European Journal of Epidemiology

, Volume 22, Issue 6, pp 389–395

Epidemiology of cleft palate alone and cleft palate with accompanying defects

  • Emily W. Harville
  • Allen J. Wilcox
  • Rolv Terje Lie
  • Frank Åbyholm
  • Hallvard Vindenes
Birth Defects

Abstract

The epidemiology of cleft palate with multiple defects is often thought to be different from that of cleft palate alone, but there are few empirical data on this question. We explored this in a population-based data set created by combining data from two sources: the 1.8 million live births recorded from 1967 to 1998 in the Norwegian Birth Registry, and the two Norwegian surgical centers that repair cleft palate. Accompanying defects were identified from either source. Stratified analysis and logistic regression were used to assess relative risks by covariates. Of 1,431 babies with cleft palate, 31 % had another birth defect recorded by one or both sources. Prevalence of isolated cleft palate was steady over time, while cleft palate with other defects increased substantially. Girls had a higher risk of isolated cleft palate (relative risk 1.4; 95% confidence interval, 1.2–1.6) but not of cleft palate accompanied by other defects (1.1; 0.88–1.3). Older parents and parents who were first cousins had no increased risk of isolated cleft palate, but were twice as likely as others to have a baby with cleft palate accompanied by other defects. Risk factors differ between cases of cleft palate with and without accompanying defects.

Keywords

Birth defects; Cleft palate; Multiple abnormalities; Sex distribution 

Abbreviations

CP

Cleft palate

ICD

International classification of diseases

RR

Relative risk

OR

Odds ratio

References

  1. 1.
    Christensen K. The 20th century Danish facial cleft population––epidemiological and genetic-epidemiological studies. Cleft Palate Craniofac J 1999;36:96–104PubMedCrossRefGoogle Scholar
  2. 2.
    Jakobsen LP, Molsted K, Christensen K. Occurrence of cleft lip and palate in the Faroe Islands and Greenland from 1950 to 1999. Cleft Palate Craniofac J 2003;40:426–30PubMedCrossRefGoogle Scholar
  3. 3.
    Kallen B, Harris J, Robert E. The epidemiology of orofacial clefts. 2. Associated malformations. J Craniofac Genet Dev Biol 1996;16:242–8PubMedGoogle Scholar
  4. 4.
    Magdalenic-Mestrovic M, Bagatin M. An epidemiological study of orofacial clefts in Croatia 1988–1998. J Craniomaxillofac Surg 2005;33:85–90PubMedGoogle Scholar
  5. 5.
    Milerad J, Larson O, Ph DD, Hagberg C, Ideberg M. Associated malformations in infants with cleft lip and palate: a prospective, population-based study. Pediatrics 1997;100:180–6PubMedCrossRefGoogle Scholar
  6. 6.
    Natsume N, Niimi T, Furukawa H, Kawai T, Ogi N, Suzuki Y, Kawai T. Survey of congenital anomalies associated with cleft lip and/or palate in 701,181 Japanese people. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;91:157–61PubMedCrossRefGoogle Scholar
  7. 7.
    Forrester MB, Merz RD. Descriptive epidemiology of oral clefts in a multiethnic population, Hawaii, 1986–2000. Cleft Palate Craniofac J 2004;41:622–8PubMedCrossRefGoogle Scholar
  8. 8.
    Shaw GM, Carmichael SL, Yang W, Harris JA, Lammer EJ. Congenital malformations in births with orofacial clefts among 3.6 million California births, 1983–1997. Am J Med Genet A 2004;125:250–6PubMedCrossRefGoogle Scholar
  9. 9.
    Stoll C, Alembik Y, Dott B, Roth MP. Associated malformations in cases with oral clefts. Cleft Palate Craniofac J 2000;37:41–7PubMedCrossRefGoogle Scholar
  10. 10.
    Tolarova MM, Cervenka J. Classification and birth prevalence of orofacial clefts. Am J Med Genet 1998;75:126–37PubMedCrossRefGoogle Scholar
  11. 11.
    Mitchell LE, Beaty TH, Lidral AC, Munger RG, Murray JC, Saal HM, Wyszynski DF. International Consortium for Oral Clefts G Guidelines for the design and analysis of studies on non-syndromic cleft lip and cleft palate in humans: summary report from a Workshop of the International Consortium for Oral Clefts Genetics. Cleft Palate Craniofac J 2002;39:93–100PubMedCrossRefGoogle Scholar
  12. 12.
    Wyszynski DF, Sarkozi A, Czeizel AE. Oral clefts with associated anomalies: methodological issues. Cleft Palate Craniofac J 2006;43:1–6PubMedCrossRefGoogle Scholar
  13. 13.
    Das SK, Runnels RS Jr, Smith JC, Cohly HH. Epidemiology of cleft lip and cleft palate in Mississippi. South Med J 1995;88:437–42PubMedGoogle Scholar
  14. 14.
    James WH. Are oral clefts a consequence of maternal hormone imbalance? evidence from the sex ratios of sibs of probands. Teratology 2000;62:342–5PubMedCrossRefGoogle Scholar
  15. 15.
    Lowry RB, Thunem NY, Uh SH. Birth prevalence of cleft lip and palate in British Columbia between 1952 and 1986: stability of rates. CMAJ 1989;140:1167–70PubMedGoogle Scholar
  16. 16.
    Menegotto BG, Salzano FM. Epidemiology of oral clefts in a large South American sample. Cleft Palate Craniofac J 1991;28:373–6 (discussion 376–377)PubMedCrossRefGoogle Scholar
  17. 17.
    Christensen K. Methodological issues in epidemiological studies of oral clefts. In: Wyszynski DF, (editor). Cleft lip & Palate. New York: Oxford University Press, 2002. 101–7Google Scholar
  18. 18.
    Stanier P, Moore GE. Genetics of cleft lip and palate: syndromic genes contribute to the incidence of non-syndromic clefts. Hum Mol Genet 2004;13(Spec No 1):R73–81PubMedCrossRefGoogle Scholar
  19. 19.
    Vieira AR, Orioli IM. Birth order and oral clefts: a meta analysis. Teratology 2002;66:209–16PubMedCrossRefGoogle Scholar
  20. 20.
    Sayetta RB, Weinrich MC, Coston GN. Incidence and prevalence of cleft lip and palate: what we think we know. Cleft Palate Journal 1989;26:242–7 (discussion 247–248)PubMedGoogle Scholar
  21. 21.
    Bixler D. Commentary. Cleft Palate Craniofac J 1991;28:376–7Google Scholar
  22. 22.
    Lie RT, Heuch I, Irgens LM. Maximum likelihood estimation of the proportion of congenital malformations using double registration systems. Biometrics 1994;50:433–44PubMedCrossRefGoogle Scholar
  23. 23.
    Czeizel A, Tusnadi G. An epidemiologic study of cleft lip with or without cleft palate and posterior cleft palate in Hungary. Human Hered 1971;21:17–38CrossRefGoogle Scholar
  24. 24.
    DeRoo LA, Gaudino JA, Edmonds LD. Orofacial cleft malformations: associations with maternal and infant characteristics in Washington State. Birth Defects Res A Clin Mol Teratol 2003;67:637–42PubMedCrossRefGoogle Scholar
  25. 25.
    Wyszynski DF, Sarkozi A, Vargha P, Czeizel AE. Birth weight and gestational age of newborns with cleft lip with or without cleft palate and with isolated cleft palate. J Clin Pediatr Dent 2003;27:185–90. PubMedGoogle Scholar
  26. 26.
    Christensen K, Holm NV, Olsen J, Kock K, Fogh-Andersen P. Selection bias in genetic-epidemiological studies of cleft lip and palate. Am J Hum Genet 1992;51:654–9. PubMedGoogle Scholar

Copyright information

© Springer Science+Business Media B.V. 2007

Authors and Affiliations

  • Emily W. Harville
    • 1
    • 7
  • Allen J. Wilcox
    • 2
  • Rolv Terje Lie
    • 3
    • 4
  • Frank Åbyholm
    • 5
  • Hallvard Vindenes
    • 6
  1. 1.Department of EpidemiologyUniversity of North Carolina-Chapel HillChapel HillUSA
  2. 2.Epidemiology BranchNational Institute of Environmental Health SciencesDurhamUSA
  3. 3.Department of Public Health and Primary Health CareUniversity of BergenBergenNorway
  4. 4.Medical Birth Registry of Norway, Norwegian Institute of Public HealthBergenNorway
  5. 5.Department of Plastic SurgeryThe National HospitalOsloNorway
  6. 6.Department of Plastic SurgeryHaukeland University HospitalBergenNorway
  7. 7.Department of EpidemiologyTulane University School of Public Health and Tropical MedicineNew OrleansUSA

Personalised recommendations