Parental craniofacial morphology in cleft lip with or without cleft palate as determined by cephalometry: a meta-analysis.
OBJECTIVE
To integrate findings from previous cephalometric studies comparing the craniofacial complex of unaffected parents with cleft lip with or without cleft palate (CL/P) children to controls with no history of the disease.


METHODS
Meta-analysis of case-control cephalometric data.


METHODS
Studies were selected if the unaffected parents of children with CL/P were included and were not combined with parents of children with isolated CP; quantitative data were obtained through cephalometry; the cephalometric variables used were not unique to a study; a case-control design was used; and the means and standard deviations for all variables were reported or could be calculated for both the experimental and the control group.


METHODS
Using raw data obtained from nine studies, mean weighted effect sizes with 95% confidence intervals were calculated for 28 cephalometric variables (mothers and fathers combined) or 18 variables (mothers and fathers separately). Heterogeneity statistics for the effect sizes were also calculated.


RESULTS
In general, unaffected parents of children with CL/P possessed significantly wider interorbital, nasal cavity and upper facial dimensions, narrower cranial vaults, longer cranial bases, longer and more protrusive mandibles, shorter upper faces and longer lower faces compared with controls. Increased width of the nasal cavity was the most robust finding. Significant effect size heterogeneity was observed in roughly half of the variables examined.


CONCLUSIONS
Unaffected parents of children with CL/P are characterized by a suite of consistent, yet subtle, craniofacial differences, which could indicate an underlying genetic liability.
Weinberg
S M
SM
Center for Craniofacial and Dental Genetics, Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA 15219, USA. smwst46@sdmgenetics.pitt.edu
Maher
B S
BS
Marazita
M L
ML
D016022:Case-Control Studies; D002508:Cephalometry; D002971:Cleft Lip / Q000235:genetics* / Q000473:pathology; D002972:Cleft Palate / Q000235:genetics* / Q000473:pathology; D005145:Face / Q000033:anatomy & histology*; D005147:Facial Bones / Q000033:anatomy & histology*; D005220:Fathers; D020022:Genetic Predisposition to Disease; D006801:Humans; D008334:Mandible / Q000033:anatomy & histology; D009035:Mothers; D009296:Nasal Cavity / Q000033:anatomy & histology; D009915:Orbit / Q000033:anatomy & histology; D010290:Parents; D012886:Skull / Q000033:anatomy & histology*; D019291:Skull Base / Q000033:anatomy & histology; D014716:Vertical Dimension