{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,26]],"date-time":"2026-06-26T21:48:35Z","timestamp":1782510515907,"version":"3.54.5"},"reference-count":36,"publisher":"SAGE Publications","license":[{"start":{"date-parts":[[2026,6,23]],"date-time":"2026-06-23T00:00:00Z","timestamp":1782172800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,6,23]],"date-time":"2026-06-23T00:00:00Z","timestamp":1782172800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"DOI":"10.13039\/100015902","name":"European Orthodontic Society","doi-asserted-by":"publisher","award":["\u201dVan der Woude syndrome \u2013 craniofacial develop"],"award-info":[{"award-number":["\u201dVan der Woude syndrome \u2013 craniofacial develop"]}],"id":[{"id":"10.13039\/100015902","id-type":"DOI","asserted-by":"publisher"}]},{"name":"The Swedish Freemasons\u2019 Foundation for Children\u2019s Welfare","award":["Van der Woude syndrome (VWS) \u2013 craniofacial deve"],"award-info":[{"award-number":["Van der Woude syndrome (VWS) \u2013 craniofacial deve"]}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["The Cleft Palate Craniofacial Journal"],"abstract":"<jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To study craniofacial growth and hypodontia in individuals born with Van der Woude syndrome (VWS) and compare with matched controls with and without a cleft.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design<\/jats:title>\n                    <jats:p>A multicenter retrospective case-control study.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Setting<\/jats:title>\n                    <jats:p>Individuals from three craniofacial centers in Sweden and from a historical archive of Swedish healthy children were included.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Patients<\/jats:title>\n                    <jats:p>Sixty individuals, with VWS (n\u2009=\u200920), non-syndromic cleft lip and\/or palate (NSCL\/P) (n\u2009=\u200920), and healthy controls (CTR) (n\u2009=\u200920), matched for age, gender and cleft type.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Interventions<\/jats:title>\n                    <jats:p>Two lateral cephalograms per individual, taken at 10 and 16 years of age, were analyzed. Orthopantomograms were used to evaluate hypodontia.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Main Outcome Measures<\/jats:title>\n                    <jats:p>Craniofacial growth followed longitudinally, with the primary variables SNA, SNB, ANB, NL\/ML, and maxillary length. Prevalence of hypodontia was also recorded.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Maxillary length increased 0.3\u2005mm in the VWS group during puberty, compared to 4\u2005mm in the NSCL\/P group and 5.5\u2005mm in the CTR group. Both cleft groups had a retruded maxilla at 16 years of age. The change in vertical jaw relation from 10 to 16 years showed a statistically significant difference between the VWS and the CTR group, with a slightly more posteriorly inclined mandible in the VWS group at 16 years. A statistically significant difference in hypodontia is seen between the cleft groups. In the VWS group, 90% had hypodontia compared to 60% in the NSCL\/P group.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Individuals born with VWS have reduced maxillary growth during puberty and a higher prevalence of hypodontia compared to matched controls in the studied population.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/10556656261463128","type":"journal-article","created":{"date-parts":[[2026,6,23]],"date-time":"2026-06-23T12:20:22Z","timestamp":1782217222000},"update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Craniofacial Growth and Hypodontia in Individuals Born with Van der Woude Syndrome"],"prefix":"10.1177","author":[{"ORCID":"https:\/\/orcid.org\/0009-0006-2921-5461","authenticated-orcid":false,"given":"Malin","family":"Vesterbacka","sequence":"first","affiliation":[{"name":"Karolinska Institutet"},{"name":"Stockholm Craniofacial Team, Department of Maxillofacial surgery, Karolinska University Hospital, Stockholm, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Konstantinos","family":"Parikakis","sequence":"additional","affiliation":[{"name":"Karolinska Institutet"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4588-3543","authenticated-orcid":false,"given":"Niels","family":"Ganzer","sequence":"additional","affiliation":[{"name":"Karolinska Institutet"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9905-4046","authenticated-orcid":false,"given":"Agneta","family":"Karsten","sequence":"additional","affiliation":[{"name":"Karolinska Institutet"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1734-2990","authenticated-orcid":false,"given":"Marie","family":"Pegelow","sequence":"additional","affiliation":[{"name":"Karolinska Institutet"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2026,6,23]]},"reference":[{"key":"e_1_3_3_2_2","doi-asserted-by":"publisher","DOI":"10.1093\/oso\/9780195118612.001.0001"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320050203"},{"issue":"1","key":"e_1_3_3_4_2","first-page":"52","article-title":"The penetrance and variable expression of the Van der Woude syndrome: Implications for genetic counseling","volume":"17","author":"Shpintzen RJ","year":"1980","unstructured":"Shpintzen RJ, Goldberg RB, Sidoti EJ. The penetrance and variable expression of the Van der Woude syndrome: Implications for genetic counseling. Cleft Palate J. 1980;17(1):52-57.","journal-title":"Cleft Palate J"},{"key":"e_1_3_3_5_2","doi-asserted-by":"publisher","DOI":"10.1038\/ng985"},{"key":"e_1_3_3_6_2","doi-asserted-by":"publisher","DOI":"10.1097\/GIM.0b013e318197a49a"},{"key":"e_1_3_3_7_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2013.11.009"},{"issue":"2","key":"e_1_3_3_8_2","first-page":"244","article-title":"Fistula labii inferioris congenita and its association with cleft lip and palate","volume":"6","author":"Van Der Woude A","year":"1954","unstructured":"Van Der Woude A. Fistula labii inferioris congenita and its association with cleft lip and palate. Am J Hum Genet. 1954;6(2):244-256.","journal-title":"Am J Hum Genet"},{"issue":"2","key":"e_1_3_3_9_2","first-page":"158","article-title":"Correlations between microforms of the Van Der Woude syndrome and cleft palate","volume":"20","author":"Ranta R","year":"1983","unstructured":"Ranta R, Rintala AE. Correlations between microforms of the Van Der Woude syndrome and cleft palate. Cleft Palate J. 1983;20(2):158-162.","journal-title":"Cleft Palate J"},{"key":"e_1_3_3_10_2","doi-asserted-by":"publisher","DOI":"10.1093\/ejo\/26.1.17"},{"key":"e_1_3_3_11_2","doi-asserted-by":"publisher","DOI":"10.1186\/s12903-021-01420-7"},{"key":"e_1_3_3_12_2","doi-asserted-by":"publisher","DOI":"10.1597\/15-186"},{"key":"e_1_3_3_13_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00784-022-04540-8"},{"key":"e_1_3_3_14_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0528.2004.00158.x"},{"key":"e_1_3_3_15_2","doi-asserted-by":"publisher","DOI":"10.1016\/0889-5406(86)90022-3"},{"issue":"3","key":"e_1_3_3_16_2","article-title":"Tooth anomalies associated with congenital sinuses of the lower lip and cleft lip\/palate","volume":"52","author":"Ranta R","year":"1982","unstructured":"Ranta R, Rintala A. Tooth anomalies associated with congenital sinuses of the lower lip and cleft lip\/palate. Angle Orthod 1982;52(3):212-221.","journal-title":"Angle Orthod"},{"key":"e_1_3_3_17_2","doi-asserted-by":"publisher","DOI":"10.1597\/04-028.1"},{"key":"e_1_3_3_18_2","doi-asserted-by":"publisher","DOI":"10.1093\/ejo\/cjaa082"},{"key":"e_1_3_3_19_2","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0b013e3181ec6aad"},{"key":"e_1_3_3_20_2","doi-asserted-by":"publisher","DOI":"10.1597\/06-075.1"},{"key":"e_1_3_3_21_2","doi-asserted-by":"publisher","DOI":"10.3109\/2000656X.2014.992904"},{"key":"e_1_3_3_22_2","doi-asserted-by":"publisher","DOI":"10.1597\/1545-1569_2002_039_0219_acsofg_2.0.co_2"},{"key":"e_1_3_3_23_2","doi-asserted-by":"publisher","DOI":"10.1111\/odi.14187"},{"issue":"1","key":"e_1_3_3_24_2","first-page":"12","article-title":"The prevalence of malocclusion of Swedish schoolchildren","volume":"81","author":"Thilander B","year":"1973","unstructured":"Thilander B, Myrberg N. The prevalence of malocclusion of Swedish schoolchildren. Scand J Dent Res. 1973;81(1):12-21.","journal-title":"Scand J Dent Res"},{"key":"e_1_3_3_25_2","first-page":"219","article-title":"Roentegencephalometric standards for the facial skeleton and soft tissue profile of Swedish children and young adults. II. Comparisons with earlier Scandinavian normative data","volume":"15","author":"Thilander B","year":"1982","unstructured":"Thilander B, Persson M, Skagius S. Roentegencephalometric standards for the facial skeleton and soft tissue profile of Swedish children and young adults. II. Comparisons with earlier Scandinavian normative data. Swed Dent J Suppl. 1982;15:219-228.","journal-title":"Swed Dent J Suppl"},{"key":"e_1_3_3_26_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajpa.1330440306"},{"issue":"10","key":"e_1_3_3_27_2","first-page":"744","article-title":"Soft tissue cephalometric analysis for orthognathic surgery","volume":"38","author":"Legan H","year":"1980","unstructured":"Legan H, Burstone CJ. Soft tissue cephalometric analysis for orthognathic surgery. J Oral Surg. 1980;38(10):744-751.","journal-title":"J Oral Surg"},{"key":"e_1_3_3_28_2","volume-title":"Statistical Methods for Medical and Biological Students","author":"Dahlberg G","year":"1940","unstructured":"Dahlberg G. Statistical Methods for Medical and Biological Students. Interscience Publications; 1940."},{"key":"e_1_3_3_29_2","volume-title":"R: A Language and Environment for Statistical Computing","author":"R Core Team","year":"2024","unstructured":"R Core Team. R: A Language and Environment for Statistical Computing. R Foundation for Statistical Computing; 2024, https:\/\/www.R-project.org\/."},{"issue":"2","key":"e_1_3_3_30_2","first-page":"65","article-title":"A simple sequentially rejective multiple test procedure","volume":"6","author":"Holm S","year":"1979","unstructured":"Holm S. A simple sequentially rejective multiple test procedure. Scan J Stat. 1979;6(2):65-70.","journal-title":"Scan J Stat"},{"key":"e_1_3_3_31_2","doi-asserted-by":"publisher","DOI":"10.1597\/14-132"},{"key":"e_1_3_3_32_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jcms.2017.01.016"},{"key":"e_1_3_3_33_2","doi-asserted-by":"publisher","DOI":"10.1177\/10556656211013690"},{"key":"e_1_3_3_34_2","doi-asserted-by":"publisher","DOI":"10.1097\/PRS.0000000000001694"},{"issue":"1","key":"e_1_3_3_35_2","first-page":"24","article-title":"Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: Maxillary growth at eight years of age","volume":"42","author":"K\u00fcseler A","year":"2020","unstructured":"K\u00fcseler A, M\u00f8lsted K, Marcusson A, Heli\u00f6vaara A, Karsten A, Bellardie H, S\u00e6le P, Brinck E, Skaare P, Rizell S, et al. Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: Maxillary growth at eight years of age. Eur J Orthod. 2020;42(1):24-29.","journal-title":"Eur J Orthod"},{"key":"e_1_3_3_36_2","doi-asserted-by":"publisher","DOI":"10.1097\/PRS.0000000000000779"},{"key":"e_1_3_3_37_2","doi-asserted-by":"publisher","DOI":"10.1093\/ejo\/cjab007"}],"container-title":["The Cleft Palate Craniofacial Journal"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/10556656261463128","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/10556656261463128","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/10556656261463128","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,26]],"date-time":"2026-06-26T21:20:27Z","timestamp":1782508827000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/10556656261463128"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,6,23]]},"references-count":36,"alternative-id":["10.1177\/10556656261463128"],"URL":"https:\/\/doi.org\/10.1177\/10556656261463128","relation":{},"ISSN":["1055-6656","1545-1569"],"issn-type":[{"value":"1055-6656","type":"print"},{"value":"1545-1569","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,6,23]]},"article-number":"10556656261463128"}}