Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting

B J Howe; M E Cooper; A R Vieira; S M Weinberg; J M Resick; N L Nidey; G L Wehby; M L Marazita; L M Moreno Uribe

doi:10.1177/0022034515588281

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Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting

Journal article

Open access

Peer reviewed

Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting

B J Howe, M E Cooper, A R Vieira, S M Weinberg, J M Resick, N L Nidey, G L Wehby, M L Marazita and L M Moreno Uribe

Journal of dental research, Vol.94(7), pp.905-912

07/2015

DOI: 10.1177/0022034515588281

PMCID: PMC4530345

PMID: 26082386

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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.

Genetic Predisposition to Disease - genetics

Malocclusion - epidemiology

Cleft Lip - epidemiology

Tooth, Deciduous - abnormalities

Humans

Mandible - pathology

Risk Factors

Male

Case-Control Studies

Genetic Heterogeneity

Phenotype

Dental Arch - pathology

Tooth Eruption, Ectopic - epidemiology

Global Health - statistics & numerical data

Female

Tooth Abnormalities - epidemiology

Cleft Palate - epidemiology

Maxilla - pathology

Child

Tooth, Impacted - epidemiology

Tooth, Supernumerary - epidemiology

Cohort Studies

Details

Title: Subtitle: Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting
Creators: B J Howe - Department of Family Dentistry, College of Dentistry, University of Iowa, Iowa City, IA, USA
M E Cooper - Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA
A R Vieira - 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
S M Weinberg - Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA
J M Resick - Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA
N L Nidey - Department of Pediatrics, College of Medicine, University of Iowa, Iowa City, IA, USA
G L Wehby - Department of Health Management and Policy, College of Public Health, University of Iowa, Iowa City, IA, USA
M L Marazita - 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
L M Moreno Uribe - 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
Resource Type: Journal article
Publication Details: Journal of dental research, Vol.94(7), pp.905-912
DOI: 10.1177/0022034515588281
PMID: 26082386
PMCID: PMC4530345
NLM abbreviation: J Dent Res
ISSN: 0022-0345
eISSN: 1544-0591
Publisher: United States
Grant note: R01 DE014667 / NIDCR NIH HHS R01 DE016148 / NIDCR NIH HHS R37-DE-08559 / NIDCR NIH HHS R37 DE008559 / NIDCR NIH HHS R01 DD000295 / NCBDD CDC HHS UL1 TR000005 / NCATS NIH HHS R01 DE106148 / NIDCR NIH HHS
Language: English
Date published: 07/2015
Academic Unit: Preventive and Community Dentistry; Orthodontics; Health Management and Policy; Stead Family Department of Pediatrics; Epidemiology; Economics; Dental Clinic Administration; Addiction Medicine; Craniofacial Anomalies Research Center; Public Policy Center (Archive); Family Dentistry; Dental Research
Record Identifier: 9984064194302771

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