To report the epidemiological characteristics of isolated cleft lip, cleft palate or both (CL ± P and CP) using population-based data in Victoria, Australia.
Design
Descriptive study of a cohort of children born between 1983 and 2000 notified to the Victorian Birth Defects Register by multiple ascertainment sources.
Participants
Data were collected on patients identified with CL ± P and CP without associated defects classified as live births, stillbirths, neonatal deaths, and terminated pregnancies < 20 weeks’ gestation following prenatal identification. Information was collected on sex, plurality, maternal age, and country of birth.
Results
The overall prevalence (per 10,000 pregnancies) of CL ± P was 7.8 (95% confidence interval [CI] = 7.30, 8.33; cleft lip [CL] 3.3; 95% CI = 2.97, 3.65, CL+P 4.5; 95% CI = 4.13, 4.91) and cleft palate (CP), 4.3 (95% CI = 3.89, 4.66). The prevalence of CL + P was higher among stillbirths, neonatal deaths, and terminated pregnancies than CL (without CP) and CP. Boys were at greater risk than girls for CL ± P and girls at greater risk than boys for CP. Regardless of cleft type, there was a nonsignificant excess of clefts among singleton births than multiple births and no related effects of maternal age or country of birth.
Conclusions
The prevalence of isolated CL ± P and CP in Victoria parallels other population-based studies of the same conditions. Inclusion of stillbirths, neonatal deaths, and terminations had little impact on rates. The effect of sex and plurality on cleft type is consistent with the literature, but the effects of maternal age and country of birth remain equivocal. Further studies focusing on certain ethnic groups are warranted to explain the higher rates observed.
AbyholmF.E.Cleft lip and palate in a Norwegian population. II. A numerical study of 1555 CLP patients admitted for surgical treatment 1954-75.Scand J Plast Reconstr Surg.1978; 12: 35–43.
2.
AmideiR.L.HammanR.F.KassebaumD.K.MarshallJ.A.Birth prevalence of cleft lip and palate in Colorado by sex distribution, seasonality, race/ethnicity, and geographic variation.Spec Care Dent.1994; 14: 233–240.
3.
AntoszewskiB.Kruk-JerominJ.Epidemiology of CL and palate in Lodz, Poland, in the years 1981-1995.Acta Chir Plast.1997; 39: 109–112.
4.
Australian Bureau of Statistics.2001 Census of Population and Housing, State of Victoria, Commonwealth of Australia. Available at: http://www.abs.gov.au. Accessed June 1, 2002.
5.
BairdP.A.SadovnickA.D.YeeI.M.L.Maternal age and oral cleft malformations: data from a population-based series of 576,815 consecutive livebirths.Teratology.1994; 49: 448–451.
6.
BellisT.H.WohlgenmuthB.The incidence of cleft lip and palate deformities in the South-East of Scotland. (1971-1990).Br J Orthod.1999; 26: 121–125.
7.
BonaitiC.BriardM.L.FeingoldJ.PavyB.PsaumeJ.Migne-TufferaudG.KaplanJ.An epidemiological and genetic study of facial clefting in France. I. Epidemiology and frequency study of facial clefting in relatives.J Med Genet.1982; 19: 8–15.
8.
BorkarA.S.MathurA.K.MahaluxmivalaS.Epidemiology of facial clefts in the central province of Saudi Arabia.Brit J Plast Surg.1993; 46: 673–675.
9.
British Paediatric Association. British Paediatric Association Classification of Diseases.London: BPA;1979: 1–220.
10.
BurdiA.R.FeingoldM.LarssonK.S.LeckI.ZimmermanE.F.FraserF.C.Etiology and pathogenesis of congenital cleft lip and cleft palate, an NIDR state of the art report.Teratology.1972; 6: 255–270.
11.
ChristenssenK.The 20th century Danish facial cleft population-epidemiological and genetic-epidemiological studies.Cleft Palate Craniofac J.1999; 36: 96–104.
12.
ChuangsuwanichA.AojanepongC.MuangsombutS.TongpiewP.Epidemiology of cleft lip and palate in Thailand.Ann Plast Surg.1998; 41: 7–10.
13.
ClementiM.TenconiR.BianchiF.StollC., EUROSCAN study group. Evaluation of prenatal diagnosis of cleft lip with or without cleft palate and cleft palate by ultrasound: experience from 20 European registries.Prenat Diagn.2000; 20: 870–875.
14.
Consultative Council on Obstetric and Paediatric Mortality and Morbidity. Annual Report for the Year 1999, incorporating the 38th Survey of Perinatal Deaths in Victoria. Melbourne; 2001.
15.
CooperM.E.StoneR.A.LiuY-EHuD-NMelnickM.MarzazitaM.L.Descriptive epidemiology of isolated cleft lip with or without cleft palate in Shanghai, China, from 1980 to 1989.Cleft Palate Craniofac J.2000; 37: 274–280.
16.
CroenL.A.ShawG.M.WassermanC.R.TolarovaM.M.Racial and ethnic variations in the prevalence of orofacial clefts in California, 1983-1992.Am J Med Genet.1998; 79: 42–47.
17.
CzeizelA.TusnadiG.A family study of cleft lip with or without cleft palate and posterior cleft palate in Hungary.Hum Hered.1972; 22: 405–416.
18.
DeanA.G.ArnerT.G.SangamS.SunkiG.G.FriedmanR.LantingaM.ZubietaJ.C.SullivanK.M.SmithD.C.Epi Info 2000, a database and statistics program for public health professionals for use on Windows 95, 98, NT, and 2000 computers.Atlanta, GA: Center for Disease Control and Prevention; 2000.
19.
DerijckeA.EerensA.CarelsC.The incidence of oral clefts: a review.Br J Oral Maxillofac Surg.1996; 34: 488–494.
20.
EmanuelI.CulverB.H.EricksonJ.D.GuthrieB.SchuldbergD.The further epidemiological differentiation of cleft lip and palate: a population study of clefts in King County, Washington, 1956-1965.Teratology.1973; 7: 271–281.
21.
EmanuelI.HuangS.W.GutmanL.T.YuF.C.LinC.C.The incidence of congenital malformations in a Chinese population: the Taipei collaborative study.Teratology.1972; 5: 159–170.
22.
EUROCAT Report 7. 15 Years of Surveillance of Congenital Anomalies in Europe 1980-1994.Brussels, Belgium: Scientific Institute of Public Health-Louis Pasteur; 1997: 1–252.
23.
FitzPatrickD.R.RaineP.A.BoormanJ.G.Facial clefts in the west of Scotland in the period 1980-1984: epidemiology and genetic diagnoses.J Med Genet.1994; 31: 126–129.
24.
Fogh-AndersonP.Inheritance of Harelip and Cleft Palate.Copenhagen: Arnold Busck;1942.
25.
GreeneJ.C.VermillonJ.R.HayS.GibbensS.F.KerschbaumS.Epidemiologic study of cleft lip and palate in four states.J Am Dent Assoc.1964; 68: 387–404.
26.
GreggT.BoydD.RichardsonA.The incidence of cleft lip and palate in Northern Ireland from 1980-1990.Br J Orthod.1994; 21: 387–392.
27.
HagbergC.LarsonO.MileradJ.Incidence of cleft lip and palate and risks of additional malformations.Cleft Palate Craniofac J.1998; 35: 40–45.
28.
HayS.Incidence of clefts and parental age.Cleft Palate J.1967; 4: 205–213.
29.
HenrikssonT.G.Cleft Lip and Palate in Sweden. A Genetic and Clinical Investigation.Uppsala, Sweden: The Institute for Medical Genetics, University of Uppsala;1971.
30.
HewsonA.R.McNamaraC.M.CL and/or palate in the west of Ireland, 1980—1996.Spec Care Dent.2000; 20: 143–146.
31.
International Classification of Diseases.9th ed.Geneva: World Health Organization; 1977.
32.
JensenB.L.KreiborgS.DahlE.Fogh-AndersonP.Cleft lip and palate in Denmark, 1976—1981: epidemiology, variability, and early somatic development.Cleft Palate J.1988; 25: 258–269.
33.
KilkennyM.RileyM.LumleyJ.Follow-up validation study of the Victorian congenital malformations register.J Paediatr Child Health.1995; 31: 323–325.
34.
KozeljV.Epidemiology of orofacial clefts in Slovenia, 1973—1993: comparison of the incidence in six European countries.J Craniomaxillofac Surg.1996; 24: 378–382.
35.
LumleyJ.PalmaS.FischerM.RobertsonH.The tip of the iceberg: a validation study of the Victorian congenital malformations/Birth Defects Register.Aust Paediat J.1988; 24: 244–246.
36.
MenegottoB.G.SalzanoF.M.Epidemiology of oral clefts in a large South American sample.Cleft Palate Craniofac J.1991; 28: 373–376.
37.
MurrayJ.C.Daack-HirschS.BuetowK.H.MungerR.EspinaL.PaglinawanN.VillanuevaE.RaryJ.MageeK.MageeW.Clinical and epidemiologic studies of cleft lip and palate in the Philippines.Cleft Palate Craniofac J.1997; 34: 7–10.
38.
NatsumeN.KawaiT.KohamaG.TeshimaT.KochiS.OhashiY.EnomotoS.IshiiM.NakanoY.MatsuyaT.KogoM.YoshimuraY.OhishiM.NakamuraN.KatsukiT.GotoM.ShimizuM.YanagisawaS.MimuraT.SunakawaH.Incidence of cleft lip or palate in 303738 Japanese babies born between 1994 and 1995.Br J Oral Maxillofac Surg.2000; 38: 605–607.
39.
NorusisM.J.SPSS for Windows: Base System Users’ Guide. Release 11.0. Chicago: SPSS;2002.
40.
OkaS.W.Commentary. Incidence and prevalence of cleft lip and palate: what we think we know.Cleft Palate J.1989; 26: 247–248.
41.
OkaS.W.Epidemiology and genetics of clefting: with implications for etiology. In: CooperH.K.HardingR.L.KrogmanW.M.MazaheriM.MillardR.T., eds. Cleft Palate and Cleft Lip: A Team Approach to Cleft Management and Rehabilitation of the Patient.Philadelphia: WB Saunders;1979: 108–143.
42.
OwensJ.R.JonesJ.W.HarrisF.Epidemiology of facial clefting.Arch Dis Child.1985; 60: 521–524.
43.
PeterkaM.PeterkovaR.TvrdekM.KuderovaJ.LiokovskyZ.Significant differences in the incidence of orofacial clefts in fifty-two Czech districts between 1983 and 1997.Acta Chir Plast.2000; 42: 124–129.
44.
RajabianM.H.SherkatM.An epidemiologic study of oral clefts in Iran: analysis of 1,669 cases.Cleft Palate Craniofac J.2000; 37: 191–196.
45.
RileyM.HallidayJ.Birth Defects in Victoria 1983-1998.Melbourne: Perinatal Data Collection Unit, Victorian Government Department of Human Services; 2000.
46.
RileyM.M.HallidayJ.L.LumleyJ.M.Congenital malformations in Victoria, Australia, 1983-95: an overview of infant characteristics.J Paediartr Child Health.1998; 34: 233–240.
47.
RobertE.KallenB.HarrisJ.The epidemiology of orofacial clefts 1. Some general epidemiological characteristics.J Craniofac Genet Dev Biol.1996; 16: 234–241.
48.
SayettaR.B.WeinrichM.C.CostonG.N.Incidence and prevalence of cleft lip and palate: what we think we know.Cleft Palate J.1989; 26: 243–247.
49.
ShawG.M.CroenL.A.CurryC.J.Isolated oral cleft malformations: associations with maternal and infant characteristics in a California population.Teratology.1991; 43: 225–228.
50.
StollC.AlembikY.DottB.RothM.P.Associated malformations in cases with oral clefts.Cleft Palate Craniofac J.2000; 37: 41–47.
51.
StollC.AlembikY.DottB.RothM.P.Epidemiological and genetic study in 207 cases of oral clefts in Alsace, northeastern France.J Med Genet.1991; 28: 325–329.
52.
TanK.L.Incidence and epidemiology of cleft lip/palate in Singapore.Ann Acad Med.1988; 17: 311–314.
53.
TolarovaM.M.Genetic findings in cleft lip and palate in Czech population. In: BardachJ.MorrisH.L., eds. Multidisciplinary Management of Cleft Lip and Palate.New York: WB Saunders;1990: 113–121.
54.
TolarovaM.M.CervenkaJ.Classification and birth prevalence of orofacial clefts.Am J Med Genet.1998; 75: 126–137.
55.
VanderasA.P.Incidence of cleft lip, cleft palate, and cleft lip and palate among races: a review.Cleft Palate J.1987; 24: 216–225.
56.
WarkenyJ.Congenital Malformations.Chicago: YearBook Medical Publications;1971.
57.
WomersleyJ.StoneD.H.Epidemiology of facial clefts.Arch Dis Child.1987; 62: 717–720.
58.
World Atlas of Birth Defects.1st ed. International Centre for Birth Defects of the International Clearinghouse for Birth Defects Monitoring Systems, Human Genetics Programme, World Health Organization; 1998.
59.
YiN.N.YeowV.K.LeeS.T.Epidemiology of cleft lip and palate in Singapore—a 10-year hospital-based study.Ann Acad Med Singapore.1999; 28: 655–659.
