Craniofacial Growth and Hypodontia in Individuals Born with Van der Woude Syndrome

ObjectiveTo study craniofacial growth and hypodontia in individuals born with Van der Woude syndrome (VWS) and compare with matched controls with and without a cleft.DesignA multicenter retrospective case-control study.SettingIndividuals from three craniofacial centers in Sweden and from a historical archive of Swedish healthy children were included.PatientsSixty individuals, with VWS (n = 20), non-syndromic cleft lip and/or palate (NSCL/P) (n = 20), and healthy controls (CTR) (n = 20), matched for age, gender and cleft type.InterventionsTwo lateral cephalograms per individual, taken at 10 and 16 years of age, were analyzed. Orthopantomograms were used to evaluate hypodontia.Main Outcome MeasuresCraniofacial growth followed longitudinally, with the primary variables SNA, SNB, ANB, NL/ML, and maxillary length. Prevalence of hypodontia was also recorded.ResultsMaxillary length increased 0.3 mm in the VWS group during puberty, compared to 4 mm in the NSCL/P group and 5.5 mm in the CTR group. Both cleft groups had a retruded maxilla at 16 years of age. The change in vertical jaw relation from 10 to 16 years showed a statistically significant difference between the VWS and the CTR group, with a slightly more posteriorly inclined mandible in the VWS group at 16 years. A statistically significant difference in hypodontia is seen between the cleft groups. In the VWS group, 90% had hypodontia compared to 60% in the NSCL/P group.ConclusionIndividuals born with VWS have reduced maxillary growth during puberty and a higher prevalence of hypodontia compared to matched controls in the studied population.