In the current article, we examined such approaches in the context of schistosomiasis via analysis of two real datasets and extensive simulation studies. Our main conclusions highlighted poor model fit in low prevalence settings and the necessity of collecting partial gold standard information in such settings in order to improve the accuracy and reduce bias of sensitivity and specificity estimates.

Highlights

  • Schistosomiasis affects over 250 million people in rural and some urban populations across much of Africa and regions in South America, the Caribbean, People’s Republic of China, and Southeast Asia [1, 2]

  • In schistosomiasis diagnostic studies, several inherent study design issues pose a real challenge for the currently available statistical tools used for diagnostic modelling and associated data analysis and conclusions

  • For Schistosoma haematobium work has been done in Western Nigeria [8], comparing the presence of parasite eggs detected on the filter paper from the filtered urine, haematuria detected by dipstick, and detection of the Dra1 DNA extracted from the filters

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Summary

Introduction

Schistosomiasis affects over 250 million people in rural and some urban populations across much of Africa and regions in South America, the Caribbean, People’s Republic of China, and Southeast Asia [1, 2]. Lodh et al [7] carried out a study in Zambia to compare KK, CCA and DNA (from urine) in an exposed population in Zambia. A total of 89 volunteers were examined, KK showed 45/89 infected, CCA 55/89 (6 false positives) and 79/89 had detectable Schistosoma mansoni DNA. If there is a schistosomiasis gold standard, it is the presence of parasite specific DNA. For Schistosoma haematobium work has been done in Western Nigeria [8], comparing the presence of parasite eggs detected on the filter paper from the filtered urine, haematuria detected by dipstick, and detection of the Dra DNA extracted from the filters. Some 400 specimens were examined in that study and the conclusion was that detection of Dra fragment is a definitive test for the presence of S. haematobium infection

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