Epidemiological and genetic study of 121 cases of oral clefts in Kuwait
Identifieur interne : 002802 ( Istex/Corpus ); précédent : 002801; suivant : 002803Epidemiological and genetic study of 121 cases of oral clefts in Kuwait
Auteurs : S A Al Ustan ; M M El Awahri ; A M Al Dsani ; R L Bang ; I. Ghunaim ; B S Maher ; S. Weinberg ; M L MarazitaSource :
- Orthodontics & Craniofacial Research [ 1601-6335 ] ; 2002-08.
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Abstract
Authors – S Al‐Bustan, M El‐Zawahri, A Al‐Adsani, R Bang, I Ghunaim, B Maher, S Weinberg, M Marazita Aim – The aim of the study was to ascertain some epidemiological factors such as sex and consanguinity that may be associated with cleft lip with or without cleft palate (CL ± CP) in Kuwait as well as to conduct genetic segregation analysis of these families. Setting and sample population – A total of 113 families ascertained through 121 CL ± CP and CP surgical probands in Kuwait. The frequencies of cleft types and the epidemiological variables were calculated using SPSS version 5.0 software. Chi‐square for goodness‐of‐fit test was used to test the significance of the associated epidemiological variables to facial clefts. Genetic segregation analysis was performed on 76 families with extended pedigrees and included only those with non‐syndromic CL ± CP (NS CL ± CP). Major locus segregation analysis was used to fit models to the observed family patterns under Class A regressive models as implemented by REGD routine in S.A.G.E. release 4.0. A test for heterogeneity was also conducted to complete data set in addition to two subsets: Arabs and nomads. Results – Of the 121 patients, 34(28.1%) had CP, 30(24.8%) had CL and 57 (47.1%) had CL + CP. The male to female ratio was 0.89 for CP, 1.14 for CL, 1.35 for CL + CP and 1.2 for all the clefts. The percentage of consanguineous families among those with a positive family history (60%) was not significantly different from that of the general population (54.3%), whereas for all the families with clefts the percent consanguineous was significantly lower (38%). No evidence of heterogeneity in the results between the Arab and nomad subsets was observed. The results for the major locus segregation analysis were inconclusive. Conclusion – No definite association was observed between consanguinity and the occurrence of facial clefts in Kuwait. General transmission models in the full data set showed no evidence of heterogeneity in the results between the Arab and nomad subsets.
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DOI: 10.1034/j.1600-0544.2002.02203.x
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<record><TEI wicri:istexFullTextTei="biblStruct"><teiHeader><fileDesc><titleStmt><title xml:lang="en">Epidemiological and genetic study of 121 cases of oral clefts in Kuwait</title><author><name sortKey="Al Ustan, S A" sort="Al Ustan, S A" uniqKey="Al Ustan S" first="S A" last="Al Ustan">S A Al Ustan</name><affiliation><mods:affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</mods:affiliation></affiliation></author><author><name sortKey="El Awahri, M M" sort="El Awahri, M M" uniqKey="El Awahri M" first="M M" last="El Awahri">M M El Awahri</name><affiliation><mods:affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</mods:affiliation></affiliation></author><author><name sortKey="Al Dsani, A M" sort="Al Dsani, A M" uniqKey="Al Dsani A" first="A M" last="Al Dsani">A M Al Dsani</name><affiliation><mods:affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</mods:affiliation></affiliation></author><author><name sortKey="Bang, R L" sort="Bang, R L" uniqKey="Bang R" first="R L" last="Bang">R L Bang</name><affiliation><mods:affiliation>Department of Surgery, Faculty of Medicine, Kuwait University, Kuwait,</mods:affiliation></affiliation><affiliation><mods:affiliation>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation></author><author><name sortKey="Ghunaim, I" sort="Ghunaim, I" uniqKey="Ghunaim I" first="I" last="Ghunaim">I. Ghunaim</name><affiliation><mods:affiliation>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation></author><author><name sortKey="Maher, B S" sort="Maher, B S" uniqKey="Maher B" first="B S" last="Maher">B S Maher</name><affiliation><mods:affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation><affiliation><mods:affiliation>Department of Human Genetics, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation></author><author><name sortKey="Weinberg, S" sort="Weinberg, S" uniqKey="Weinberg S" first="S" last="Weinberg">S. Weinberg</name><affiliation><mods:affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation><affiliation><mods:affiliation>Department of Anthropology, University of Pittsburgh, PA, USA</mods:affiliation></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><mods:affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation><affiliation><mods:affiliation>Department of Human Genetics, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">ISTEX</idno><idno type="RBID">ISTEX:DC67F621EEF6681683E2CC863139B0F3116938D9</idno><date when="2002" year="2002">2002</date><idno type="doi">10.1034/j.1600-0544.2002.02203.x</idno><idno type="url">https://api.istex.fr/document/DC67F621EEF6681683E2CC863139B0F3116938D9/fulltext/pdf</idno><idno type="wicri:Area/Istex/Corpus">002802</idno></publicationStmt><sourceDesc><biblStruct><analytic><title level="a" type="main" xml:lang="en">Epidemiological and genetic study of 121 cases of oral clefts in Kuwait</title><author><name sortKey="Al Ustan, S A" sort="Al Ustan, S A" uniqKey="Al Ustan S" first="S A" last="Al Ustan">S A Al Ustan</name><affiliation><mods:affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</mods:affiliation></affiliation></author><author><name sortKey="El Awahri, M M" sort="El Awahri, M M" uniqKey="El Awahri M" first="M M" last="El Awahri">M M El Awahri</name><affiliation><mods:affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</mods:affiliation></affiliation></author><author><name sortKey="Al Dsani, A M" sort="Al Dsani, A M" uniqKey="Al Dsani A" first="A M" last="Al Dsani">A M Al Dsani</name><affiliation><mods:affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</mods:affiliation></affiliation></author><author><name sortKey="Bang, R L" sort="Bang, R L" uniqKey="Bang R" first="R L" last="Bang">R L Bang</name><affiliation><mods:affiliation>Department of Surgery, Faculty of Medicine, Kuwait University, Kuwait,</mods:affiliation></affiliation><affiliation><mods:affiliation>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation></author><author><name sortKey="Ghunaim, I" sort="Ghunaim, I" uniqKey="Ghunaim I" first="I" last="Ghunaim">I. Ghunaim</name><affiliation><mods:affiliation>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation></author><author><name sortKey="Maher, B S" sort="Maher, B S" uniqKey="Maher B" first="B S" last="Maher">B S Maher</name><affiliation><mods:affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation><affiliation><mods:affiliation>Department of Human Genetics, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation></author><author><name sortKey="Weinberg, S" sort="Weinberg, S" uniqKey="Weinberg S" first="S" last="Weinberg">S. Weinberg</name><affiliation><mods:affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation><affiliation><mods:affiliation>Department of Anthropology, University of Pittsburgh, PA, USA</mods:affiliation></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><mods:affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation><affiliation><mods:affiliation>Department of Human Genetics, University of Pittsburgh, PA, USA,</mods:affiliation></affiliation></author></analytic><monogr></monogr><series><title level="j">Orthodontics & Craniofacial Research</title><idno type="ISSN">1601-6335</idno><idno type="eISSN">1601-6343</idno><imprint><publisher>Munksgaard International Publishers</publisher><pubPlace>Oxford UK</pubPlace><date type="published" when="2002-08">2002-08</date><biblScope unit="volume">5</biblScope><biblScope unit="issue">3</biblScope><biblScope unit="page" from="154">154</biblScope><biblScope unit="page" to="160">160</biblScope></imprint><idno type="ISSN">1601-6335</idno></series><idno type="istex">DC67F621EEF6681683E2CC863139B0F3116938D9</idno><idno type="DOI">10.1034/j.1600-0544.2002.02203.x</idno><idno type="ArticleID">OCR2O203</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">1601-6335</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Kuwait</term><term>cleft lip</term><term>cleft palate</term><term>epidemiology</term><term>genetics</term><term>segregation analysis</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Authors – S Al‐Bustan, M El‐Zawahri, A Al‐Adsani, R Bang, I Ghunaim, B Maher, S Weinberg, M Marazita Aim – The aim of the study was to ascertain some epidemiological factors such as sex and consanguinity that may be associated with cleft lip with or without cleft palate (CL ± CP) in Kuwait as well as to conduct genetic segregation analysis of these families. Setting and sample population – A total of 113 families ascertained through 121 CL ± CP and CP surgical probands in Kuwait. The frequencies of cleft types and the epidemiological variables were calculated using SPSS version 5.0 software. Chi‐square for goodness‐of‐fit test was used to test the significance of the associated epidemiological variables to facial clefts. Genetic segregation analysis was performed on 76 families with extended pedigrees and included only those with non‐syndromic CL ± CP (NS CL ± CP). Major locus segregation analysis was used to fit models to the observed family patterns under Class A regressive models as implemented by REGD routine in S.A.G.E. release 4.0. A test for heterogeneity was also conducted to complete data set in addition to two subsets: Arabs and nomads. Results – Of the 121 patients, 34(28.1%) had CP, 30(24.8%) had CL and 57 (47.1%) had CL + CP. The male to female ratio was 0.89 for CP, 1.14 for CL, 1.35 for CL + CP and 1.2 for all the clefts. The percentage of consanguineous families among those with a positive family history (60%) was not significantly different from that of the general population (54.3%), whereas for all the families with clefts the percent consanguineous was significantly lower (38%). No evidence of heterogeneity in the results between the Arab and nomad subsets was observed. The results for the major locus segregation analysis were inconclusive. Conclusion – No definite association was observed between consanguinity and the occurrence of facial clefts in Kuwait. General transmission models in the full data set showed no evidence of heterogeneity in the results between the Arab and nomad subsets.</div></front></TEI><istex><corpusName>wiley</corpusName><author><json:item><name>S A Al‐Bustan</name><affiliations><json:string>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</json:string></affiliations></json:item><json:item><name>M M El‐Zawahri</name><affiliations><json:string>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</json:string></affiliations></json:item><json:item><name>A M Al‐Adsani</name><affiliations><json:string>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</json:string></affiliations></json:item><json:item><name>R L Bang</name><affiliations><json:string>Department of Surgery, Faculty of Medicine, Kuwait University, Kuwait,</json:string><json:string>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</json:string></affiliations></json:item><json:item><name>I Ghunaim</name><affiliations><json:string>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</json:string></affiliations></json:item><json:item><name>B S Maher</name><affiliations><json:string>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</json:string><json:string>Department of Human Genetics, University of Pittsburgh, PA, USA,</json:string></affiliations></json:item><json:item><name>S Weinberg</name><affiliations><json:string>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</json:string><json:string>Department of Anthropology, University of Pittsburgh, PA, USA</json:string></affiliations></json:item><json:item><name>M L Marazita</name><affiliations><json:string>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</json:string><json:string>Department of Human Genetics, University of Pittsburgh, PA, USA,</json:string></affiliations></json:item></author><subject><json:item><lang><json:string>eng</json:string></lang><value>cleft lip</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>cleft palate</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>epidemiology</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>genetics</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>Kuwait</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>segregation analysis</value></json:item></subject><articleId><json:string>OCR2O203</json:string></articleId><language><json:string>eng</json:string></language><abstract>Authors – S Al‐Bustan, M El‐Zawahri, A Al‐Adsani, R Bang, I Ghunaim, B Maher, S Weinberg, M Marazita Aim – The aim of the study was to ascertain some epidemiological factors such as sex and consanguinity that may be associated with cleft lip with or without cleft palate (CL ± CP) in Kuwait as well as to conduct genetic segregation analysis of these families. Setting and sample population – A total of 113 families ascertained through 121 CL ± CP and CP surgical probands in Kuwait. The frequencies of cleft types and the epidemiological variables were calculated using SPSS version 5.0 software. Chi‐square for goodness‐of‐fit test was used to test the significance of the associated epidemiological variables to facial clefts. Genetic segregation analysis was performed on 76 families with extended pedigrees and included only those with non‐syndromic CL ± CP (NS CL ± CP). Major locus segregation analysis was used to fit models to the observed family patterns under Class A regressive models as implemented by REGD routine in S.A.G.E. release 4.0. A test for heterogeneity was also conducted to complete data set in addition to two subsets: Arabs and nomads. Results – Of the 121 patients, 34(28.1%) had CP, 30(24.8%) had CL and 57 (47.1%) had CL + CP. The male to female ratio was 0.89 for CP, 1.14 for CL, 1.35 for CL + CP and 1.2 for all the clefts. The percentage of consanguineous families among those with a positive family history (60%) was not significantly different from that of the general population (54.3%), whereas for all the families with clefts the percent consanguineous was significantly lower (38%). No evidence of heterogeneity in the results between the Arab and nomad subsets was observed. The results for the major locus segregation analysis were inconclusive. Conclusion – No definite association was observed between consanguinity and the occurrence of facial clefts in Kuwait. General transmission models in the full data set showed no evidence of heterogeneity in the results between the Arab and nomad subsets.</abstract><qualityIndicators><score>7.116</score><pdfVersion>1.3</pdfVersion><pdfPageSize>595 x 788 pts</pdfPageSize><refBibsNative>true</refBibsNative><keywordCount>6</keywordCount><abstractCharCount>2045</abstractCharCount><pdfWordCount>4116</pdfWordCount><pdfCharCount>24335</pdfCharCount><pdfPageCount>7</pdfPageCount><abstractWordCount>323</abstractWordCount></qualityIndicators><title>Epidemiological and genetic study of 121 cases of oral clefts in Kuwait</title><genre.original><json:string>article</json:string></genre.original><genre><json:string>article</json:string></genre><host><volume>5</volume><publisherId><json:string>OCR</json:string></publisherId><pages><total>7</total><last>160</last><first>154</first></pages><issn><json:string>1601-6335</json:string></issn><issue>3</issue><genre><json:string>journal</json:string></genre><language><json:string>unknown</json:string></language><eissn><json:string>1601-6343</json:string></eissn><title>Orthodontics & Craniofacial Research</title><doi><json:string>10.1111/(ISSN)1601-6343</json:string></doi></host><publicationDate>2002</publicationDate><copyrightDate>2002</copyrightDate><doi><json:string>10.1034/j.1600-0544.2002.02203.x</json:string></doi><id>DC67F621EEF6681683E2CC863139B0F3116938D9</id><fulltext><json:item><original>true</original><mimetype>application/pdf</mimetype><extension>pdf</extension><uri>https://api.istex.fr/document/DC67F621EEF6681683E2CC863139B0F3116938D9/fulltext/pdf</uri></json:item><json:item><original>false</original><mimetype>application/zip</mimetype><extension>zip</extension><uri>https://api.istex.fr/document/DC67F621EEF6681683E2CC863139B0F3116938D9/fulltext/zip</uri></json:item><istex:fulltextTEI uri="https://api.istex.fr/document/DC67F621EEF6681683E2CC863139B0F3116938D9/fulltext/tei"><teiHeader><fileDesc><titleStmt><title level="a" type="main" xml:lang="en">Epidemiological and genetic study of 121 cases of oral clefts in Kuwait</title></titleStmt><publicationStmt><authority>ISTEX</authority><publisher>Munksgaard International Publishers</publisher><pubPlace>Oxford UK</pubPlace><availability><p>WILEY</p></availability><date>2002</date></publicationStmt><notesStmt><note type="content">*To cite this article: Orthod. Craniofacial Res. 5, 2002; 154–160 Al‐Bustan SA, El‐Zawahri MM, Al‐Adsani AM, Bang RL, Ghunaim I, Maher BS, Weinberg S, Marazita ML: Epidemiological and genetic study of 121 cases of oral clefts in Kuwait.</note></notesStmt><sourceDesc><biblStruct type="inbook"><analytic><title level="a" type="main" xml:lang="en">Epidemiological and genetic study of 121 cases of oral clefts in Kuwait</title><author><persName><forename type="first">S A</forename><surname>Al‐Bustan</surname></persName><affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</affiliation></author><author><persName><forename type="first">M M</forename><surname>El‐Zawahri</surname></persName><affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</affiliation></author><author><persName><forename type="first">A M</forename><surname>Al‐Adsani</surname></persName><affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</affiliation></author><author><persName><forename type="first">R L</forename><surname>Bang</surname></persName><affiliation>Department of Surgery, Faculty of Medicine, Kuwait University, Kuwait,</affiliation><affiliation>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</affiliation></author><author><persName><forename type="first">I</forename><surname>Ghunaim</surname></persName><affiliation>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</affiliation></author><author><persName><forename type="first">B S</forename><surname>Maher</surname></persName><affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</affiliation><affiliation>Department of Human Genetics, University of Pittsburgh, PA, USA,</affiliation></author><author><persName><forename type="first">S</forename><surname>Weinberg</surname></persName><affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</affiliation><affiliation>Department of Anthropology, University of Pittsburgh, PA, USA</affiliation></author><author><persName><forename type="first">M L</forename><surname>Marazita</surname></persName><affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</affiliation><affiliation>Department of Human Genetics, University of Pittsburgh, PA, USA,</affiliation></author></analytic><monogr><title level="j">Orthodontics & Craniofacial Research</title><idno type="pISSN">1601-6335</idno><idno type="eISSN">1601-6343</idno><idno type="DOI">10.1111/(ISSN)1601-6343</idno><imprint><publisher>Munksgaard International Publishers</publisher><pubPlace>Oxford UK</pubPlace><date type="published" when="2002-08"></date><biblScope unit="volume">5</biblScope><biblScope unit="issue">3</biblScope><biblScope unit="page" from="154">154</biblScope><biblScope unit="page" to="160">160</biblScope></imprint></monogr><idno type="istex">DC67F621EEF6681683E2CC863139B0F3116938D9</idno><idno type="DOI">10.1034/j.1600-0544.2002.02203.x</idno><idno type="ArticleID">OCR2O203</idno></biblStruct></sourceDesc></fileDesc><profileDesc><creation><date>2002</date></creation><langUsage><language ident="en">en</language></langUsage><abstract xml:lang="en"><p>Authors – S Al‐Bustan, M El‐Zawahri, A Al‐Adsani, R Bang, I Ghunaim, B Maher, S Weinberg, M Marazita Aim – The aim of the study was to ascertain some epidemiological factors such as sex and consanguinity that may be associated with cleft lip with or without cleft palate (CL ± CP) in Kuwait as well as to conduct genetic segregation analysis of these families. Setting and sample population – A total of 113 families ascertained through 121 CL ± CP and CP surgical probands in Kuwait. The frequencies of cleft types and the epidemiological variables were calculated using SPSS version 5.0 software. Chi‐square for goodness‐of‐fit test was used to test the significance of the associated epidemiological variables to facial clefts. Genetic segregation analysis was performed on 76 families with extended pedigrees and included only those with non‐syndromic CL ± CP (NS CL ± CP). Major locus segregation analysis was used to fit models to the observed family patterns under Class A regressive models as implemented by REGD routine in S.A.G.E. release 4.0. A test for heterogeneity was also conducted to complete data set in addition to two subsets: Arabs and nomads. Results – Of the 121 patients, 34(28.1%) had CP, 30(24.8%) had CL and 57 (47.1%) had CL + CP. The male to female ratio was 0.89 for CP, 1.14 for CL, 1.35 for CL + CP and 1.2 for all the clefts. The percentage of consanguineous families among those with a positive family history (60%) was not significantly different from that of the general population (54.3%), whereas for all the families with clefts the percent consanguineous was significantly lower (38%). No evidence of heterogeneity in the results between the Arab and nomad subsets was observed. The results for the major locus segregation analysis were inconclusive. Conclusion – No definite association was observed between consanguinity and the occurrence of facial clefts in Kuwait. General transmission models in the full data set showed no evidence of heterogeneity in the results between the Arab and nomad subsets.</p></abstract><textClass xml:lang="en"><keywords scheme="keyword"><list><head>keywords</head><item><term>cleft lip</term></item><item><term>cleft palate</term></item><item><term>epidemiology</term></item><item><term>genetics</term></item><item><term>Kuwait</term></item><item><term>segregation analysis</term></item></list></keywords></textClass></profileDesc><revisionDesc><change when="2002-08">Published</change></revisionDesc></teiHeader></istex:fulltextTEI><json:item><original>false</original><mimetype>text/plain</mimetype><extension>txt</extension><uri>https://api.istex.fr/document/DC67F621EEF6681683E2CC863139B0F3116938D9/fulltext/txt</uri></json:item></fulltext><metadata><istex:metadataXml wicri:clean="Wiley, elements deleted: body"><istex:xmlDeclaration>version="1.0" encoding="UTF-8" standalone="yes"</istex:xmlDeclaration><istex:document><component version="2.0" type="serialArticle" xml:lang="en"><header><publicationMeta level="product"><publisherInfo><publisherName>Munksgaard International Publishers</publisherName><publisherLoc>Oxford UK</publisherLoc></publisherInfo><doi origin="wiley" registered="yes">10.1111/(ISSN)1601-6343</doi><issn type="print">1601-6335</issn><issn type="electronic">1601-6343</issn><idGroup><id type="product" value="OCR"></id><id type="publisherDivision" value="ST"></id></idGroup><titleGroup><title type="main" sort="ORTHODONTICS CRANIOFACIAL RESEARCH">Orthodontics & Craniofacial Research</title></titleGroup></publicationMeta><publicationMeta level="part" position="08003"><doi origin="wiley">10.1111/ocr.2002.5.issue-3</doi><numberingGroup><numbering type="journalVolume" number="5">5</numbering><numbering type="journalIssue" number="3">3</numbering></numberingGroup><coverDate startDate="2002-08">August 2002</coverDate></publicationMeta><publicationMeta level="unit" type="article" position="0015400" status="forIssue"><doi origin="wiley">10.1034/j.1600-0544.2002.02203.x</doi><idGroup><id type="unit" value="OCR2O203"></id></idGroup><countGroup><count type="pageTotal" number="7"></count></countGroup><titleGroup><title type="tocHeading1">Original Articles</title></titleGroup><eventGroup><event type="firstOnline" date="2003-01-13"></event><event type="publishedOnlineFinalForm" date="2003-01-13"></event><event type="xmlConverted" agent="Converter:BPG_TO_WML3G version:2.3.2 mode:FullText source:FullText result:FullText" date="2010-03-04"></event><event type="xmlConverted" agent="Converter:WILEY_ML3G_TO_WILEY_ML3GV2 version:3.8.8" date="2014-02-06"></event><event type="xmlConverted" agent="Converter:WML3G_To_WML3G version:4.1.7 mode:FullText,remove_FC" date="2014-11-03"></event></eventGroup><numberingGroup><numbering type="pageFirst" number="154">154</numbering><numbering type="pageLast" number="160">160</numbering></numberingGroup><correspondenceTo> Dr. Suzanne A. Al‐Bustan Department of Biological Sciences, Faculty of Science, Kuwait University PO Box: 5969, Safat 13060 Kuwait Tel.:(965) 4811188 ext. 7130/5607 Fax:(965) 5335149 E‐mail: <email>suzanne@kuc01.kuniv.edu.kw</email></correspondenceTo><linkGroup><link type="toTypesetVersion" href="file:OCR.OCR2o203.pdf"></link></linkGroup></publicationMeta><contentMeta><countGroup><count type="tableTotal" number="0"></count><count type="referenceTotal" number="39"></count><count type="linksPubMed" number="22"></count><count type="linksCrossRef" number="0"></count></countGroup><titleGroup><title type="main">Epidemiological and genetic study of 121 cases of oral clefts in Kuwait<link href="#fn1"></link></title><title type="shortAuthors">Al‐Bustan et al.</title><title type="short">Epidemiology and genetics of CL*CP in Kuwait</title></titleGroup><creators><creator creatorRole="author" xml:id="cr1" affiliationRef="#a1"><personName><givenNames>S A</givenNames><familyName>Al‐Bustan</familyName></personName></creator><creator creatorRole="author" xml:id="cr2" affiliationRef="#a1"><personName><givenNames>M M</givenNames><familyName>El‐Zawahri</familyName></personName></creator><creator creatorRole="author" xml:id="cr3" affiliationRef="#a1"><personName><givenNames>A M</givenNames><familyName>Al‐Adsani</familyName></personName></creator><creator creatorRole="author" xml:id="cr4" affiliationRef="#a2 #a3"><personName><givenNames>R L</givenNames><familyName>Bang</familyName></personName></creator><creator creatorRole="author" xml:id="cr5" affiliationRef="#a3"><personName><givenNames>I</givenNames><familyName>Ghunaim</familyName></personName></creator><creator creatorRole="author" xml:id="cr6" affiliationRef="#a4 #a5"><personName><givenNames>B S</givenNames><familyName>Maher</familyName></personName></creator><creator creatorRole="author" xml:id="cr7" affiliationRef="#a4 #a6"><personName><givenNames>S</givenNames><familyName>Weinberg</familyName></personName></creator><creator creatorRole="author" xml:id="cr8" affiliationRef="#a4 #a5"><personName><givenNames>M L</givenNames><familyName>Marazita</familyName></personName></creator></creators><affiliationGroup><affiliation xml:id="a1" countryCode="KW"><unparsedAffiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</unparsedAffiliation></affiliation><affiliation xml:id="a2" countryCode="KW"><unparsedAffiliation>Department of Surgery, Faculty of Medicine, Kuwait University, Kuwait,</unparsedAffiliation></affiliation><affiliation xml:id="a3"><unparsedAffiliation>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</unparsedAffiliation></affiliation><affiliation xml:id="a4"><unparsedAffiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</unparsedAffiliation></affiliation><affiliation xml:id="a5"><unparsedAffiliation>Department of Human Genetics, University of Pittsburgh, PA, USA,</unparsedAffiliation></affiliation><affiliation xml:id="a6" countryCode="US"><unparsedAffiliation>Department of Anthropology, University of Pittsburgh, PA, USA</unparsedAffiliation></affiliation></affiliationGroup><keywordGroup xml:lang="en"><keyword xml:id="k1">cleft lip</keyword><keyword xml:id="k2">cleft palate</keyword><keyword xml:id="k3">epidemiology</keyword><keyword xml:id="k4">genetics</keyword><keyword xml:id="k5">Kuwait</keyword><keyword xml:id="k6">segregation analysis</keyword></keywordGroup><abstractGroup><abstract type="main" xml:lang="en"><title type="main">Structured Abstract</title><p> <i><b>Authors –</b></i> S Al‐Bustan, M El‐Zawahri, A Al‐Adsani, R Bang, I Ghunaim, B Maher, S Weinberg, M Marazita</p><p> <i><b>Aim –</b></i> The aim of the study was to ascertain some epidemiological factors such as sex and consanguinity that may be associated with cleft lip with or without cleft palate (CL ± CP) in Kuwait as well as to conduct genetic segregation analysis of these families.</p><p> <i><b>Setting and sample population –</b></i> A total of 113 families ascertained through 121 CL ± CP and CP surgical probands in Kuwait. The frequencies of cleft types and the epidemiological variables were calculated using SPSS version 5.0 software. Chi‐square for goodness‐of‐fit test was used to test the significance of the associated epidemiological variables to facial clefts. Genetic segregation analysis was performed on 76 families with extended pedigrees and included only those with non‐syndromic CL ± CP (NS CL ± CP). Major locus segregation analysis was used to fit models to the observed family patterns under Class A regressive models as implemented by REGD routine in S.A.G.E. release 4.0. A test for heterogeneity was also conducted to complete data set in addition to two subsets: Arabs and nomads.</p><p> <i><b>Results –</b></i> Of the 121 patients, 34(28.1%) had CP, 30(24.8%) had CL and 57 (47.1%) had CL + CP. The male to female ratio was 0.89 for CP, 1.14 for CL, 1.35 for CL + CP and 1.2 for all the clefts. The percentage of consanguineous families among those with a positive family history (60%) was not significantly different from that of the general population (54.3%), whereas for all the families with clefts the percent consanguineous was significantly lower (38%). No evidence of heterogeneity in the results between the Arab and nomad subsets was observed. The results for the major locus segregation analysis were inconclusive.</p><p> <i><b>Conclusion –</b></i> No definite association was observed between consanguinity and the occurrence of facial clefts in Kuwait. General transmission models in the full data set showed no evidence of heterogeneity in the results between the Arab and nomad subsets.</p></abstract></abstractGroup></contentMeta></header></component></istex:document></istex:metadataXml><mods version="3.6"><titleInfo lang="en"><title>Epidemiological and genetic study of 121 cases of oral clefts in Kuwait</title></titleInfo><titleInfo type="abbreviated"><title>Epidemiology and genetics of CL*CP in Kuwait</title></titleInfo><titleInfo type="alternative" contentType="CDATA" lang="en"><title>Epidemiological and genetic study of 121 cases of oral clefts in Kuwait</title></titleInfo><name type="personal"><namePart type="given">S A</namePart><namePart type="family">Al‐Bustan</namePart><affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">M M</namePart><namePart type="family">El‐Zawahri</namePart><affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">A M</namePart><namePart type="family">Al‐Adsani</namePart><affiliation>Department of Biological Sciences, Faculty of Science, Kuwait University, Kuwait,</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">R L</namePart><namePart type="family">Bang</namePart><affiliation>Department of Surgery, Faculty of Medicine, Kuwait University, Kuwait,</affiliation><affiliation>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">I</namePart><namePart type="family">Ghunaim</namePart><affiliation>Al‐Babtain Center for Burns and Plastic Surgery, School of Dental Medicine, University of Pittsburgh, PA, USA,</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">B S</namePart><namePart type="family">Maher</namePart><affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</affiliation><affiliation>Department of Human Genetics, University of Pittsburgh, PA, USA,</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">S</namePart><namePart type="family">Weinberg</namePart><affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</affiliation><affiliation>Department of Anthropology, University of Pittsburgh, PA, USA</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">M L</namePart><namePart type="family">Marazita</namePart><affiliation>Division of Oral Biology, School of Dental Medicine, University of Pittsburgh, PA, USA,</affiliation><affiliation>Department of Human Genetics, University of Pittsburgh, PA, USA,</affiliation><role><roleTerm type="text">author</roleTerm></role></name><typeOfResource>text</typeOfResource><genre type="article" displayLabel="article"></genre><originInfo><publisher>Munksgaard International Publishers</publisher><place><placeTerm type="text">Oxford UK</placeTerm></place><dateIssued encoding="w3cdtf">2002-08</dateIssued><copyrightDate encoding="w3cdtf">2002</copyrightDate></originInfo><language><languageTerm type="code" authority="rfc3066">en</languageTerm><languageTerm type="code" authority="iso639-2b">eng</languageTerm></language><physicalDescription><internetMediaType>text/html</internetMediaType><extent unit="references">39</extent></physicalDescription><abstract lang="en">Authors – S Al‐Bustan, M El‐Zawahri, A Al‐Adsani, R Bang, I Ghunaim, B Maher, S Weinberg, M Marazita Aim – The aim of the study was to ascertain some epidemiological factors such as sex and consanguinity that may be associated with cleft lip with or without cleft palate (CL ± CP) in Kuwait as well as to conduct genetic segregation analysis of these families. Setting and sample population – A total of 113 families ascertained through 121 CL ± CP and CP surgical probands in Kuwait. The frequencies of cleft types and the epidemiological variables were calculated using SPSS version 5.0 software. Chi‐square for goodness‐of‐fit test was used to test the significance of the associated epidemiological variables to facial clefts. Genetic segregation analysis was performed on 76 families with extended pedigrees and included only those with non‐syndromic CL ± CP (NS CL ± CP). Major locus segregation analysis was used to fit models to the observed family patterns under Class A regressive models as implemented by REGD routine in S.A.G.E. release 4.0. A test for heterogeneity was also conducted to complete data set in addition to two subsets: Arabs and nomads. Results – Of the 121 patients, 34(28.1%) had CP, 30(24.8%) had CL and 57 (47.1%) had CL + CP. The male to female ratio was 0.89 for CP, 1.14 for CL, 1.35 for CL + CP and 1.2 for all the clefts. The percentage of consanguineous families among those with a positive family history (60%) was not significantly different from that of the general population (54.3%), whereas for all the families with clefts the percent consanguineous was significantly lower (38%). No evidence of heterogeneity in the results between the Arab and nomad subsets was observed. The results for the major locus segregation analysis were inconclusive. Conclusion – No definite association was observed between consanguinity and the occurrence of facial clefts in Kuwait. General transmission models in the full data set showed no evidence of heterogeneity in the results between the Arab and nomad subsets.</abstract><note type="content">*To cite this article: Orthod. Craniofacial Res. 5, 2002; 154–160 Al‐Bustan SA, El‐Zawahri MM, Al‐Adsani AM, Bang RL, Ghunaim I, Maher BS, Weinberg S, Marazita ML: Epidemiological and genetic study of 121 cases of oral clefts in Kuwait.</note><subject lang="en"><genre>keywords</genre><topic>cleft lip</topic><topic>cleft palate</topic><topic>epidemiology</topic><topic>genetics</topic><topic>Kuwait</topic><topic>segregation analysis</topic></subject><relatedItem type="host"><titleInfo><title>Orthodontics & Craniofacial Research</title></titleInfo><genre type="journal">journal</genre><identifier type="ISSN">1601-6335</identifier><identifier type="eISSN">1601-6343</identifier><identifier type="DOI">10.1111/(ISSN)1601-6343</identifier><identifier type="PublisherID">OCR</identifier><part><date>2002</date><detail type="volume"><caption>vol.</caption><number>5</number></detail><detail type="issue"><caption>no.</caption><number>3</number></detail><extent unit="pages"><start>154</start><end>160</end><total>7</total></extent></part></relatedItem><identifier type="istex">DC67F621EEF6681683E2CC863139B0F3116938D9</identifier><identifier type="DOI">10.1034/j.1600-0544.2002.02203.x</identifier><identifier type="ArticleID">OCR2O203</identifier><recordInfo><recordContentSource>WILEY</recordContentSource><recordOrigin>Munksgaard International Publishers</recordOrigin></recordInfo></mods></metadata><serie></serie></istex></record>Pour manipuler ce document sous Unix (Dilib)
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