Hostname: page-component-586b7cd67f-l7hp2 Total loading time: 0 Render date: 2024-11-27T19:53:34.503Z Has data issue: false hasContentIssue false
  • English
  • Français

The Association of Twinning and Neural Tube Defects: Studies in Los Angeles, California, and Norway

Published online by Cambridge University Press:  01 August 2014

Gayle C. Windham ,
Tor Bjerkedal  and
Lowell E. Sever
Gayle C. Windham*
Affiliation:
Institute of Preventive Medicine, University of Oslo, Los Angeles Division of Epidemiology, School of Public Health, University of California, Los Angeles
Tor Bjerkedal
Affiliation:
Institute of Preventive Medicine, University of Oslo, Los Angeles
Lowell E. Sever*
Affiliation:
Division of Epidemiology, School of Public Health, University of California, Los Angeles
*
National Institute of Public Health, Postuttak Oslo 1, Norway
Battelle Pacific Northwest Laboratories, Richland, WA 99352, USA

Abstract

Core share and HTML view are not available for this content. However, as you have access to this content, a full PDF is available via the ‘Save PDF’ action button.

Accurate, unbiased malformation rates in twins must be obtained unselectively from population-based studies that include livebirths and stillbirths after a thorough ascertainment of cases. This type of study was conducted in Los Angeles County, California, where 28 twins with a neural tube defect (NTD) were identified. The prevalence in twins (1.6/1,000) was significantly higher than in singletons (1.1/1,000). The study then was expanded to include population-based data from the Medical Birth Registry of Norway which has a comparable overall NTD prevalence (1.0/1,000) and twinning rate (2%). The combined material shows a higher prevalence of anencephaly and encephalocele but not of spina bifida in twins compared to singletons. The male/female ratios in total twin and singleton cases were comparable (0.8), but varied by specific defect. Like-sex twin females appeared at highest risk for NTD as well as for fetal death.

This study supports theories which associate NTDs with monozygotic twins, either through developmental disruptions that cause susceptibility to environmental agents or through a common etiology. Furthermore, it suggests that twins and singletons differ in their response to etiologic factors for the development of NTDs and that the development of each type of NTD may be related to different factors.

Keywords

Neural tube defectsTwinningAnencephalySpina bifidaEncephaloceleCase ascertainmentMalformations in twins
Type
Research Article
Information
Acta geneticae medicae et gemellologiae: twin research , Volume 31 , Issue 3-4: Special Issue on Twin Pregnancies , July 1982 , pp. 165 - 172
Copyright
Copyright © The International Society for Twin Studies 1982

References

REFERENCES

1. Bjerkedal, T, Bakketeig, LS (1975): Surveillance of congenital malformations and other conditions of the newborn. Int J Epidemiol 4:31–36.Google Scholar
2. Buckley, MR, Erten, O (1979): The epidemiology of anencephaly and spina bifida in Izmir, Turkey, in the light of recent aetiological theories. J Epidemiol Commun Health 33:186–190.CrossRefGoogle Scholar
3. Carter, CO, Evans, K (1973): Spina bifida and anencephalus in greater London. J Med Genet 10:209–234.CrossRefGoogle ScholarPubMed
4. Cederbaum, SD (1978): Genetic aspects of neural tube defects. In Crandall, BF, Brazier, MAB (eds): “The Prevention of Neural Tube Defects: The Role of Alpha-Fetoprotein.” New York: Academic Press, pp 91–97.Google Scholar
5. Elwood, JM, Elwood, JH (1980): “Epidemiology of Anencephalus and Spina Bifida.” Oxford: Oxford University Press.Google Scholar
6. Feldman, JG, Stein, SC, Klein, RJ, Koyl, S, Casey, G (1982): The prevalence of neural tube defects among ethnic groups in Brooklyn, New York. J Chronic Dis 35:53–60.CrossRefGoogle ScholarPubMed
7. Field, B, Kerr, C (1974): Twinning and neural tube defects. Lancet 2:964–965.Google Scholar
8. Gittelsohn, AM, Milham, S (1965): Vital record incidence of congenital malformations in New York State. In Neel, JV, Shaw, MJ, Shull, WJ (ed): “Genetics and the Epidemiology of Chronic Diseases.” Washington: U.S. Department of Health, Education and Welfare, pp 305–319.Google Scholar
9. Granroth, G, Hakama, M, Saxen, L (1977): Defects of the central nervous system in Finland: I. Variations in time and space, sex distribution, and parental age. Br J Prev Soc Med 31:164–170.Google ScholarPubMed
10. Granroth, G, Haapakoski, J, Hakama, M (1978): Defects of the central nervous system in Finland: II. Birth order, outcome of previous pregnancies and family history. Teratology 17:213–222.Google Scholar
11. Guha-Ray, DK (1977): Anencephaly-survey of 60 cases. Penn Med 80:46–49.Google ScholarPubMed
12. Hay, S, Wehrung, DA (1970): Congenital malformations in twins. Am J Hum Genet 22:662–678.Google Scholar
13. James, WH (1976): Twinning and anencephaly. Ann Hum Biol 3:401–409.CrossRefGoogle ScholarPubMed
14. James, WH (1981): Differences between the events preceding spina bifida and anencephaly. J. Med Genet 18:17–21.Google Scholar
15. Janerich, DT, Piper, J (1978): Shifting genetic patterns in anencephaly and spina bifida. J Med Genet 15:101–105.Google Scholar
16. Layde, PM, Erickson, JD, Falek, A, McCarthy, BJ (1980): Congenital malformations in twins. Am J Hum Genet 32:69–78.Google ScholarPubMed
17. McBride, ML (1979): Sib risks of anencephaly and spina bifida in British Columbia. Am J Med Genet 3:377–387.Google Scholar
18. Myrianthopoulos, NC (1978): Congenital malformations: The contribution of twin studies. Birth Defects 14:151–165.Google ScholarPubMed
19. Renwick, JH (1972): Hypothesis: Anencephaly and spina bifida are usually preventable by the avoidance of a specific but unidentified substance present in certain potato tubers. Br J Prev Soc Med 26:67–77.Google Scholar
20. Schinzel, AAGL, Smith, DW, Miller, JR (1979): Monozygotic twinning and structural defects. J Pediatr 95:921–930.Google Scholar
21. Sever, LE (1976): Anencephalus and spina bifida: An ecological approach. Hum Ecol 4:209–221.Google Scholar
22. Sever, LE (1982): An epidemiologic study of neural tube defects in Los Angeles County. II. Etiologic factors in an area with low prevalence at birth. Teratology 25:323–334.Google Scholar
23. Sever, LE (1978): Epidemiologic aspects of neural tube defects. In Crandall, BF, Brazier, MAB (eds): “The Prevention of Neural Tube Defects: The Role of Alpha-Fetoprotein.” New York: Academic Press, pp 75–89.Google Scholar
24. Shapiro, LR, Zemek, L, Shulman, MJ (1978): Genetic etiology for monozygotic twinning. Birth Defects 15:219–222.Google Scholar
25. Strassburg, MA (1981): The Epidemiology of Anencephalus and Spina Bifida in Los Angeles County. Doctor of Public Health Dissertation, University of California, Los Angeles.Google Scholar
26. Windham, GC, Sever, LE (1982): Neural tube defects among twin births. Am J Hum Genet 34:(in press).Google Scholar
27. Windham, GC, Bjerkedal, T (1982): Secular trends of neural tube defects by demographic subgroups in Norway, 1967–1981. Natl Inst Public Health Ann 6:(in press).Google Scholar