Article

Economic evaluation of a neural tube defect recurrence-prevention program

National Center on Birth Defects and Developmental Disabilities, CDC, Atlanta, Georgia 30333, USA.
American journal of preventive medicine (Impact Factor: 4.28). 11/2008; 35(6):572-7. DOI: 10.1016/j.amepre.2008.07.008
Source: PubMed

ABSTRACT Women with a pregnancy affected by a neural tube defect (NTD) are encouraged to take folic acid prior to a subsequent pregnancy, but it is unknown whether organized attempts to identify and counsel such women to prevent recurrent NTDs are cost effective.
Data from the South Carolina recurrence-prevention program for October 2001-September 2002 were analyzed between October 2002 and December 2003 to calculate costs. Cost-effectiveness modeling of the program during 1992-2006 was conducted during 2007. Results were calculated for three scenarios based on recurrence risk, supplement use, and the effectiveness of folic acid in preventing recurrences. For each scenario, quality-adjusted life years (QALYs) were calculated separately using prevented NTD-affected live births; prevented NTD-affected births (including fetal deaths); and all prevented NTD-affected pregnancies.
The prevention program cost approximately $155,000 per year in 2003 dollars to protect 35 pregnancies and prevent approximately one NTD. The direct costs associated with an NTD depend on type and outcome, but are approximately $560,000 in 2003 dollars for a live birth with spina bifida. The base-case cost-effectiveness ratio was $39,600 per QALY gained from avoided NTD-affected live births and stillbirths, and $14,700 per QALY gained from the avoidance of all NTD-affected pregnancies. The baseline NTD recurrence risk and the use of folic acid supplements by women who are at high risk for an NTD-affected pregnancy were influential parameters.
The South Carolina NTD recurrence-prevention program appears comparable in cost effectiveness to other preventive services. Other states might consider including NTD recurrence prevention in birth defect-prevention programs.

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    • "As a result of major advances in medical care, the majority of babies born with SB survive to adulthood with varying degrees of morbidity (Centers for Disease Control, 1992). Spina bifida represents a significant burden for those affected and their families with an estimated $806,000 in 2007 dollars for lifetime medical care costs when living up to 65 years of age (Grosse et al., 2008). This financial burden based on medical costs alone does not even begin to account for the societal costs or the emotional impact on the affected individuals and their families. "
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    ABSTRACT: Neural tube defects (NTDs) are congenital anomalies caused by a combination of genetic and environmental influences. A defect below the head region resulting in protuberance of meninges and nervous tissue is termed myelomeningocele (MM). MM, the most common NTD compatible with survival, occurs in approximately 1 in 1000 births worldwide. Maternal preconceptional and periconceptional folate supplementation reduces the risk of NTDs by up to 70%. A key enzyme in folate metabolism is 5, 10-methylene-tetrahydrofolate reductase (MTHFR). Sequence the 12 exons of the MTHFR gene among 96 subjects with MM to identify variants potentially contributing to the disease trait. Exons were amplified by polymerase chain reaction, and the products were sequenced with the Sanger method to reveal sequence variants compared to MTHFR reference sequences. Association of variants was examined by Fisher's test. A novel variant c.171+3G>T was identified in intron 1 in one affected subject. The variant was not found in the subject's unaffected mother's DNA, and the unaffected father's DNA was unavailable. We found significant differences in allele frequencies for seven SNPs in MM subjects compared with ethnically matched reference populations reported in the single nucleotide polymorphism database. We identified a novel variant c.171+3G>T in the MTHFR gene that potentially affects splicing in an affected subject. In addition, we observed five SNPs (rs13306561, rs2274976, rs2066462, rs12121543, and rs1476413) in the MTHFR gene not previously shown to associate with MM. The current study provides additional evidence that multiple variations in the MTHFR gene are associated with MM.
    Birth Defects Research Part A Clinical and Molecular Teratology 02/2012; 94(2):84-90. DOI:10.1002/bdra.22884
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    • "Subsequently, most westernized medical administrations, such as the United States Public Health Services, has recommended that women of childbearing age should consume 400 µg of folic acid daily during the periconceptional period (Fohr et al., 2002) due to its ability to alleviate congenital abnormalities (from 30 to 70%) (Fohr et al., 2002; Houcher et al., 2009). Furthermore, one study has reported that NTD-affected pregnant mothers who did not receive any folic acid supplementations exhibited a 2-3% recurrence during pregnancy (Grosse et al., 2008). "
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    ABSTRACT: Neural tube defects (NTDs) are severe but common congenital malformations. Neonates who suffer from NTDs may experience long-term complications throughout their lives. These NTD complications which have been reduced worldwide are primarily due to environmental and genetic factors. Multicenter NTD studies conducted in Malaysia report a prevalence ranging from 0.79 to 1.29 per 1000 live births based on NTD etiologies, such as anti-epileptic drug consumption and maternal folate levels. In addition, intervention studies concluded that daily consumption of 400 µg of folic acid effectively reduced NTD risk; however, this data has not been robustly tested on the entire population due to the inefficiency of the three interrelated folate transport mechanisms and autoantibody generation. In this review, we evaluated the studies indicating that folic acid supplementation may not be the sole factor in reducing NTD incidence and that autoantibodies may have an important role in the NTD etiological pathway.
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    • "nually , with more aborted spontaneously and electively terminated ( reviewed by Detrait , George et al . 2005 ) . Anencephaly is fatal in all cases whereas children with spina bifida frequently suffer from severe disability ( Date , Yagyu et al . 1993 ) and require continued medical treatment ( Zurmohle , Homann et al . 1998 ) . In 2003 dollars , Grosse et al . ( 2008 ) estimated the direct lifetime costs of spina bifida to be $ 560 , 000 per child . Randomized controlled trials indicated that folic acid supplementation could prevent at least one - half of all NTDs ( Medical Research"
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    ABSTRACT: With a birth prevalence of 1 in 1000, neural tube defects (NTD)s contribute considerably to morbidity and healthcare costs. Known genetic and environmental (non-inherited) risk factors for NTDs account for a small portion of risk, suggesting unidentified risk factors. In animal studies, maternal alcohol and pesticide exposures, independently, led to excess neural cell death, resulting in too few cells for neural tube closure. Human studies report no association between alcohol exposure and NTDs, but small to moderate positive associations for pesticide exposure. Such human etiologic studies of NTDs require a large base population, but frequently include only live births. Exclusion of cases by pregnancy outcomes may create ascertainment and response bias, complicating interpretation of findings. Using data from the National Birth Defects Prevention Study (NBDPS) and the Iowa Registry for Congenital and Inherited Disorders (IRCID), the independent effects of maternal periconceptional (1 month prior through 2 months postconception) alcohol and occupational pesticide exposure on the development of NTDs were examined, and differences in Iowa NTD cases were characterized by pregnancy outcome. Maternal reports of alcohol exposure were obtained for 1223 NTD case infants and 6807 control infants. Adjusted odds ratios, estimated using multivariate logistic regression, were near unity for NTDs by any maternal alcohol exposure, binge episode(s), and type(s) of alcohol consumed. Occupational pesticide exposure was assigned by industrial hygienists for mothers of 502 case and 2950 control infants. Adjusted odds ratios for any exposure and cumulative exposure to any pesticide, insecticides only, and insecticides + herbicides + fungicides were near unity for NTDs. Insecticide + herbicide exposure was positively associated with spina bifida. Among the 279 Iowa NTD case infants ascertained by the IRCID, 167 live births and 112 were other pregnancy outcomes (fetal deaths and elective terminations), which increased in proportion over time. Selected infant and maternal characteristics of live births and other pregnancy outcomes were similar. NBDPS eligibility varied significantly by pregnancy outcome, but participation rates did not. NTD case mothers were similar to Iowa NBDPS control mothers. Efforts were made to improve upon prior etiologic studies of these exposures and NTDs, including increased sample size and improved exposure specificity. Some exposure strata (e.g., herbicides only) and outcome strata (e.g., other rare subtypes) were limited by small numbers. All results may have been affected by response and ascertainment bias. Future studies should aim to use similarly detailed exposure classification methods, increase sample size in less prevalent NTD subtypes, and improve ascertainment of fetal deaths.
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