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      Temporal and Spatial Analysis of Neural Tube Defects and Detection of Geographical Factors in Shanxi Province, China

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          Abstract

          Background

          Neural tube defects (NTDs) are congenital birth defects that occur in the central nervous system, and they have the highest incidence among all birth defects. Shanxi Province in China has the world’s highest rate of NTDs. Since the 1990s, China’s government has worked on many birth defect prevention programs to reduce the occurrence of NTDs, such as pregnancy planning, health education, genetic counseling, antenatal ultrasonography and serological screening. However, the rate of NTDs in Shanxi Province is still higher than the world’s average morbidity rate after intervention. In addition, Shanxi Province has abundant coal reserves, and is the largest coal production province in China. The objectives of this study are to determine the temporal and spatial variation of the NTD rate in rural areas of Shanxi Province, China, and identify geographical environmental factors that were associated with NTDs in the risk area.

          Methods

          In this study, Heshun County and Yuanping County in Shanxi Province, which have high incidence of NTDs, were selected as the study areas. Two paired sample T test was used to analyze the changes in the risk of NTDs from the time dimension. Ripley’s k function and spatial filtering were combined with geographic information system (GIS) software to study the changes in the risk of NTDs from the spatial dimension. In addition, geographical detectors were used to identify the risk geographical environmental factors of NTDs in the study areas, especially the areas close to the coal sites and main roads.

          Results

          In both Heshun County and Yuanping County, the incidence of NTDs was significantly (P<0.05) reduced after intervention. The results from spatial analysis showed that significant spatial heterogeneity existed in both counties. NTD clusters were still identified in areas close to coal sites and main roads after interventions. This study also revealed that the elevation, fault and soil types always had a larger influence on the incidence of NTDs in our study areas. In addition, distance to the river was a risk factor of NTDs in areas close to the coal sites and main roads.

          Conclusion

          The existing interventions may have played an important role to reduce the incidence of NTDs. However, there is still spatial heterogeneity in both counties after using the traditional intervention methods. The government needs to take more measures to strengthen the environmental restoration to prevent the occurrence of NTDs, especially those areas close to coal sites and main roads. The outcome of this research provides an important theoretical basis and technical support for the government to prevent the occurrence of NTDs.

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          Most cited references13

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          Epidemiology of neural tube defects.

          Neural tube defects (NTDs)-malformations secondary to abnormal neural tube closure between the third and fourth weeks of gestational age-have a complex and imperfectly understood etiology in which both genetic and environmental factors appear to be involved. A number of specific chromosomal or single-gene disorders, presumably not affected by environmental influences, are associated with the development of NTDs, but such syndromal cases account for a small proportion of NTDs in live-born infants. Analysis of recurrence patterns within families and of twin-concordance data provides evidence of a genetic influence in nonsyndromal cases, but factors such as socioeconomic status and geographic area (independent of race or ethnicity) are also associated with variations in the incidence of NTDs. The prevalence at birth of both anencephaly and spina bifida has decreased, but the advent of antenatal diagnosis and elective termination of affected pregnancies has undermined the reliability of birth prevalence rate as an estimate of incidence. Some occupational and other exposures, including maternal use of antiepileptic drugs (AEDs), are associated with increased risk for NTDs. Among women who have had an NTD-affected pregnancy, recurrence risk is markedly higher than the risk for a first NTD-affected pregnancy in the general population. There is strong evidence, overall, for a protective effect of adequate folate consumption. In some high-risk groups, however, such as women taking AEDs, folate supplementation has not been proven to reduce NTD risk.
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            Altitude as a risk factor for congenital anomalies.

            The birth prevalence of specific types of congenital anomalies at low and high altitudes in South America were compared after adjustment for prenatal growth, ethnicity, and socioeconomic status. The material includes all 1,668,722 consecutive births occurring in 53 hospitals participating in the Latin-American Collaborative Study of Congenital Malformations (ECLAMC), having registered at least 100 malformed/control pairs between 1967 and 1995. The lowland subsample (below 2,000 m above the sea level) included 46,729 case-control pairs, ascertained in 1,539,432 births from 49 hospitals in 38 cities. The highlands (above 2,000 m) comprised 3,498 case-control pairs from 129,301 births, occurring in four hospitals from three cities. Unconditional logistic regression was used to estimate the relative risks for the exposure at high altitudes, adjusted by ethnicity (Amerindian yes/no), type of health service (public/private), and birth weight (below/equal and greater than 2, 500 g). The adjusted relative risks showed significantly (P < 0.01) higher values in the high than in the lowlands for four types of defects: cleft lip [relative risks (RR): 1.57; 95% confidence interval (CI): 1.27-1.94], microtia (RR: 3.21; 95% CI: 2.35-4.79), preauricular tag (RR: 2.09; 95% CI: 1.86-2.36), branchial arch anomaly complex (RR: 1.79; 95% CI: 1.23-2.61), constriction band complex (RR: 1.92; 95% CI: 1.11-3.31), and anal atresia (RR: 1.61; 95% CI: 1.01-2.57). Conversely, lower risks in the highlands were registered for two neural tube defects: anencephaly (RR: 0.33; 95% CI: 0.20-0.54), spina bifida (RR: 0.57; 95% CI: 0.37-0.78), as well as for hydrocephaly (RR: 0.41; 95% CI: 0.22-0.77) and pes equinovarus (RR: 0.70; 95% CI: 0.51-0.96). Even though some of these differences may be caused by undetected confounders, the coincidental finding of four types of craniofacial defects with higher, and two types of neural tube defect with lower frequencies in the highlands, suggest a real biological foundation. Copyright 1999 Wiley-Liss, Inc.
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              Indoor air pollution from coal combustion and the risk of neural tube defects in a rural population in Shanxi Province, China.

              The authors evaluated indoor air pollution from coal combustion (IAPCC) as a potential risk factor for neural tube defects (NTDs) in a rural population in Shanxi Province, China. The studied rural population has both high IAPCC exposure and a high prevalence of NTDs. A population-based case-control study was used to identify 610 NTD cases and 837 normal controls between November 2002 and December 2007. Information was collected within 1 week following delivery or pregnancy termination due to prenatal NTD diagnosis. The authors derived an exposure index by integrating a series of IAPCC-related characteristics concerning dwelling and lifestyle. Compared with women with no IAPCC exposure, women with any exposure at all had a 60% increased risk of having a child with an NTD (adjusted odds ratio (OR) = 1.6, 95% confidence interval (CI): 1.1, 2.1). An increased NTD risk was linked to both residential heating (adjusted OR = 1.7, 95% CI: 1.1, 2.4) and cooking (adjusted OR = 1.5, 95% CI: 1.1, 2.1). The risk increased with increases in the exposure index, showing a dose-response trend (P < 0.001). This is the first known study to link IAPCC to NTDs. Additional studies are needed to confirm the link between IAPCC and NTDs.
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                Author and article information

                Contributors
                Role: Editor
                Journal
                PLoS One
                PLoS ONE
                plos
                plosone
                PLoS ONE
                Public Library of Science (San Francisco, CA USA )
                1932-6203
                21 April 2016
                2016
                : 11
                : 4
                : e0150332
                Affiliations
                [1 ]The State Key Laboratory of Resources and Environmental Information System, Institute of Geographic Sciences and Natural Resources Research, Chinese Academy of Sciences, Beijing, 100101, China
                [2 ]The Key Laboratory of Surveillance and Early-Warning on Infectious Disease, Chinese Center for Disease Control and Prevention, Beijing, 102206, China
                [3 ]Institute of Agricultural Economics and Information, Henan Academy of Agricultural Sciences, Zhengzhou, 450002, China
                [4 ]Department of Statistics, University of California, Berkeley, California, 94720, United States of America
                [5 ]Department of Spatial Sciences, Curtin University, Bentley, WA, 6845, Australia
                [6 ]Chang'an University, Xi’an, 710054, China
                [7 ]Jiangsu Center for Collaborative Innovation in Geographical Information Resource Development and Application, Nanjing, 210023, China
                Tabriz University of Medical Sciences, ISLAMIC REPUBLIC OF IRAN
                Author notes

                Competing Interests: The authors have declared that no competing interests exist.

                Conceived and designed the experiments: LYL. Performed the experiments: ZY HL. Analyzed the data: LX ZNX XB. Contributed reagents/materials/analysis tools: WJF. Wrote the paper: LX.

                Article
                PONE-D-15-44393
                10.1371/journal.pone.0150332
                4839670
                27100082
                666e681b-c286-4c86-9fae-f5b35ebbf476
                © 2016 Liao et al

                This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

                History
                : 12 October 2015
                : 14 December 2015
                Page count
                Figures: 3, Tables: 5, Pages: 14
                Funding
                This study was supported by the National Natural Science Foundation of China, http://www.nsfc.gov.cn/publish/portal1/ (Nos. 41471377, 41101431), and the Young Talent Fund of the Institute of Geographic Sciences and Natural Resources Research, CAS, http://english.igsnrr.cas.cn/ (2012RC201).
                Categories
                Research Article
                Engineering and Technology
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                Fuels
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