Journal article
Parents of Children With Nonsyndromic Orofacial Clefting Show Altered Palate Shape
The Cleft palate-craniofacial journal, Vol.58(7), pp.847-853
07/01/2021
DOI: 10.1177/1055665620967235
PMCID: PMC8079510
PMID: 33111571
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.
Details
- Title: Subtitle
- Parents of Children With Nonsyndromic Orofacial Clefting Show Altered Palate Shape
- Creators
- Ahmed M. El Sergani - University of PittsburghStephanie Brandebura - University of PittsburghCarmencita Padilla - University of the Philippines ManilaAzeez Butali - University of IowaWasiu L. Adeyemo - University of LagosConsuelo Valencia-Ramirez - 542797Fundación Clínica Noel, Carrera, Medellin, Colombia.Claudia P. Restrepo Muneton - 542797Fundación Clínica Noel, Carrera, Medellin, Colombia.Lina M. Moreno - University of IowaCarmen J. Buxo - University of Puerto Rico, Medical Sciences CampusRoss E. Long - Clinic for Special ChildrenKatherine Neiswanger - University of PittsburghJohn R. Shaffer - University of PittsburghMary L. Marazita - University of PittsburghSeth M. Weinberg - University of Pittsburgh
- Resource Type
- Journal article
- Publication Details
- The Cleft palate-craniofacial journal, Vol.58(7), pp.847-853
- DOI
- 10.1177/1055665620967235
- PMID
- 33111571
- PMCID
- PMC8079510
- NLM abbreviation
- Cleft Palate Craniofac J
- ISSN
- 1055-6656
- eISSN
- 1545-1569
- Publisher
- Sage
- Number of pages
- 7
- Grant note
- R01-DE016148; R00-DE022378; R01-DE015667; R00-DE024571; S21-MD001830; U54-MD007587 / NIH/NIDCR; United States Department of Health & Human Services; National Institutes of Health (NIH) - USA; NIH National Institute of Dental & Craniofacial Research (NIDCR)
- Language
- English
- Date published
- 07/01/2021
- Academic Unit
- Orthodontics; Oral Pathology, Radiology and Medicine; Stead Family Department of Pediatrics; Craniofacial Anomalies Research Center; Dental Research
- Record Identifier
- 9984354006402771
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