Geographic distribution of live births with tetralogy of Fallot in North Carolina 2003 to 2012

Nelson, Jennifer S.; Stebbins, Rebecca C.; Strassle, Paula D.; Meyer, Robert E.

doi:10.1002/bdra.23566

Cited by 12 publications

(9 citation statements)

References 36 publications

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“…Even so, the data analysed do not fit any definite spatial country-wide pattern. The study conducted by Nelson et al in North Carolina (USA) showed a cluster of TOF cases in mainly agricultural areas but did not identify any specific environmental risk factors [26]. In their spatial analysis of CHD prevalence in the Valencian Region [27], the authors also found a heterogeneous geographical distribution which they felt might be due to differences in diagnostic accuracy and coding at hospitals.…”

Section: Discussionmentioning

confidence: 99%

“…In China, a progressive increase in the prevalence of atrial septal defect and patent ductus arteriosus was observed in areas of higher altitude [25]. A cluster of births with TOF was found in a largely agricultural area of North Carolina (USA) but there was insufficient evidence to conclude that this was due to any given environmental factor [26]. In Spain, a temporal and spatial analysis of CHD prevalence was carried out in the Valencian Region ( Comunidad Valenciana ) in the east of Spain, where the authors found a heterogeneous geographic pattern [27].…”

Section: Introductionmentioning

confidence: 99%

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Tetralogy of Fallot in Spain: a nationwide registry-based mortality study across 36 years

Llamosas-Falcón

Bermejo‐Sánchez

Sánchez-Díaz

et al. 2019

Orphanet J Rare Dis

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Background Tetralogy of Fallot (TOF) is the most frequent cyanotic congenital heart defect. TOF mortality has fallen remarkably in recent years due to therapeutic advances. Accordingly, the aim of this study was to assess temporal and spatial variability in TOF-related mortality in Spain across the period 1981–2016, using data drawn from the nationwide population-based registry. Methods Annual deaths due to TOF were sourced from the Spanish National Institute of Statistics database by reference to International Classification of Diseases (ICD), 9th and 10th Revision codes, namely, ICD-9 code 745.2 (period 1981–1998) and ICD-10 code Q21.3 (period 1999–2016). Age-specific and age-adjusted mortality rates were calculated, as were standardised mortality ratios (SMRs) by province, district and municipality for the period 1999–2016. Results A total of 1035 deaths were attributed to TOF (57.78% of them were men and 42.22% were women). The age-adjusted mortality rate ranged from 0.75 per 1,000,000 inhabitants (95% confidence interval [CI]: 0–1.36) in 1981 to 0.03 per 1,000,000 (95% CI: 0.01–0.06) in 2016 for both sexes. In 2011, there was a change in the mortality trend, with a significant decrease of 49.22% per year ( p < 0.001). In terms of geographical analysis, some areas with a significantly higher risk of TOF mortality were identified in the south of Spain, though no specific spatial pattern was in evidence. Conclusion The decrease in TOF mortality may be related to improvements in diagnostic and treatment techniques. More studies are needed to analyse regions with a higher mortality risk, in order to improve medical planning and resource allocation, and identify risk factors and preventive measures.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Tetralogy of Fallot in Spain: a nationwide registry-based mortality study across 36 years

Llamosas-Falcón

Bermejo‐Sánchez

Sánchez-Díaz

et al. 2019

Orphanet J Rare Dis

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“…Most of the literature studies in this field focused on congenital neural tube defects, [24] gastroschisis [25] or congenital heart defects. [26] To the best of our knowledge, only seven studies have analyzed the spatial distribution of cases of hypospadias. The statistical methodology varies, and the results remain inconsistent, with spatial cluster detection in three studies, ecological pattern association in three studies (with three different definitions of rural/urban areas), and association to socio-economic status in one article.…”

Section: Introductionmentioning

confidence: 99%

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

Prevost

Génin

OCCELLI

et al. 2020

Preprint

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Background 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 in our region, taking into account of clinical data, and evaluate association to socio-economic and ecological data. Methods All 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 whole study population and then the subset of boys lacking confounding factors. Results The study sample of 975 cases of hypospadias 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). Conclusions Our 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.

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“…Left surgically unrepaired, TOF has a 66% survival rate at 1 year; with surgery, greater than 95% of infants can expect to survive into adulthood (Bertranou et al, ; Barron, ). Although infant mortality in TOF has sharply declined since the advent of surgical palliation in 1945, a recent study demonstrated that 15% of children born with TOF in North Carolina (NC) between 2003 and 2012 died in infancy (Egbe et al, ; Nelson et al, ). Little is known about the causes of death and risk factors for death among infants with TOF.…”

Section: Introductionmentioning

confidence: 99%

Effect of Concomitant Birth Defects and Genetic Anomalies on Infant Mortality in Tetralogy of Fallot

Jernigan

Strassle

Stebbins

et al. 2017

Birth Defects Research

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Geographic distribution of live births with tetralogy of Fallot in North Carolina 2003 to 2012

Cited by 12 publications

References 36 publications

Tetralogy of Fallot in Spain: a nationwide registry-based mortality study across 36 years

Tetralogy of Fallot in Spain: a nationwide registry-based mortality study across 36 years

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

Effect of Concomitant Birth Defects and Genetic Anomalies on Infant Mortality in Tetralogy of Fallot

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