To evaluate the parental craniofacial morphology in Chinese patients with sporadic nonsyndromic cleft lip with or without palate.
Methods:
A total of 98 parental pairs of nonsyndromic unilateral incomplete cleft lip children, 207 parental pairs of nonsyndromic complete cleft lip and palate children, and 206 normal persons from Sichuan University were involved in this study. A conventional cephalometric analysis was used to measure angles, linear distances, and their ratios. Two-sample Student's t tests and a multivariate discriminant analysis were applied to the data.
Results:
Data indicate that the unaffected parents of nonsyndromic cleft lip children had on average significantly more acute cranial base angle (Angle N-S-Ba) and larger nasal width (NC-NC′) (p < .01). The healthy parents of nonsyndromic cleft lip and palate children consistently displayed a more acute cranial base angle (Angle N-S-Ba), shorter palatal length (A- PNS) and maxillary length (PNS-ANS), a more obtuse gonial angle (Angle Me-Go-Ar), and a larger y-axis length (S-Gn) and nasal width (NC-NC′) (p < .01).
Conclusions:
All these results indicate that the healthy parents of patients with nonsyndromic cleft lip with or without palate show distinct characteristics in craniofacial morphology. These parental craniofacial features are more obvious in patients with cleft lip with palate than those with cleft lip only. In general, the characteristics seem to be more distinct in the fathers than in the mothers of cleft patients.
AlEmranS.E., FataniE., HassanainJ.E.. Craniofacial variability in parents of children with cleft lip and palate.J Clin Pediatr Dent.1999; 23: 337–341.
2.
BeiraghiS., ForoudT., DiouhyS., BixlerD., ConneallyP.M., Delozier- BlanchetD., HodesM.E.. Possible localization of a major gene for cleft lip and palate to 4q.Clin Genet.1994; 46: 255–256.
3.
BlancoR., CifuentesL., MaldonadoM.J., RameauX., MunozM.A.. Cleft lip and cleft palate: cephalometric characteristics of affected individuals, their relatives and a control population.Rev MedChil.1992; 120: 13–19.
4.
CoccaroP.J., D'AmicoR., ChavoorA.. Craniofacial morphology of parents with and without cleft lip and palate children.Cleft Palate Craniofac J.1972; 9: 28–38.
FraserF.C., PashayanH.. Relation of face shape to susceptibility to congenital cleft lip.J Med Genet.1970a; 7: 112–117.
7.
FraserF.C., PashayanH.. The genetics of cleft lip and palate.Am J Hum Genet.1970b; 22: 336–352.
8.
GrummonsD.C., KappeyneM.A.. A frontal asymmetry analysis.J Clin Orthod.1987; 21: 448–465.
9.
KurisuK., NiswanderJ.D., JohnstonM.C., MazaheriM.. Facial morphology as an indicator of genetic predisposition to cleft lip and palate.Am J Hum Genet.1974; 26: 702–714.
10.
MaulinaI., UrtaneI., JakobsoneG.. The craniofacial morphology of the parents of children with cleft lip and/or palate: a review of cephalometric studies.Stomatologija.2006; 8: 16–20.
11.
McIntyreG.T., MosseyP.A.. Posteroanterior cephalometric analysis of the parental craniofacial morphology in orofacial clefting.Cleft Palate Craniofac J.2003; 40: 416–425.
12.
McIntyreG.T., MosseyP.A.. The craniofacial morphology of the parents of children with orofacial clefting: a systematic review of cephalometric studies.J Orthod.2002; 29: 23–29.
13.
MitchellL.E., BeatyT.H., LidralA.C., MungerR.G., MurrayJ.C., SaalH.M., WyszynskiD.F.. Guidelines for the design and analysis of studies on nonsyndromic cleft lip and cleft palate in humans: summary report from a Workshop of the International Consortium for Oral Cleft Genetics.Cleft Palate Craniofac J.2002; 39: 93–100.
14.
MoorreesC.F.A., KeanM.R.. Natural head position: a basic consideration in the interpretation of cephalometric radiographs.Am J Phys Anthropol.1958; 16: 213–234.
15.
MosseyP.A., ArngrimssonR., McCollJ., VintinerG.M., ConnerJ.M.. Prediction of liability to orofacial clefting using genetic and craniofacial data from parents.J Med Genet.1998a; 35: 371–378.
16.
MosseyP.A., LittleJ.. Epidemiology of oral clefts: an international perspective. In: WyszynskiD.F., ed. Cleft Lip and Palate: From Origin to Treatment.Oxford: Oxford University Press;2002: 127–158.
17.
MosseyP.A., McCollJ., O'HaraM.. Cephalometric features in the parents of children with orofacial clefting.Br J Oral Maxillofac Surg.1998b; 36: 202–212.
18.
MosseyP.A., McCollJ., StirrupsD.R.. Differentiation between cleft lip with or without cleft palate and isolated cleft palate using parental cephalometric parameters.Cleft Palate Craniofac J.1997; 34: 27–35.
19.
NakasimaA., IchinoseM.. Characteristics of craniofacial structures of parents of children with cleft lip and/or palate.Am J Orthod.1983; 84: 140–146.
20.
NakasimaA., IchinoseM.. Size of the cranium in parents and their children with cleft lip.Cleft Palate Craniofac J.1984; 21: 193–203.
21.
PerkiomakiM.R., YoonY.J., TallentsR.H., BarillasI., Herrera-GuidoR., MossM.E., FongC.T.. Kyrkanides S Association of distinct craniofacial features in nonsyndromic cleft lip and palate family members.Cleft Palate Craniofac J.2003; 40: 397–402.
22.
ProchazkovaJ., TolarovaM.. Craniofacial morphological features in parents of children with isolated cleft palate.Acta Chir Plast.1986; 28: 194–204.
23.
ProchazkovaJ., VinsovaJ.. Craniofacial morphology as a marker of predisposition to isolated cleft palate.J Craniofac Genet Dev Biol.1995; 15: 162–168.
24.
RaghavanR., SidhuS.S., KharbandaO.P.. Crainofacial pattern of parents of children having cleft lip and/or cleft palate anomaly.Angle Orthod.1994; 64: 137–144.
25.
SatoT.. Craniofacial morphology of parents with cleft lip and palate children.Shikwa Gakuho.1989; 89: 1479–1506.
26.
ShibasakiY., OhtsukaS., HattoriM., NukatsukaS.. A cephalometric study on craniofacial morphology of parents of children with cleft lip and palate.J Jpn Cleft Palate Assoc.1978; 3: 31–43.
27.
SolowB., SandhamA.. Cranio-cervical posture: a factor in the development and function of the dentofacial structures.Eur J Orthod.2002; 24: 447–456.
28.
SolowB., TallgrenA.. Natural head position in standing subjects.Acta Odont Scand.1971; 29: 591–607.
29.
SuzukiA., TakenoshitaY., HondaY., MatsuuraC.. Dentocraniofacial morphology in parents of children with cleft lip and/or palate.Cleft Palate Craniofac J.1999; 36: 131–138.
30.
TraslerD.G.. Pathogenesis of cleft lip and its relation to embryonic face shape in A-J and C57BL mice.Teratology.1968; 1: 33–49.
31.
WardR.E., BixlerD., JamisonP.. Cephalometric evidence for a dominantly inherited predisposition to cleft lip-cleft palate in a single large kindred.Am J Med Genet.1994; 50: 57–63.
32.
WardR.E., MooreE.S., HartsfieldJ.K.. Morphometric characteristics of subjects with oral facial clefts and their relatives. In: WyszynskiD.F., ed. Cleft Lip and Palate: From Origin to Treatment.Oxford: Oxford University Press;2002: 66–86.
33.
WeinbergS.M., NeiswangerK., MartineR.A., MooneyM.P., KaneA.A., WengerS.L., LooseJ., DeleyiannisF., MaL., DeSalamancaJ.E.. The Pittsburgh oral-facial cleft study: expanding the cleft phenotype. Background and justification.Cleft Palate Craniofac J.2006; 43: 7–20.
34.
YoonY.J., PerkiomakiM.R., TallentsR.H., BarillasI., Herrera-GuidoR., FongC.T., KyrkanidesS.. Association of nasomaxillary asymmetry in children with unilateral cleft lip and palate and their parents.Cleft Palate Craniofac J.2003; 40: 493–497.
35.
YoonY.J., PerkiomakiM.R., TallentsR.H., BarillasI., Herrera-GuidoR., FongC.T., KyrkanidesS.. Transverse craniofacial features and their genetic predisposition in families with nonsyndromic unilateral cleft lip and palate.Cleft Palate Craniofac J.2004; 41: 256–261.
36.
ZuccheroT.M., CooperM.E., MaherB.S., Daack-HirschS., NepomucenoB., RibeiroL., CaprauD., ChristensenK., SuzukiY., MachidaJ.. Interferon regulatory factor 6 (IRF6) gene variants and the risk of isolated cleft lip or palate.JV Engl J Med.2004; 19: 769–780.
