Parents of Children with Non-syndromic Orofacial Clefting Show Altered Palate Shape.
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Date
2020-10-28
Authors
El Sergani, A.M.
Brandebura, S.
Padilla, C.
Butali, A.
Adeyemo, W.L.
Valencia-Ramírez, C.
Muñeton, C.P.R.
Moreno, L.M.
Buxo, C.J
Long, R.E.
Journal Title
Journal ISSN
Volume Title
Publisher
Sage Publishing
Abstract
Objective: The unaffected relatives of individuals with nonsyndromic orofacial clefts have been shown to exhibit subtle craniofacial differences compared with the general population. Here, we investigate whether these morphological differences extend to the shape of the palate.
Design: We conducted a geometric morphometric analysis to compare palate shape in the clinically unaffected parents of children with nonsyndromic cleft lip with or without cleft palate and adult controls of European, Asian, and African ancestry. We conducted pairwise group comparisons using canonical variates analysis, and then confirmed and characterized findings of shape differences using Euclidean distance matrix analysis.
Results: Significant differences in palate shape were detected in unaffected mothers (but not fathers) compared to demographically matched controls. The differences in shape were ancestry-specific; mothers of Asian-derived and African-derived ancestry showed wider and shorter palates with higher posterior palatal vaults, while mothers of European-derived ancestry showed narrower palates with higher anterior palatal vaults.
Conclusions: Our findings suggest that altered palate shape is a subclinical phenotypic feature, which may be indicative of elevated orofacial cleft risk. The risk phenotype varied by sex and ancestry, suggesting possible etiologic heterogeneity among demographic groups. Understanding the genetic basis of these informative palate shape traits may reveal new genes and pathways relevant to nonsyndromic orofacial clefting.
Keywords: cleft lip and palate; craniofacial biology; craniofacial morphology; geometric morphometrics; palatal morphology.
Description
Scholarly articles
Keywords
Cleft palate , Palatal morphology , Craniofacial , Nonsyndromic orofacial clefts , Research Subject Categories::ODONTOLOGY
Citation
El-Sergani AM, Brandebura S, Padilla C, Butali A, Adeyemo WL, Valencia-Ramirez C, Restrepo-Muneton CP, Moreno LM, Buxo CJ, Long Jr RE, Neiswanger K, Shafer JR, Marazita ML, Weinberg SM. Parents of Children with Non-syndromic Orofacial Clefting Show Altered Palate Shape. Cleft Palate Craniofac J 2020.doi: 10.1177/1055665620967235.