Prevalence of dental anomalies in cleft affected subjects and their family members

Abstract

Introduction: A wide range of dental anomalies are found in populations affected with orofacial clefts. However, an underwhelming body of evidence is available regarding the prevalence of dental anomalies in their unaffected relatives. This multi-center study aims to quantify the difference in prevalence of dental anomalies between subjects with non-syndromic clefts, their unaffected siblings and parents, and controls. Methods: A total of 1,025 subjects were rated, including: 110 subjects with overt orofacial clefting, 316 unaffected relatives, 599 controls. Intraoral photographs were utilized to rate dental anomalies. Case-control differences were evaluated via chi squared statistics and t-tests. Results: Compared with matched controls, the case proband group had more subjects with agenesis, displacements and mamelons, yet differences in prevalence for these anomalies varied by dentition and arch. Analyses by cleft type revealed that agenesis in the maxillary arch was significantly more prevalent in the CLP group compared to other cleft types or controls. After correction for multiple testing, dental anomalies in unaffected parents and siblings were not significantly different from matched controls except for incisal fissures that were more prevalent in controls than unaffected parents. However, several trends were detected. Discussion: Generally our findings are consistent with previous work that found an elevated rate of dental anomalies in the cleft-affected population. However, based on our study a claim cannot be made that unaffected family members are at significantly higher risk for dental anomalies, suggesting the higher rate of anomalies in cleft-affected subjects is not entirely due to genetics. Conclusions: These findings suggest that families with history of clefting do not carry a genetic risk for dental anomalies greater than the general population. Higher rates of dental anomalies in the case probands is most likely due to the physical cleft and corrective surgical interventions.