Spatial analysis of hypospadias cases in northern France: taking clinical data into account

Arthur Lauriot Dit Prevost,Caroline Lanier,Florent Occelli,Dyuti Sharma,Michael Genin,René-Hilaire Priso,Remi Besson

doi:10.1186/s12887-020-02332-1

Arthur Lauriot Dit Prevost, Caroline Lanier + Show 5 more

Open Access

https://doi.org/10.1186/s12887-020-02332-1

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Abstract

BackgroundStrong evidence for a causal role of environmental factors in a congenital anomaly is still difficult to produce. The collection of statistical data is crucial for gaining a better understanding of the epidemiology and pathophysiology of these anomalies. We aimed to evaluate spatial variations in hypospadias within our region and it’s association to socioeconomic and ecological factors, taking clinical data into account.MethodsAll boys with hypospadias born in northern France and seen in Lille University Medical Center (Lille, France) between 1999 and 2012 were included in the analysis. We retrospectively collected geographic data, clinical data (especially known confounding factors associated with an elevated risk of hypospadias), and demographic, socio-economic and ecological data. We analyzed the entire study population and subsequently the subset of boys lacking confounding factors.ResultsThe study sample of 975 cases of hypospadias over the 13-year period resulted in an incidence of 25.4/10,000 male births, and was characterized by significant spatial heterogeneity (p < 0.005) and autocorrelation (p < 0.001). We detected two high-incidence clusters that differed with regard to their land use. After the exclusion of 221 patients with confounding factors, two high-incidence clusters with significant disease risks (1.65 and 1.75, respectively; p < 0.001) and a significant difference in land use (p < 0.001) again appeared. The first cluster contained a higher median [interquartile range] proportion of artificialized land (0.40 [0.22;0.47]) than the remaining “neutral areas” (0.19 [0.08;0.53]) did (p < 0.001). Conversely, the second cluster contained a higher median proportion of rural land (0.90 [0.78;0.96]) than the “neutral areas” (0.81 [0.47;0.92]) did (p < 0.001). The median deprivation index was significantly lower in the urban cluster (0.47 [0.42;0.55]) and significantly higher in the rural cluster (0.69 [0.56;0.73]) (p < 0.001).ConclusionsOur results evidenced the heterogeneous spatial distribution of cases of hypospadias in northern France. We identified two clusters with different environmental and social patterns – even after the exclusion of known confounding factors.

Highlights

Strong evidence for a causal role of environmental factors in a congenital anomaly is still difficult to produce
The suspected causal factors include genetic factors, [4,5,6,7] iatrogenic factors (e.g. medications taken during pregnancy [8,9,10] or conception via assisted reproductive technologies (ART) [11, 12]) and environmental factors [13,14,15,16,17]
The collection of statistical data on all the potential genetic and environmental causal factors is crucial for gaining a better understanding of the epidemiology and pathophysiology of hypospadias and, for guiding public health measures capable of mitigating exogenous risk factors to the greatest extent possible [5, 18]

Summary

Introduction

Strong evidence for a causal role of environmental factors in a congenital anomaly is still difficult to produce. The collection of statistical data is crucial for gaining a better understanding of the epidemiology and pathophysiology of these anomalies. We aimed to evaluate spatial variations in hypospadias within our region and it’s association to socioeconomic and ecological factors, taking clinical data into account. The collection of statistical data on all the potential genetic and environmental causal factors is crucial for gaining a better understanding of the epidemiology and pathophysiology of hypospadias and, for guiding public health measures capable of mitigating exogenous risk factors to the greatest extent possible [5, 18]. Given that many environmental factors are ubiquitous and not measurable, strong evidence for a causal role in a congenital anomaly is still difficult to produce. Most of the studies involving spatial analysis used massive administrative datasets from national registries; the latter did not contain much clinical information

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