Defining Subphenotypes for Oral Clefts Based on Dental Development

Letra, Ariadne; Menezes, Renato; Granjeiro, José Mauro; Vieira, Alexandre R.

doi:10.1177/154405910708601013

Cited by 167 publications

(292 citation statements)

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“…In that context, we have shown that dental anomalies outside the cleft area could be used as additional features for the generation of more sophisticated cleft subphenotypes [Letra et al, 2007].…”

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confidence: 99%

Studies of genes in the FGF signaling pathway and oral clefts with or without dental anomalies

Menezes

Letra

Ruff

et al. 2008

American J of Med Genetics Pt A

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confidence: 99%

Studies of genes in the FGF signaling pathway and oral clefts with or without dental anomalies

Menezes

Letra

Ruff

et al. 2008

American J of Med Genetics Pt A

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“…found that dental anomalies increase with cleft severity (Stahl et al 2006;Letra et al 2007;Aizenbud et al 2011). Mutations in genes influencing both palatogenesis and dental development could also partly account for the greater prevalence of dental anomalies in individuals with clefts.…”

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confidence: 99%

Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting

et al. 2015

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Children with oral clefts show a wide range of dental anomalies, adding complexity to understanding the phenotypic spectrum of orofacial clefting. The evidence is mixed, however, on whether the prevalence of dental anomalies is elevated in unaffected relatives and is mostly based on small samples. In the largest international cohort to date of children with nonsyndromic clefts, their relatives, and controls, this study characterizes the spectrum of cleft-related dental anomalies and evaluates whether families with clefting have a significantly higher risk for such anomalies compared with the general population. A total of 3,811 individuals were included: 660 cases with clefts, 1,922 unaffected relatives, and 1,229 controls. Dental anomalies were identified from in-person dental exams or intraoral photographs, and case-control differences were tested using χ2 statistics. Cases had higher rates of dental anomalies in the maxillary arch than did controls for primary (21% vs. 4%, P = 3 × 10−8) and permanent dentitions (51% vs. 8%, P = 4 × 10−62) but not in the mandible. Dental anomalies were more prevalent in cleft lip with cleft palate than other cleft types. More anomalies were seen in the ipsilateral side of the cleft. Agenesis and tooth displacements were the most common dental anomalies found in case probands for primary and permanent dentitions. Compared with controls, unaffected siblings (10% vs. 2%, P = 0.003) and parents (13% vs. 7%, P = 0.001) showed a trend for increased anomalies of the maxillary permanent dentition. Yet, these differences were nonsignificant after multiple-testing correction, suggesting genetic heterogeneity in some families carrying susceptibility to both overt clefts and dental anomalies. Collectively, the findings suggest that most affected families do not have higher genetic risk for dental anomalies than the general population and that the higher prevalence of anomalies in cases is primarily a physical consequence of the cleft and surgical interventions.

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“…We proposed to use dental development to subphenotype clefts. Our preliminary analysis suggests dental anomalies are preferentially associated with clefts in some families [Letra et al, 2007]. In order to extend these earlier studies, we proposed to revisit the subset of the initially genotyped families with two or more siblings affected by CL/P and perform a dental examination to broader the phenotypic description of the families.…”

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confidence: 99%