{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,30]],"date-time":"2026-04-30T04:13:05Z","timestamp":1777522385527,"version":"3.51.4"},"reference-count":44,"publisher":"SAGE Publications","issue":"2","license":[{"start":{"date-parts":[[2013,3,1]],"date-time":"2013-03-01T00:00:00Z","timestamp":1362096000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["The Cleft Palate Craniofacial Journal"],"published-print":{"date-parts":[[2013,3]]},"abstract":"<jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>Ectrodactyly-ectodermal dysplasia-clefting syndrome is a congenital anomaly characterized by ectodermal dysplasia, ectrodactyly, cleft lip and palate, and lacrimal duct anomalies. Because this syndrome is frequently accompanied by a congenital lack of teeth, narrow palate, and malocclusion, comprehensive orthodontic intervention is required.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design<\/jats:title>\n                    <jats:p>To highlight the specific dental and maxillofacial characteristics of ectrodactylyectodermal dysplasia-clefting syndrome, six Japanese individuals diagnosed with the syndrome are described here.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Patients<\/jats:title>\n                    <jats:p>The subjects consisted of two boys and four girls (age range, 6.0 to 13.9 years) diagnosed with ectrodactyly-ectodermal dysplasia-clefting syndrome by medical and dental specialists. Their conditions included ectodermal dysplasia (hypodontia, microdontia, enamel hypoplasia, and abnormalities in hair and nails), cleft lip and\/or palate, and ectrodactyly. Cephalograms, panoramic x-rays, and dental casts were taken; systemic complications were recorded at the first visit to our dental hospital.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>All individuals had severe oligodontia with 9 to 18 missing teeth. The missing teeth were mainly maxillary and mandibular incisors and second bicuspids, arranged in a symmetrical manner. Cephalometric analysis showed retruded and short maxilla due to cleft lip and\/or palate. It is interesting that all individuals showed a characteristically shaped mandibular symphysis with a retruded point B. It is likely that this unusual symphyseal morphology is due to the lack of mandibular incisors.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>This study demonstrates the presence of severe oligodontia in the incisal and premolar regions and describes a characteristic maxillary and mandibular structure in Japanese individuals with ectrodactyly-ectodermal dysplasia-clefting syndrome.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1597\/11-123","type":"journal-article","created":{"date-parts":[[2012,1,11]],"date-time":"2012-01-11T18:33:24Z","timestamp":1326306804000},"page":"192-200","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":9,"title":["Dental and Maxillofacial Characteristics of Six Japanese Individuals with Ectrodactyly-Ectodermal Dysplasia-Clefting Syndrome"],"prefix":"10.1177","volume":"50","author":[{"given":"Erika","family":"Okamura","sequence":"first","affiliation":[{"name":"Maxillofacial Orthognathics, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Naoto","family":"Suda","sequence":"additional","affiliation":[{"name":"Division of Orthodontics, Department of Human Development and Fostering, Meikai University School of Dentistry, Saitama, Japan."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoshiyuki","family":"Baba","sequence":"additional","affiliation":[{"name":"Maxillofacial Orthognathics, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan."},{"name":"Division of Dentistry, Department of Surgical Specialties, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hiroki","family":"Fukuoka","sequence":"additional","affiliation":[{"name":"Maxillofacial Orthognathics, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan."},{"name":"Global COE Program of JSPS, International Research Center for Molecular Science in Tooth and Bone Diseases."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takuya","family":"Ogawa","sequence":"additional","affiliation":[{"name":"Maxillofacial Orthognathics, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan."},{"name":"Global COE Program of JSPS, International Research Center for Molecular Science in Tooth and Bone Diseases."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mizue","family":"Ohkuma","sequence":"additional","affiliation":[{"name":"Maxillofacial Orthognathics, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nozomi","family":"Ahiko","sequence":"additional","affiliation":[{"name":"Maxillofacial Orthognathics, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Akihiro","family":"Yasue","sequence":"additional","affiliation":[{"name":"Department of Orthodontics and Dentofacial Orthopedics, Institute of Health Biosciences, The University of Tokushima Graduate School, Tokushima, Japan."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Toshimoto","family":"Tengan","sequence":"additional","affiliation":[{"name":"Department of Clinical Neuroscience Oral and Maxillofacial Functional Rehabilitation, Faculty of Medicine, University of the Ryukyus, Okinawa, Japan."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Momotoshi","family":"Shiga","sequence":"additional","affiliation":[{"name":"Maxillofacial Orthognathics, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michiko","family":"Tsuji","sequence":"additional","affiliation":[{"name":"Maxillofacial Orthognathics, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan."},{"name":"Global COE Program of JSPS, International Research Center for Molecular Science in Tooth and Bone Diseases."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Keiji","family":"Moriyama","sequence":"additional","affiliation":[{"name":"Maxillofacial Orthognathics, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan."},{"name":"Global COE Program of JSPS, International Research Center for Molecular Science in Tooth and Bone Diseases."}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2013,3,1]]},"reference":[{"key":"bibr1-11-123","doi-asserted-by":"publisher","DOI":"10.1597\/08-275.1"},{"key":"bibr2-11-123","first-page":"1017","volume-title":"Inborn Errors of Development.","author":"Bamshad M.J.","year":"2004"},{"key":"bibr3-11-123","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.39.8.559"},{"key":"bibr4-11-123","doi-asserted-by":"publisher","DOI":"10.1097\/01.prs.0000263567.62923.fd"},{"key":"bibr5-11-123","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.1972.tb01724.x"},{"key":"bibr6-11-123","doi-asserted-by":"publisher","DOI":"10.1016\/0002-9416(48)90083-9"},{"key":"bibr7-11-123","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.32.9.716"},{"key":"bibr8-11-123","doi-asserted-by":"publisher","DOI":"10.1016\/S0092-8674(00)81646-3"},{"key":"bibr9-11-123","first-page":"38","volume":"41","author":"Chiang T.P.","year":"1974","journal-title":"J Dent Child."},{"key":"bibr10-11-123","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2133.2009.09496.x"},{"key":"bibr11-11-123","volume-title":"Statistical Methods for Medical and Biological Students.","author":"Dahlberg G.","year":"1940"},{"key":"bibr12-11-123","volume-title":"\u00dceber eine sehr Komplicierte Hasenscharte.","author":"Eckoldt J.G.","year":"1804"},{"key":"bibr13-11-123","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.27.3.165"},{"key":"bibr14-11-123","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.38.6.405"},{"key":"bibr15-11-123","doi-asserted-by":"publisher","DOI":"10.1046\/j.1523-1747.2002.19526.x"},{"key":"bibr16-11-123","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.1991.tb03077.x"},{"key":"bibr17-11-123","unstructured":"HennekamR.C.M.KrantzI.D.AllansonJ.E. Gorlin's Syndromes of the Head and Neck. 5th ed. New York: Oxford University Press; 2010: 974\u2013978."},{"key":"bibr18-11-123","first-page":"18","volume":"25","author":"Iizuka T.","year":"1958","journal-title":"J Stomatol Soc Jpn."},{"key":"bibr19-11-123","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.1987.tb10571.x"},{"key":"bibr20-11-123","first-page":"31","volume":"17","author":"Kamiyama T.","year":"1958","journal-title":"J Jpn Orthod Soc."},{"key":"bibr21-11-123","first-page":"731","volume":"25","author":"King N.M.","year":"1994","journal-title":"Quintessence Int."},{"key":"bibr22-11-123","first-page":"85","volume":"9","author":"Komurasaki H.","year":"1997","journal-title":"J Chugoku-Shikoku Orthod."},{"key":"bibr23-11-123","doi-asserted-by":"publisher","DOI":"10.1034\/j.1399-0004.2001.600411.x"},{"key":"bibr24-11-123","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19960614)63:3<472::AID-AJMG11>3.0.CO;2-J"},{"key":"bibr25-11-123","first-page":"436","volume":"39","author":"Masaki F.","year":"1980","journal-title":"J Jpn Orthod Soc."},{"key":"bibr26-11-123","unstructured":"Ministry of Education, Culture, Sports, Science, and Technology, Physical development of students 2002. http:\/\/www.e-stat.go.jp\/SG1\/estat\/List.do?bid=000001016203&cycode=0. Accessed April 10, 2011."},{"key":"bibr27-11-123","unstructured":"Ministry of Health, Labor, and Welfare, National growth survey of Japanese infants and children 2000. http:\/\/www.e-stat.go.jp\/SG1\/estat\/List.do?lid=000001047588. Accessed April 10, 2011."},{"key":"bibr28-11-123","first-page":"109","volume":"84","author":"Nielsen H.G.","year":"1976","journal-title":"Scand J Dent Res."},{"key":"bibr29-11-123","first-page":"192","volume":"8","author":"O'Dwyer M.R.","year":"1984","journal-title":"J Pedod."},{"key":"bibr30-11-123","doi-asserted-by":"publisher","DOI":"10.1086\/301687"},{"key":"bibr31-11-123","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.13.4.281"},{"key":"bibr32-11-123","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320530207"},{"key":"bibr33-11-123","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2004.00287.x"},{"key":"bibr34-11-123","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.31271"},{"key":"bibr35-11-123","doi-asserted-by":"publisher","DOI":"10.1097\/00019605-199604000-00003"},{"key":"bibr36-11-123","doi-asserted-by":"publisher","DOI":"10.1001\/archpedi.1970.02100070104016"},{"key":"bibr37-11-123","first-page":"1","volume":"18","author":"Sakamoto T.","year":"1959","journal-title":"J Jpn Orthod Soc."},{"key":"bibr38-11-123","first-page":"31","volume":"63","author":"Schalk van der Weide Y.","year":"1993","journal-title":"Angle Orthod."},{"key":"bibr39-11-123","first-page":"306","volume":"52","author":"Sekiya T.","year":"1993","journal-title":"J Jpn Orthod Soc."},{"key":"bibr40-11-123","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33768"},{"key":"bibr41-11-123","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-263X.1992.tb00017.x"},{"key":"bibr42-11-123","doi-asserted-by":"publisher","DOI":"10.1086\/323123"},{"key":"bibr43-11-123","doi-asserted-by":"publisher","DOI":"10.1086\/341450"},{"key":"bibr44-11-123","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0b013e3181ebccc5"}],"container-title":["The Cleft Palate Craniofacial Journal"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1597\/11-123","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1597\/11-123","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1597\/11-123","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,28]],"date-time":"2026-04-28T16:17:23Z","timestamp":1777393043000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1597\/11-123"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2013,3]]},"references-count":44,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2013,3]]}},"alternative-id":["10.1597\/11-123"],"URL":"https:\/\/doi.org\/10.1597\/11-123","relation":{},"ISSN":["1055-6656","1545-1569"],"issn-type":[{"value":"1055-6656","type":"print"},{"value":"1545-1569","type":"electronic"}],"subject":[],"published":{"date-parts":[[2013,3]]}}}