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Published in: European Journal of Pediatrics 11/2011

Open Access 01-11-2011 | Review

Economic burden of neural tube defects and impact of prevention with folic acid: a literature review

Authors: Yunni Yi, Marion Lindemann, Antje Colligs, Claire Snowball

Published in: European Journal of Pediatrics | Issue 11/2011

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Abstract

Neural tube defects (NTDs) are the second most common group of serious birth defects. Although folic acid has been shown to reduce effectively the risk of NTDs and measures have been taken to increase the awareness, knowledge, and consumption of folic acid, the full potential of folic acid to reduce the risk of NTDs has not been realized in most countries. To understand the economic burden of NTDs and the economic impact of preventing NTDs with folic acid, a systematic review was performed on relevant studies. A total of 14 cost of illness studies and 10 economic evaluations on prevention of NTDs with folic acid were identified. Consistent findings were reported across all of the cost of illness studies. The lifetime direct medical cost for patients with NTDs is significant, with the majority of cost being for inpatient care, for treatment at initial diagnosis in childhood, and for comorbidities in adult life. The lifetime indirect cost for patients with spina bifida is even greater due to increased morbidity and premature mortality. Caregiver time costs are also significant. The results from the economic evaluations demonstrate that folic acid fortification in food and preconception folic acid consumption are cost-effective ways to reduce the incidence and prevalence of NTDs. This review highlights the significant cost burden that NTDs pose to healthcare systems, various healthcare payers, and society and concludes that the benefits of prevention of NTDs with folic acid far outweigh the cost. Further intervention with folic acid is justified in countries where the full potential of folic acid to reduce the risk of NTDs has not been realized.
Literature
1.
go back to reference Armour BS, Ouyang L, Thibadeau J, Grosse SD, Campbell VA, Joseph D (2009) Hospitalization for urinary tract infections and the quality of preventive health care received by people with spina bifida. Disabil Health J 2:145–152PubMedCrossRef Armour BS, Ouyang L, Thibadeau J, Grosse SD, Campbell VA, Joseph D (2009) Hospitalization for urinary tract infections and the quality of preventive health care received by people with spina bifida. Disabil Health J 2:145–152PubMedCrossRef
2.
go back to reference Bamer AM, Connell FA, Dudgeon BJ, Johnson KL (2010) Frequency of purchase and associated costs of assistive technology for Washington State Medicaid program enrollees with spina bifida by age. Disabil Health J 3:155–161PubMedCrossRef Bamer AM, Connell FA, Dudgeon BJ, Johnson KL (2010) Frequency of purchase and associated costs of assistive technology for Washington State Medicaid program enrollees with spina bifida by age. Disabil Health J 3:155–161PubMedCrossRef
3.
go back to reference Bea M, Diaz LI, Martinez A, Lopez A, Garcia AV, Forner V (1994) A multicentre study of the hospital care of 1500 patients with myelomeningocele. Paraplegia 32:561–564CrossRef Bea M, Diaz LI, Martinez A, Lopez A, Garcia AV, Forner V (1994) A multicentre study of the hospital care of 1500 patients with myelomeningocele. Paraplegia 32:561–564CrossRef
4.
go back to reference Bentley TG, Weinstein MC, Willett WC, Kuntz KM (2008) A cost-effectiveness analysis of folic acid fortification policy in the US. Public Health Nutr 12:455–467PubMedCrossRef Bentley TG, Weinstein MC, Willett WC, Kuntz KM (2008) A cost-effectiveness analysis of folic acid fortification policy in the US. Public Health Nutr 12:455–467PubMedCrossRef
5.
go back to reference Berry RJ, Li Z, Erickson JD, Li S, Moore CA, Wang H et al (1999) Prevention of neural-tube defects with folic acid in China. China–US Collaborative Project for Neural Tube Defect Prevention. N Engl J Med 341:1485–1490PubMedCrossRef Berry RJ, Li Z, Erickson JD, Li S, Moore CA, Wang H et al (1999) Prevention of neural-tube defects with folic acid in China. China–US Collaborative Project for Neural Tube Defect Prevention. N Engl J Med 341:1485–1490PubMedCrossRef
6.
go back to reference Blencowe H, Cousens S, Modell B, Lawn J (2010) Folic acid to reduce neonatal mortality from neural tube disorders. Int J Epidemiol 39:i110–i121PubMedCrossRef Blencowe H, Cousens S, Modell B, Lawn J (2010) Folic acid to reduce neonatal mortality from neural tube disorders. Int J Epidemiol 39:i110–i121PubMedCrossRef
7.
go back to reference Botto LD, Moore CA, Khoury MJ, Erickson JD (1999) Neural-tube defects. N Engl J Med 341:1509–1519PubMedCrossRef Botto LD, Moore CA, Khoury MJ, Erickson JD (1999) Neural-tube defects. N Engl J Med 341:1509–1519PubMedCrossRef
8.
go back to reference Case AP, Canfield MA (2009) Methods for developing useful estimation of the costs associated with birth defects. Birth Defects Res A Clin Mol Teratol 85:920–924PubMedCrossRef Case AP, Canfield MA (2009) Methods for developing useful estimation of the costs associated with birth defects. Birth Defects Res A Clin Mol Teratol 85:920–924PubMedCrossRef
9.
go back to reference Castilla E, Orioli I, Lopez C, Dutra M, Nazer H (2003) Preliminary data on changes in neural tube defect prevalence rates after folic acid fortification in South America. Am J Med Genet A 123:123–128CrossRef Castilla E, Orioli I, Lopez C, Dutra M, Nazer H (2003) Preliminary data on changes in neural tube defect prevalence rates after folic acid fortification in South America. Am J Med Genet A 123:123–128CrossRef
10.
go back to reference Centers for Disease Control and Prevention (1992) Recommendations for the use of folic acid to reduce the number of cases of spina bifida and other neural tube defects. MMWR 41:1–7 Centers for Disease Control and Prevention (1992) Recommendations for the use of folic acid to reduce the number of cases of spina bifida and other neural tube defects. MMWR 41:1–7
11.
go back to reference Centers for Disease Control and Prevention (1999) Annex A: fact sheets for candidates diseases for elimination or eradication. MMWR Supplements 48(SU01):154–203 Centers for Disease Control and Prevention (1999) Annex A: fact sheets for candidates diseases for elimination or eradication. MMWR Supplements 48(SU01):154–203
12.
go back to reference Centers for Disease Control and Prevention (2004) Spina bifida and anencephaly before and after folic acid mandate—United States, 1995–1996 and 1999–2000. MMWR 53:362–365 Centers for Disease Control and Prevention (2004) Spina bifida and anencephaly before and after folic acid mandate—United States, 1995–1996 and 1999–2000. MMWR 53:362–365
13.
go back to reference Centers for Disease Control and Prevention (2008) Use of supplements containing folic acid among women of childbearing age—United States. MMWR 57(1):5–8 Centers for Disease Control and Prevention (2008) Use of supplements containing folic acid among women of childbearing age—United States. MMWR 57(1):5–8
15.
go back to reference Chen L, Rivera M (2004) The Costa Rican experience: reduction of neural tube defects following food fortification programs. Nutr Rev 62:S40–S43PubMedCrossRef Chen L, Rivera M (2004) The Costa Rican experience: reduction of neural tube defects following food fortification programs. Nutr Rev 62:S40–S43PubMedCrossRef
17.
go back to reference Cooper NJ (2000) Economic burden of rheumatoid arthritis: a systematic review. Rheumatology 39:28–33PubMedCrossRef Cooper NJ (2000) Economic burden of rheumatoid arthritis: a systematic review. Rheumatology 39:28–33PubMedCrossRef
18.
go back to reference Dalziel KS, Segal L, Katz R (2010) Cost-effectiveness of mandatory folate fortification v. other options for the prevention of neural tube defects: results from Australia and New Zealand. Public Health Nutr 13:566–578PubMedCrossRef Dalziel KS, Segal L, Katz R (2010) Cost-effectiveness of mandatory folate fortification v. other options for the prevention of neural tube defects: results from Australia and New Zealand. Public Health Nutr 13:566–578PubMedCrossRef
19.
go back to reference Date I, Yagyu Y, Asari S, Ohmoto T (1993) Long-term outcome in surgically treated spina bifida cystica. Surg Neurol 40:471–475PubMedCrossRef Date I, Yagyu Y, Asari S, Ohmoto T (1993) Long-term outcome in surgically treated spina bifida cystica. Surg Neurol 40:471–475PubMedCrossRef
20.
go back to reference De Wals P, Tairou F, Van A, Uh S, Lowry R, Sibbald B et al (2007) Reduction in neural-tube defects after folic acid fortification in Canada. N Engl J Med 357:135–142PubMedCrossRef De Wals P, Tairou F, Van A, Uh S, Lowry R, Sibbald B et al (2007) Reduction in neural-tube defects after folic acid fortification in Canada. N Engl J Med 357:135–142PubMedCrossRef
21.
go back to reference Grace HJ (1981) Prenatal screening for neural tube defects in South Africa An assessment. S Afr Med J 60:324–329PubMed Grace HJ (1981) Prenatal screening for neural tube defects in South Africa An assessment. S Afr Med J 60:324–329PubMed
22.
go back to reference Grosse SD, Ouyang L, Collins JS et al (2008) Economic evaluation of a neural tube defect recurrence-prevention program. Am J Prev Med 35:572–577PubMedCrossRef Grosse SD, Ouyang L, Collins JS et al (2008) Economic evaluation of a neural tube defect recurrence-prevention program. Am J Prev Med 35:572–577PubMedCrossRef
23.
go back to reference Grosse SD, Waitzman NJ, Romano PS, Mulinare J (2005) Reevaluating the benefits of folic acid fortification in the United States: economic analysis, regulation, and public health. Am J Public Health 95:1917–1922PubMedCrossRef Grosse SD, Waitzman NJ, Romano PS, Mulinare J (2005) Reevaluating the benefits of folic acid fortification in the United States: economic analysis, regulation, and public health. Am J Public Health 95:1917–1922PubMedCrossRef
24.
go back to reference Harris MB, Banta JV (1990) Cost of skin care in the myelomeningocele population. J Pediatr Orthop 10:355–361PubMedCrossRef Harris MB, Banta JV (1990) Cost of skin care in the myelomeningocele population. J Pediatr Orthop 10:355–361PubMedCrossRef
25.
go back to reference Hertrampf E, Cortes F (2008) National food-fortification program with folic acid in Chile. Food Nutr Bull 29:S231–S237PubMed Hertrampf E, Cortes F (2008) National food-fortification program with folic acid in Chile. Food Nutr Bull 29:S231–S237PubMed
26.
go back to reference Heseker HB, Mason JB, Selhub J, Rosenberg IH, Jacques PF (2009) Not all cases of neural-tube defect can be prevented by increasing the intake of folic acid. Br J Nutr 102:173–180PubMedCrossRef Heseker HB, Mason JB, Selhub J, Rosenberg IH, Jacques PF (2009) Not all cases of neural-tube defect can be prevented by increasing the intake of folic acid. Br J Nutr 102:173–180PubMedCrossRef
27.
go back to reference Honein MA, Paulozzi LJ, Mathews TJ, Erickson JD, Wong LY (2001) Impact of folic acid fortification of the US food supply on the occurrence of neural tube defects. JAMA 285:2981–2986PubMedCrossRef Honein MA, Paulozzi LJ, Mathews TJ, Erickson JD, Wong LY (2001) Impact of folic acid fortification of the US food supply on the occurrence of neural tube defects. JAMA 285:2981–2986PubMedCrossRef
28.
go back to reference Ireys HT, Anderson GF, Shaffer TJ, Neff JM (1997) Expenditures for care of children with chronic illnesses enrolled in the Washington State Medicaid program, fiscal year 1993. Pediatrics 100:197–204PubMedCrossRef Ireys HT, Anderson GF, Shaffer TJ, Neff JM (1997) Expenditures for care of children with chronic illnesses enrolled in the Washington State Medicaid program, fiscal year 1993. Pediatrics 100:197–204PubMedCrossRef
29.
go back to reference Jaquier M, Klein A, Boltshauser E (2006) Spontaneous pregnancy outcome after prenatal diagnosis of anencephaly. BJOG 113:951–953PubMedCrossRef Jaquier M, Klein A, Boltshauser E (2006) Spontaneous pregnancy outcome after prenatal diagnosis of anencephaly. BJOG 113:951–953PubMedCrossRef
30.
go back to reference Jentink J, van de Vrie-Hoekstra NW, de Jong-van den Berg LT, Postma MJ (2008) Economic evaluation of folic acid food fortification in the Netherlands. Eur J Public Health 18:270–274PubMedCrossRef Jentink J, van de Vrie-Hoekstra NW, de Jong-van den Berg LT, Postma MJ (2008) Economic evaluation of folic acid food fortification in the Netherlands. Eur J Public Health 18:270–274PubMedCrossRef
31.
go back to reference Kinsman SL, Doehring MC (1996) The cost of preventable conditions in adults with spina bifida. Eur J Pediatr Surg 6:17–20PubMedCrossRef Kinsman SL, Doehring MC (1996) The cost of preventable conditions in adults with spina bifida. Eur J Pediatr Surg 6:17–20PubMedCrossRef
32.
go back to reference Landon J, Thorpe L (1998) Changing preconceptions: volume 1: the HEA folic acid campaign 1995–1998: summary report. Landon J, Thorpe L (1998) Changing preconceptions: volume 1: the HEA folic acid campaign 1995–1998: summary report.
34.
go back to reference Lipscomb J (1986) Human capital, willingness-to-pay and cost-effectiveness analyses of screening for birth defects in North Carolina. Duke University, Durham Lipscomb J (1986) Human capital, willingness-to-pay and cost-effectiveness analyses of screening for birth defects in North Carolina. Duke University, Durham
35.
go back to reference Liu S, West R, Randell E, Longerich L, Connor KS, Scott H et al (2004) A comprehensive evaluation of food fortification with folic acid for the primary prevention of neural tube defects. BMC Pregnancy Childbirth 4:10CrossRef Liu S, West R, Randell E, Longerich L, Connor KS, Scott H et al (2004) A comprehensive evaluation of food fortification with folic acid for the primary prevention of neural tube defects. BMC Pregnancy Childbirth 4:10CrossRef
36.
go back to reference Llanos A, Hertrampf E, Cortes F, Pardo A, Grosse SD, Uauy R (2007) Cost-effectiveness of a folic acid fortification program in Chile. Health Policy 83:295–303PubMedCrossRef Llanos A, Hertrampf E, Cortes F, Pardo A, Grosse SD, Uauy R (2007) Cost-effectiveness of a folic acid fortification program in Chile. Health Policy 83:295–303PubMedCrossRef
37.
go back to reference National Health and Medical Research Council (1993) Revised statement on the relationship between dietary folic acid and neural tube defects such as spina bifida. 15th Session of the National Health and Medical Research Council, Australia National Health and Medical Research Council (1993) Revised statement on the relationship between dietary folic acid and neural tube defects such as spina bifida. 15th Session of the National Health and Medical Research Council, Australia
38.
go back to reference Mosley B, Hobbs C, Flowers B, Smith V, Robbins J (2007) Folic acid and the decline in neural tube defects in Arkansas. J Ark Med Soc 103:247–250PubMed Mosley B, Hobbs C, Flowers B, Smith V, Robbins J (2007) Folic acid and the decline in neural tube defects in Arkansas. J Ark Med Soc 103:247–250PubMed
40.
go back to reference Ouyang LJ, Grosse SD, Armour BS, Waitzman NJ (2007) Health care expenditures of children and adults with spina bifida in a privately insured US population. Birth Defects Res A Clin Mol Teratol 79:552–558PubMedCrossRef Ouyang LJ, Grosse SD, Armour BS, Waitzman NJ (2007) Health care expenditures of children and adults with spina bifida in a privately insured US population. Birth Defects Res A Clin Mol Teratol 79:552–558PubMedCrossRef
41.
go back to reference Postma MJ, Londeman J, Veenstra M, De Walle HEK, de Jong van den Berg LTW (2002) Cost-effectiveness of periconceptional supplementation of folic acid. Pharm World Sci 24:8–11PubMedCrossRef Postma MJ, Londeman J, Veenstra M, De Walle HEK, de Jong van den Berg LTW (2002) Cost-effectiveness of periconceptional supplementation of folic acid. Pharm World Sci 24:8–11PubMedCrossRef
42.
go back to reference Ray J, Meier C, Vermeulen M, Boss S, Wyatt P, Cole D (2002) Association of neural tube defects and folic acid food fortification in Canada. Lancet 360:2047–2048PubMedCrossRef Ray J, Meier C, Vermeulen M, Boss S, Wyatt P, Cole D (2002) Association of neural tube defects and folic acid food fortification in Canada. Lancet 360:2047–2048PubMedCrossRef
43.
go back to reference Robbins JM, Bird TM, Tilford JM et al (2007) Hospital stays, hospital charges, and in-hospital deaths among infants with selected birth defects: United States, 2003. MMWR 56(2):25–29 Robbins JM, Bird TM, Tilford JM et al (2007) Hospital stays, hospital charges, and in-hospital deaths among infants with selected birth defects: United States, 2003. MMWR 56(2):25–29
44.
go back to reference Romano PS, Waitzman NJ, Scheffler RM, Pi RD (1995) Folic acid fortification of grain: an economic analysis. Am J Publ Health 85:667–676CrossRef Romano PS, Waitzman NJ, Scheffler RM, Pi RD (1995) Folic acid fortification of grain: an economic analysis. Am J Publ Health 85:667–676CrossRef
45.
go back to reference Sayed AR, Bourne D, Pattinson R, Nixon J, Henderson B (2008) Decline in the prevalence of neural tube defects following folic acid fortification and its cost-benefit in South Africa. Birth Defects Res A Clin Mol Teratol 82:211–216PubMedCrossRef Sayed AR, Bourne D, Pattinson R, Nixon J, Henderson B (2008) Decline in the prevalence of neural tube defects following folic acid fortification and its cost-benefit in South Africa. Birth Defects Res A Clin Mol Teratol 82:211–216PubMedCrossRef
46.
go back to reference Scofield JC, Campbell PR (2001) Integrating the spina bifida patient into the general dental practice. The Journal of Practical Hygiene. May and June 27–31 Scofield JC, Campbell PR (2001) Integrating the spina bifida patient into the general dental practice. The Journal of Practical Hygiene. May and June 27–31
47.
go back to reference Tilford JM, Grosse SD, Goodman AC, Li K (2009) Labor market productivity costs for caregivers of children with spina bifida: a population-based analysis. Med Decis Mak 29:23–32CrossRef Tilford JM, Grosse SD, Goodman AC, Li K (2009) Labor market productivity costs for caregivers of children with spina bifida: a population-based analysis. Med Decis Mak 29:23–32CrossRef
48.
go back to reference Tilford JM, Robbins JM, Hobbs CA (2001) Improving estimates of caregiver time cost and family impact associated with birth defects. Teratology 64:S37–S41PubMedCrossRef Tilford JM, Robbins JM, Hobbs CA (2001) Improving estimates of caregiver time cost and family impact associated with birth defects. Teratology 64:S37–S41PubMedCrossRef
49.
go back to reference Waitzman, NJ, Romano, PS, Grosse, SD (2004) The half-life of cost-of-illness estimates: the case of spina bifida. Working Paper No: 2004–07. Department of Economics, University of Utah Waitzman, NJ, Romano, PS, Grosse, SD (2004) The half-life of cost-of-illness estimates: the case of spina bifida. Working Paper No: 2004–07. Department of Economics, University of Utah
50.
go back to reference Waitzman NJ, Scheffler RM, Romano PS (1996) The cost of birth defects: estimates of the value of prevention. University Press of America, Lanham Waitzman NJ, Scheffler RM, Romano PS (1996) The cost of birth defects: estimates of the value of prevention. University Press of America, Lanham
51.
go back to reference Williams P, Williams A, Graff J, Hanson S, Stanton A, Hafeman C et al (2002) Interrelationships among variables affecting well siblings and mothers in families of children with a chronic illness or disability. J Behav Med 25:411–424PubMedCrossRef Williams P, Williams A, Graff J, Hanson S, Stanton A, Hafeman C et al (2002) Interrelationships among variables affecting well siblings and mothers in families of children with a chronic illness or disability. J Behav Med 25:411–424PubMedCrossRef
52.
go back to reference Wolff T, Witkop CT, Miller T, Syed S (2009) Folic acid supplementation for the prevention of neural tube defects: an update of the evidence for the US preventive services task force. Ann Intern Med 150:632–639PubMed Wolff T, Witkop CT, Miller T, Syed S (2009) Folic acid supplementation for the prevention of neural tube defects: an update of the evidence for the US preventive services task force. Ann Intern Med 150:632–639PubMed
53.
go back to reference Young N, Steele C, Fehlings D, Jutai J, Olmsted N, Williams J, Williams JI (2005) Use of health care among adults with chronic and complex physical disabilities of childhood. Disabil Rehabil 27:1455–1460PubMedCrossRef Young N, Steele C, Fehlings D, Jutai J, Olmsted N, Williams J, Williams JI (2005) Use of health care among adults with chronic and complex physical disabilities of childhood. Disabil Rehabil 27:1455–1460PubMedCrossRef
54.
go back to reference Zlotogora J, Amitai Y, Leventhal A (2006) Surveillance of neural tube defects in Israel: the effect of the recommendation for periconceptional folic acid. Isr Med Assoc J 8:601–604PubMed Zlotogora J, Amitai Y, Leventhal A (2006) Surveillance of neural tube defects in Israel: the effect of the recommendation for periconceptional folic acid. Isr Med Assoc J 8:601–604PubMed
55.
go back to reference Zurmohle UM, Homann T, Schroeter C, Rothgerber H, Hommel G, Ermert J (1998) Psychosocial adjustment of children with spina bifida. J Child Neurol 13:64–70PubMedCrossRef Zurmohle UM, Homann T, Schroeter C, Rothgerber H, Hommel G, Ermert J (1998) Psychosocial adjustment of children with spina bifida. J Child Neurol 13:64–70PubMedCrossRef
Metadata
Title
Economic burden of neural tube defects and impact of prevention with folic acid: a literature review
Authors
Yunni Yi
Marion Lindemann
Antje Colligs
Claire Snowball
Publication date
01-11-2011
Publisher
Springer-Verlag
Published in
European Journal of Pediatrics / Issue 11/2011
Print ISSN: 0340-6199
Electronic ISSN: 1432-1076
DOI
https://doi.org/10.1007/s00431-011-1492-8

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