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<title xml:lang="en">Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting</title>
<author>
<name sortKey="Howe, B J" sort="Howe, B J" uniqKey="Howe B" first="B. J." last="Howe">B. J. Howe</name>
<affiliation>
<nlm:aff id="aff1-0022034515588281">Department of Family Dentistry, College of Dentistry, University of Iowa, Iowa City, IA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Cooper, M E" sort="Cooper, M E" uniqKey="Cooper M" first="M. E." last="Cooper">M. E. Cooper</name>
<affiliation>
<nlm:aff id="aff2-0022034515588281">Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Vieira, A R" sort="Vieira, A R" uniqKey="Vieira A" first="A. R." last="Vieira">A. R. Vieira</name>
<affiliation>
<nlm:aff id="aff2-0022034515588281">Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
<affiliation>
<nlm:aff id="aff3-0022034515588281">Department of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Weinberg, S M" sort="Weinberg, S M" uniqKey="Weinberg S" first="S. M." last="Weinberg">S. M. Weinberg</name>
<affiliation>
<nlm:aff id="aff2-0022034515588281">Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Resick, J M" sort="Resick, J M" uniqKey="Resick J" first="J. M." last="Resick">J. M. Resick</name>
<affiliation>
<nlm:aff id="aff2-0022034515588281">Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Nidey, N L" sort="Nidey, N L" uniqKey="Nidey N" first="N. L." last="Nidey">N. L. Nidey</name>
<affiliation>
<nlm:aff id="aff4-0022034515588281">Department of Pediatrics, College of Medicine, University of Iowa, Iowa City, IA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Wehby, G L" sort="Wehby, G L" uniqKey="Wehby G" first="G. L." last="Wehby">G. L. Wehby</name>
<affiliation>
<nlm:aff id="aff5-0022034515588281">Department of Health Management and Policy, College of Public Health, University of Iowa, Iowa City, IA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Marazita, M L" sort="Marazita, M L" uniqKey="Marazita M" first="M. L." last="Marazita">M. L. Marazita</name>
<affiliation>
<nlm:aff id="aff2-0022034515588281">Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
<affiliation>
<nlm:aff id="aff3-0022034515588281">Department of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Moreno Uribe, L M" sort="Moreno Uribe, L M" uniqKey="Moreno Uribe L" first="L. M." last="Moreno Uribe">L. M. Moreno Uribe</name>
<affiliation>
<nlm:aff id="aff6-0022034515588281">Department of Orthodontics, College of Dentistry, University of Iowa, Iowa City, IA, USA</nlm:aff>
</affiliation>
<affiliation>
<nlm:aff id="aff7-0022034515588281">Dows Institute for Dental Research, College of Dentistry, University of Iowa, Iowa City, IA, USA</nlm:aff>
</affiliation>
</author>
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<idno type="pmid">26082386</idno>
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<idno type="RBID">PMC:4530345</idno>
<idno type="doi">10.1177/0022034515588281</idno>
<date when="2015">2015</date>
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<title xml:lang="en" level="a" type="main">Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting</title>
<author>
<name sortKey="Howe, B J" sort="Howe, B J" uniqKey="Howe B" first="B. J." last="Howe">B. J. Howe</name>
<affiliation>
<nlm:aff id="aff1-0022034515588281">Department of Family Dentistry, College of Dentistry, University of Iowa, Iowa City, IA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Cooper, M E" sort="Cooper, M E" uniqKey="Cooper M" first="M. E." last="Cooper">M. E. Cooper</name>
<affiliation>
<nlm:aff id="aff2-0022034515588281">Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Vieira, A R" sort="Vieira, A R" uniqKey="Vieira A" first="A. R." last="Vieira">A. R. Vieira</name>
<affiliation>
<nlm:aff id="aff2-0022034515588281">Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
<affiliation>
<nlm:aff id="aff3-0022034515588281">Department of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Weinberg, S M" sort="Weinberg, S M" uniqKey="Weinberg S" first="S. M." last="Weinberg">S. M. Weinberg</name>
<affiliation>
<nlm:aff id="aff2-0022034515588281">Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Resick, J M" sort="Resick, J M" uniqKey="Resick J" first="J. M." last="Resick">J. M. Resick</name>
<affiliation>
<nlm:aff id="aff2-0022034515588281">Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Nidey, N L" sort="Nidey, N L" uniqKey="Nidey N" first="N. L." last="Nidey">N. L. Nidey</name>
<affiliation>
<nlm:aff id="aff4-0022034515588281">Department of Pediatrics, College of Medicine, University of Iowa, Iowa City, IA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Wehby, G L" sort="Wehby, G L" uniqKey="Wehby G" first="G. L." last="Wehby">G. L. Wehby</name>
<affiliation>
<nlm:aff id="aff5-0022034515588281">Department of Health Management and Policy, College of Public Health, University of Iowa, Iowa City, IA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Marazita, M L" sort="Marazita, M L" uniqKey="Marazita M" first="M. L." last="Marazita">M. L. Marazita</name>
<affiliation>
<nlm:aff id="aff2-0022034515588281">Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
<affiliation>
<nlm:aff id="aff3-0022034515588281">Department of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</nlm:aff>
</affiliation>
</author>
<author>
<name sortKey="Moreno Uribe, L M" sort="Moreno Uribe, L M" uniqKey="Moreno Uribe L" first="L. M." last="Moreno Uribe">L. M. Moreno Uribe</name>
<affiliation>
<nlm:aff id="aff6-0022034515588281">Department of Orthodontics, College of Dentistry, University of Iowa, Iowa City, IA, USA</nlm:aff>
</affiliation>
<affiliation>
<nlm:aff id="aff7-0022034515588281">Dows Institute for Dental Research, College of Dentistry, University of Iowa, Iowa City, IA, USA</nlm:aff>
</affiliation>
</author>
</analytic>
<series>
<title level="j">Journal of Dental Research</title>
<idno type="ISSN">0022-0345</idno>
<idno type="eISSN">1544-0591</idno>
<imprint>
<date when="2015">2015</date>
</imprint>
</series>
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<textClass></textClass>
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</teiHeader>
<front>
<div type="abstract" xml:lang="en">
<p>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
<sup>2</sup>
statistics. Cases had higher rates of dental anomalies in the maxillary arch than did controls for primary (21% vs. 4%,
<italic>P</italic>
= 3 × 10
<sup>−8</sup>
) and permanent dentitions (51% vs. 8%,
<italic>P</italic>
= 4 × 10
<sup>−62</sup>
) 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%,
<italic>P</italic>
= 0.003) and parents (13% vs. 7%,
<italic>P</italic>
= 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.</p>
</div>
</front>
</TEI>
<pmc article-type="research-article">
<pmc-comment>The publisher of this article does not allow downloading of the full text in XML form.</pmc-comment>
<front>
<journal-meta>
<journal-id journal-id-type="nlm-ta">J Dent Res</journal-id>
<journal-id journal-id-type="iso-abbrev">J. Dent. Res</journal-id>
<journal-id journal-id-type="publisher-id">JDR</journal-id>
<journal-id journal-id-type="hwp">spjdr</journal-id>
<journal-title-group>
<journal-title>Journal of Dental Research</journal-title>
</journal-title-group>
<issn pub-type="ppub">0022-0345</issn>
<issn pub-type="epub">1544-0591</issn>
<publisher>
<publisher-name>SAGE Publications</publisher-name>
<publisher-loc>Sage CA: Los Angeles, CA</publisher-loc>
</publisher>
</journal-meta>
<article-meta>
<article-id pub-id-type="pmid">26082386</article-id>
<article-id pub-id-type="pmc">4530345</article-id>
<article-id pub-id-type="doi">10.1177/0022034515588281</article-id>
<article-id pub-id-type="publisher-id">10.1177_0022034515588281</article-id>
<article-categories>
<subj-group subj-group-type="heading">
<subject>Research Reports</subject>
<subj-group subj-group-type="heading">
<subject>Clinical</subject>
</subj-group>
</subj-group>
</article-categories>
<title-group>
<article-title>Spectrum of Dental Phenotypes in Nonsyndromic Orofacial Clefting</article-title>
</title-group>
<contrib-group>
<contrib contrib-type="author">
<name>
<surname>Howe</surname>
<given-names>B.J.</given-names>
</name>
<xref ref-type="aff" rid="aff1-0022034515588281">1</xref>
<xref ref-type="author-notes" rid="fn1-0022034515588281">*</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Cooper</surname>
<given-names>M.E.</given-names>
</name>
<xref ref-type="aff" rid="aff2-0022034515588281">2</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Vieira</surname>
<given-names>A.R.</given-names>
</name>
<xref ref-type="aff" rid="aff2-0022034515588281">2</xref>
<xref ref-type="aff" rid="aff3-0022034515588281">3</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Weinberg</surname>
<given-names>S.M.</given-names>
</name>
<xref ref-type="aff" rid="aff2-0022034515588281">2</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Resick</surname>
<given-names>J.M.</given-names>
</name>
<xref ref-type="aff" rid="aff2-0022034515588281">2</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Nidey</surname>
<given-names>N.L.</given-names>
</name>
<xref ref-type="aff" rid="aff4-0022034515588281">4</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Wehby</surname>
<given-names>G.L.</given-names>
</name>
<xref ref-type="aff" rid="aff5-0022034515588281">5</xref>
</contrib>
<contrib contrib-type="author">
<name>
<surname>Marazita</surname>
<given-names>M.L.</given-names>
</name>
<xref ref-type="aff" rid="aff2-0022034515588281">2</xref>
<xref ref-type="aff" rid="aff3-0022034515588281">3</xref>
</contrib>
<contrib contrib-type="author" corresp="yes">
<name>
<surname>Moreno Uribe</surname>
<given-names>L.M.</given-names>
</name>
<xref ref-type="aff" rid="aff6-0022034515588281">6</xref>
<xref ref-type="aff" rid="aff7-0022034515588281">7</xref>
<xref ref-type="author-notes" rid="fn1-0022034515588281">*</xref>
</contrib>
</contrib-group>
<aff id="aff1-0022034515588281">
<label>1</label>
Department of Family Dentistry, College of Dentistry, University of Iowa, Iowa City, IA, USA</aff>
<aff id="aff2-0022034515588281">
<label>2</label>
Center for Craniofacial and Dental Genetics School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</aff>
<aff id="aff3-0022034515588281">
<label>3</label>
Department of Oral Biology, School of Dental Medicine, University of Pittsburgh, Pittsburgh, PA, USA</aff>
<aff id="aff4-0022034515588281">
<label>4</label>
Department of Pediatrics, College of Medicine, University of Iowa, Iowa City, IA, USA</aff>
<aff id="aff5-0022034515588281">
<label>5</label>
Department of Health Management and Policy, College of Public Health, University of Iowa, Iowa City, IA, USA</aff>
<aff id="aff6-0022034515588281">
<label>6</label>
Department of Orthodontics, College of Dentistry, University of Iowa, Iowa City, IA, USA</aff>
<aff id="aff7-0022034515588281">
<label>7</label>
Dows Institute for Dental Research, College of Dentistry, University of Iowa, Iowa City, IA, USA</aff>
<author-notes>
<corresp id="corresp1-0022034515588281">L.M. Moreno Uribe, Dows Institute-Research, N401 Dental Science Building, University of Iowa, Iowa City, IA 52242, USA. Email:
<email>lina-moreno@uiowa.edu</email>
</corresp>
<fn fn-type="equal" id="fn1-0022034515588281">
<label>*</label>
<p>Authors contributing equally to this article.</p>
</fn>
</author-notes>
<pub-date pub-type="epub-ppub">
<month>7</month>
<year>2015</year>
</pub-date>
<pmc-comment>Fake ppub date generated by PMC from publisher pub-date/@pub-type='epub-ppub' </pmc-comment>
<pub-date pub-type="ppub">
<month>7</month>
<year>2015</year>
</pub-date>
<pub-date pub-type="pmc-release">
<day>1</day>
<month>7</month>
<year>2016</year>
</pub-date>
<pmc-comment> PMC Release delay is 12 months and 0 days and was based on the . </pmc-comment>
<volume>94</volume>
<issue>7</issue>
<fpage>905</fpage>
<lpage>912</lpage>
<permissions>
<copyright-statement>© International & American Associations for Dental Research 2015</copyright-statement>
<copyright-year>2015</copyright-year>
<copyright-holder content-type="society">International & American Associations for Dental Research</copyright-holder>
</permissions>
<abstract>
<p>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
<sup>2</sup>
statistics. Cases had higher rates of dental anomalies in the maxillary arch than did controls for primary (21% vs. 4%,
<italic>P</italic>
= 3 × 10
<sup>−8</sup>
) and permanent dentitions (51% vs. 8%,
<italic>P</italic>
= 4 × 10
<sup>−62</sup>
) 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%,
<italic>P</italic>
= 0.003) and parents (13% vs. 7%,
<italic>P</italic>
= 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.</p>
</abstract>
<kwd-group>
<kwd>genetic susceptibility</kwd>
<kwd>nonsyndromic cleft lip with or without cleft palate</kwd>
<kwd>tooth agenesis</kwd>
<kwd>microdontia</kwd>
<kwd>supernumerary teeth</kwd>
<kwd>tooth abnormalities</kwd>
</kwd-group>
</article-meta>
</front>
</pmc>
</record>

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