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Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting.
J Dent Res. 2015 Jul; 94(7):905-12.JD

Abstract

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 χ(2) 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.

Authors+Show Affiliations

Department of Family Dentistry, College of Dentistry, University of Iowa, Iowa City, IA, USA.Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA.Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA Department of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA.Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA.Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA.Department of Pediatrics, College of Medicine, University of Iowa, Iowa City, IA, USA.Department of Health Management and Policy, College of Public Health, University of Iowa, Iowa City, IA, USA.Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA Department of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA.Department of Orthodontics, College of Dentistry, University of Iowa, Iowa City, IA, USA Dows Institute for Dental Research, College of Dentistry, University of Iowa, Iowa City, IA, USA lina-moreno@uiowa.edu.

Pub Type(s)

Comparative Study
Journal Article
Research Support, N.I.H., Extramural
Research Support, U.S. Gov't, P.H.S.

Language

eng

PubMed ID

26082386

Citation

Howe, B J., et al. "Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting." Journal of Dental Research, vol. 94, no. 7, 2015, pp. 905-12.
Howe BJ, Cooper ME, Vieira AR, et al. Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting. J Dent Res. 2015;94(7):905-12.
Howe, B. J., Cooper, M. E., Vieira, A. R., Weinberg, S. M., Resick, J. M., Nidey, N. L., Wehby, G. L., Marazita, M. L., & Moreno Uribe, L. M. (2015). Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting. Journal of Dental Research, 94(7), 905-12. https://doi.org/10.1177/0022034515588281
Howe BJ, et al. Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting. J Dent Res. 2015;94(7):905-12. PubMed PMID: 26082386.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting. AU - Howe,B J, AU - Cooper,M E, AU - Vieira,A R, AU - Weinberg,S M, AU - Resick,J M, AU - Nidey,N L, AU - Wehby,G L, AU - Marazita,M L, AU - Moreno Uribe,L M, PY - 2015/6/18/entrez PY - 2015/6/18/pubmed PY - 2015/9/10/medline KW - genetic susceptibility KW - microdontia KW - nonsyndromic cleft lip with or without cleft palate KW - supernumerary teeth KW - tooth abnormalities KW - tooth agenesis SP - 905 EP - 12 JF - Journal of dental research JO - J. Dent. Res. VL - 94 IS - 7 N2 - 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 χ(2) 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. SN - 1544-0591 UR - https://www.unboundmedicine.com/medline/citation/26082386/Spectrum_of_Dental_Phenotypes_in_Nonsyndromic_Orofacial_Clefting_ L2 - http://journals.sagepub.com/doi/full/10.1177/0022034515588281?url_ver=Z39.88-2003&rfr_id=ori:rid:crossref.org&rfr_dat=cr_pub=pubmed DB - PRIME DP - Unbound Medicine ER -