BackgroundSpatial modelling of STH and schistosomiasis epidemiology is now commonplace. Spatial epidemiological studies help inform decisions regarding the number of people at risk as well as the geographic areas that need to be targeted with mass drug administration; however, limited attention has been given to propagated uncertainties, their interpretation, and consequences for the mapped values. Using currently published literature on the spatial epidemiology of helminth infections we identified: (1) the main uncertainty sources, their definition and quantification and (2) how uncertainty is informative for STH programme managers and scientists working in this domain.Methodology/Principal FindingsWe performed a systematic literature search using the Preferred Reporting Items for Systematic reviews and Meta-Analysis (PRISMA) protocol. We searched Web of Knowledge and PubMed using a combination of uncertainty, geographic and disease terms. A total of 73 papers fulfilled the inclusion criteria for the systematic review. Only 9% of the studies did not address any element of uncertainty, while 91% of studies quantified uncertainty in the predicted morbidity indicators and 23% of studies mapped it. In addition, 57% of the studies quantified uncertainty in the regression coefficients but only 7% incorporated it in the regression response variable (morbidity indicator). Fifty percent of the studies discussed uncertainty in the covariates but did not quantify it. Uncertainty was mostly defined as precision, and quantified using credible intervals by means of Bayesian approaches.Conclusion/SignificanceNone of the studies considered adequately all sources of uncertainties. We highlighted the need for uncertainty in the morbidity indicator and predictor variable to be incorporated into the modelling framework. Study design and spatial support require further attention and uncertainty associated with Earth observation data should be quantified. Finally, more attention should be given to mapping and interpreting uncertainty, since they are relevant to inform decisions regarding the number of people at risk as well as the geographic areas that need to be targeted with mass drug administration.

Uncertainties in spatial modeling studies of schistosomiasis (SCH) are relevant for the reliable identification of at-risk populations. Ecological fallacy occurs when ecological or group-level analyses, such as spatial aggregations at a specific administrative level, are carried out for an individual-level inference. This could lead to the unreliable identification of at-risk populations, and consequently to fallacies in the drugs’ allocation strategies and their cost-effectiveness. A specific form of ecological fallacy is pure specification bias. The present research aims to quantify its effect on the parameter estimates of various environmental covariates used as drivers for SCH infection. This is done by (i) using a spatial convolution model that removes pure specification bias, (ii) estimating group and individual-level covariate regression parameters, and (iii) quantifying the difference between the parameter estimates and the predicted disease outcomes from the convolution and ecological models. We modeled the prevalence of Schistosoma japonicum using group-level health outcome data, and city-level environmental data as a proxy for individual-level exposure. We included environmental data such as water and vegetation indexes, distance to water bodies, day and night land surface temperature, and elevation. We estimated and compared the convolution and ecological model parameter estimates using Bayesian statistics. Covariate parameter estimates from the convolution and ecological models differed between 0.03 for the nearest distance to water bodies (NDWB), and 0.28 for the normalized difference water index (NDWI). The convolution model presented lower uncertainties in most of the parameter estimates, except for NDWB. High differences in uncertainty were found in night land surface temperature (0.23) and elevation (0.13). No significant differences were found between the predicted values and their uncertainties from both models. The proposed convolution model is able to correct for a pure specification bias by presenting less uncertain parameter estimates. It shows a good predictive performance for the mean prevalence values and for a positive number of infected people. Further research is needed to better understand the spatial extent and support of analysis to reliably explore the role of environmental variables.

BackgroundSpatial modelling studies of schistosomiasis (SCH) are now commonplace. Covariate values are commonly extracted at survey locations, where infection does not always take place, resulting in an unknown positional exposure mismatch. The present research aims to: (i) describe the nature of the positional exposure mismatch in modelling SCH helminth infections; (ii) delineate exposure areas to correct for such positional mismatch; and (iii) validate exposure areas using human positive cases.MethodsTo delineate exposure areas to Schistosoma japonicum, a spatial Bayesian network (sBN) was constructed. It uses data on exposure risk factors such as: potential sites for snails’ accessibility, geographical distribution of snail infection rate, and cost of the community to access nearby water bodies. Prior and conditional probabilities were obtained from the literature and inserted as weights based on their relative contribution to exposure; these probabilities were then used to calculate joint probabilities of exposure within the sBN.ResultsHigh values of probability of S. japonicum exposure correspond to polygons where snails could potentially be present, for instance in wet soils and areas with low slopes, but also where people can easily access water bodies. Low correlation (R2 = 0.3) was found between the percentage of human cases and the delineated probabilities of exposure when validation buffers are generated over the human cases.ConclusionsThe utility of a probabilistic method for the identification of exposure areas for S. japonicum, with wider application for other water-borne infections, was demonstrated. From a public health perspective, the schistosomiasis exposure sBN developed in this study could be used to guide local schistosomiasis control teams to specific potential areas of exposure, and improve efficiency of mass drug administration campaigns in places where people are likely to be exposed to the infection.Electronic supplementary materialThe online version of this article (10.1186/s13071-018-3039-6) contains supplementary material, which is available to authorized users.

Background: The modifiable areal unit problem (MAUP) arises when the support size of a spatial variable affects the relationship between prevalence and environmental risk factors. Its effect on schistosomiasis modelling studies could lead to unreliable parameter estimates. The present research aims to quantify MAUP effects on environmental drivers of Schistosoma japonicum infection by (i) bringing all covariates to the same spatial support, (ii) estimating individuallevel regression parameters at 30 m, 90 m, 250 m, 500 m and 1 km spatial supports, and (iii) quantifying the differences between parameter estimates using five models. Methods: We modelled the prevalence of Schistosoma japonicum using sub-provinces health outcome data and pixel-level environmental data. We estimated and compared regression coefficients from convolution models using Bayesian statistics. Results: Increasing the spatial support to 500 m gradually increased the parameter estimates and their associated uncertainties. Abrupt changes in the parameter estimates occur at 1 km spatial support, resulting in loss of significance of almost all the covariates. No significant differences were found between the predicted values and their uncertainties from the five models. We provide suggestions to define an appropriate spatial data structure for modelling that gives more reliable parameter estimates and a clear relationship between risk factors and the disease. Conclusions: Inclusion of quantified MAUP effects was important in this study on schistosomiasis. This will support helminth control programmes by providing reliable parameter estimates at the same spatial support and suggesting the use of an adequate spatial data structure, to generate reliable maps that could guide efficient mass drug administration campaigns.

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