Risk factors of the appearance of anencephaly in Tunisia

##plugins.themes.academic_pro.article.sidebar##

Published Jan 2, 2025
https://doi.org/10.62438/tunismed.v103i1.5086
Download
pdf
Vol. 103 No. 1 (2025): Tunis Med Jan 2025

##plugins.themes.academic_pro.article.main##

Kaouther Nasri
Department of embryo-fetopathology, La Rabta Maternity and Neonatology Center, El Manar II University, Tunis. Faculty of Sciences of Bizerte, University of Carthage, Zarzouna, Bizerte Tunisia.
Nadia Ben Jemaa
Department of histology-embryology, Faculty of Medicine of Tunis, Tunis El Manar University, Tunisia.
Soumaya Siala Gaigi
Department of embryo-fetopathology, La Rabta Maternity and Neonatology Center, El Manar II University, Tunis, Tunisia.

Abstract

 


Introduction: Anencephaly is a serious developmental defect of the central nervous system in which the brain and cranial vault are grossly malformed. The cerebrum and cerebellum are reduced or absent, but the hindbrain is present. Anencephaly is a part of the neural tube defect spectrum. This defect results when the neural tube fails to close during the third to fourth weeks of development, leading to fetal loss, stillbirth, or neonatal death.


Aim: To find out probable principal risk factors for the appearance of anencephaly.


Methods: This study was conducted to compare between pregnancies affected by anencephaly in 2002-2011 with those notified in the period 1991–2001. Statistical analysis was undertaken using chi-squared tests.


Results: Results had shown that anencephaly fetuses with a weight less than 1500 g were significantly higher in the period 2002-2011 than in 1991-2001 (P=0.003; OR= 4.32; CI= 1.62-11.53). Anencephaly cases aged more than 20 weeks of gestation (WG) were statistically elevated than cases aged less than 20 WG (P= 0.003). Maternal parity was associated with the appearance of anencephaly, where uni- or multiparous cases mothers were more likely to have an offspring with anencephaly than nulliparous mothers. Consanguinity presented a significant risk factor for the occurrence of anencephaly (P= 0.003). A logistic regression was run to examine the impact of several variables, only the maternal age was statistically significant.


Conclusion: This study clarified fields where efforts should be intensified, and surveillance data developed to prevent this malformation.

Keywords:

Anencephaly, Epidemiology, Feto-maternal characteristics, Risk factors, Tunisia

##plugins.themes.academic_pro.article.details##

Creative Commons License

This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.

References

  1. Rampersaud E, Bassuk AG, Enterline DS, George TM, Siegel DG, Melvin EC et al. Whole genomewide linkage screen for neural tube defects reveals regions of interest on chromosomes 7 and 10. J Med Genet. 2005;42(12):940-6.
  2. Golalipour M, Najafi L, Keshtkar A. Neural tube defects in native fars ethnicity in northern iran. Iran J Public Health. 2010;39(3):116-23.
  3. Khattak ST, Naheed T, Akhtar S, Jamal T. Incidence and risk factors for neural tube defects in Peshawar. Gomal J Med Sci. 2008;6: 1–4.
  4. Nili F, Jahangiri M. Risk factors for neural tube defects: a study at university-affiliated hospitals in Tehran. Arch Iran Med. 2006;9(1):20-5.
  5. De Marco P, Merello E, Calevo MG, Mascelli S, Pastorino D, Crocetti L, et al. Maternal periconceptional factors affect the risk of spina bifida-affected pregnancies: an Italian case-control study. Childs Nerv Syst. 2011;27(7):1073-81.
  6. Yin Z, Xu W, Xu C, Zhang S, Zheng Y, Wang W, et al. A population based case-control study of risk factors for neural tube defects in Shenyang, China. Childs Nerv Syst. 2011;27(1):149-54.
  7. Canfield MA, Marengo L, Ramadhani TA, Suarez L, Brender JD, Scheuerle A. The prevalence and predictors of anencephaly and spina bifida in Texas. Paediatr Perinat Epidemiol. 2009;23(1):41-50.
  8. Mandiracioglu A, Ulman I, Luleci E, Ulman C. The incidence and risk factors of neural tube defects in Izmir, Turkey: a nested case control study. Turk J Pediatr. 2004;46(3):214-20.
  9. Golalipour M, Najafi L, Keshtkar A. Prevalence of Anencephaly in Gorgan, Northern Iran. Arch Iran Med. 2010;13(1):34-7.
  10. Zheng XY, Song XM, Chen G. Epidemiology of birth defects in high-prevalence areas of China. Zhonghua Liu Xing Bing Xue Za Zhi. 2007;28(1):5-9.
  11. Tan KBL, Tan SH, Tan KH, Yeo GSH. Anencephaly in Singapore: a ten-year series 1993 – 2002. Singapore Med J. 2007;48(1):12-5.
  12. Loncarek K, Mustac E, Frkovic A, Prodan M. Prevalence of anencephaly in the region of Rijeka, Croatia. Eur J Epidemiol. 2001;17(3):241-4.
  13. Nassaralla SM, Nassaralla JJ. High incidence of anencephaly and legal rights. Braz J Med Biol Res. 1996;29(10):1301-6.
  14. Nasri K, Ben Fradj MK, Hamdi T, Aloui M, Ben Jemaa N, Nahdi S, Guesmi R, Masmoudi A, Elmay MV, Marrakchi R, Gaigi SS. Epidemiology of neural tube defect subtypes in Tunisia, 1991-2011. Pathol Res Pract. 2014;210(12):944-52.
  15. Sayed AR, Bourne D, Pattinson R, Nixon J, Henderson B. 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. 2008;82(4):211-6.
  16. De Wals P, Tairou F, van Allen MI, Uh S, Lowry RB, Sibbald B, Evans JA, Van Den Hof MC, Zimmer P, Crowley M et al. Reduction in neural-tube defects after folic acid fortification in Canada. N Engl J Med. 2007;357(2):135-42.
  17. Mocan H, Bozkaya H, Mocan MZ, Furtun EM. Changing incidence of anencephaly in the eastern Black Sea region of Turkey and Chernobyl. Paediatr Perinat Epidemiol. 1990;4(3):264-8.
  18. Berg Bruce. Anencephaly. Encyclopedia of the Neurological Sciences. 2003;157-158.
  19. Zlotogora J. Hereditary disorders among Iranian Jews. Am J Med Genet. 1995;58(1):32-7.
  20. Benjelloun H, Bouaouda H, Sendid M, Oukhouia B, Osstowar K. Anencephaly and prolonged pregnancy. Apropos of the etiology of prolonged pregnancy. J Gynecol Obstet Biol Reprod (Paris). 1983;12(1):65-71.
  21. Agopian A.J, Mark A. Canfield, Richard S. Olney, Philip J. Lupo, Tunu Ramadhani, Laura E. Mitchell, Gary M. Shaw, Cynthia A. Moore, and the National Birth Defects Prevention Study. Spina Bifida Subtypes and Sub-Phenotypes by Maternal Race/Ethnicity in the National Birth Defects Prevention Study. Am J Med Genet A. 2012;158A(1):109-15.
  22. Fornoff JE, Egler T, Shen T. Prevalence of Neural Tube Defects in Illinois 1989-2002. Epidemiological Report Series 04:02. Springfield: Illinois Department of Public Health, 2004.
  23. Whiteman D, Murphy M, Hey K, O’Donnell M, Goldacre M. Reproductive factors, subfertility and risk of neural tube defects: a case-control study based on the Oxford Record Linkage Study Register. Am J Epidemiol. 2000;152(9):823-8.
  24. Hendricks KA, Simpson JS, Larsen RD. Neural tube defects along the Texas-Mexico border, 1993-1995. Am J Epidemiol. 1999;149(12):1119-27.
  25. Machado IN, Martinez SD, Barini R. Anencephaly: Do the Pregnancy and Maternal Characteristics Impact the Pregnancy Outcome?. ISRN Obstet Gynecol. 2012;2012:127490.
  26. Jaquier. M, A. Klein, and E. Boltshauser. Spontaneous pregnancy outcome after prenatal diagnosis of anencephaly. BJOG. 2006;113(8):951-3.
  27. James W. H. The sex ratio in anencephaly. J Med Genet. 1979;16(2):129-33.
  28. Obeidi. N, N. Russell, J. R. Higgins, and K. O’Donoghue. The natural history of anencephaly. Prenat Diagn. 2010;30(4):357-60.
  29. Gaigi SS, Masmoudi A, Mahjoub S, Jabnoun S, Ouni S. Étude foetopathologique de 88 cas de spina bifida létaux. [Fetal pathology study of 88 cases of letal spina bifida]. Tunis Med. 2000;78(12):727-30.
  30. Nasri K, Ben Fradj MK, Aloui M, Ben Jemaa N, Masmoudi A, Elmay MV, Marrakchi R, Gaigi SS. An increase in spina bifida cases in Tunisia, 2008–2011. Pathol Res Pract. 2015;211(5):369-73.
  31. Stillbirth Collaborative Research Network Writing Group. Association between stillbirth and risk factors known at pregnancy confirmation. JAMA. 2011;306(22):2469-79.
  32. Willinger M, Ko CW, Reddy UM. Racial disparities in stillbirth risk across gestation in the United States. Am J Obstet Gynecol. 2009;201(5):469.e1-8.
  33. Landry T. Trisomie 21: Etude de consanguinité et d’apparentement au Saguenay Lac ST-Jean. Mémoire présenté à l'université Laval comme exigence partielle de la maîtrise en médecine expérimentale. Université Du Québec À Chicguïïmi, 1997 Octobre: 92 pages.
  34. Lacombe T. Malformations congénitales à Lubumbashi: fréquence et facteurs favorisants "de janvier 2007 à décembre 2011". Institut numérique; valable en: http://www.institut-numerique.org/malformations-congenitales-a-lubumbashi-frequence-et-facteurs-favorisants-de janvier-2007-a-decembre-2011-506c0f807210c?PHPSESSID=20bb9750544d3c564e09527f4aa37ca9
  35. Aguiar M.J.B, A.Campos, R.A.L.P.Aguiar, A.M.A.Lana, R. L. Magalh˜ aes, L. T. Babeto. Neural tube defects and associated factors in liveborn and stillborn infants. J Pediatr (Rio J). 2003;79(2):129-34.