{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,24]],"date-time":"2026-06-24T00:54:47Z","timestamp":1782262487019,"version":"3.54.5"},"reference-count":29,"publisher":"SAGE Publications","issue":"6","license":[{"start":{"date-parts":[[2015,11,1]],"date-time":"2015-11-01T00:00:00Z","timestamp":1446336000000},"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":[[2015,11]]},"abstract":"<jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To summarize the clinical characteristics and surgical and speech outcomes for patients with Van der Woude\/popliteal pterygium syndromes (VWS\/PPS) and to compare them with a historic cohort of patients with nonsyndromic cleft lip\/cleft palate (CL\/P)<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design<\/jats:title>\n                    <jats:p>Retrospective chart review.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Setting<\/jats:title>\n                    <jats:p>Tertiary care center.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Patients<\/jats:title>\n                    <jats:p>All patients with VWS\/PPS seen at Boston Children's Hospital from 1979 to 2012: 28 patients with VWS (n = 21)\/PPS (n = 7) whose mean age was 17.3 \u00b1 10.4 years, including 18 females (64%) and 10 males (36%); 18 patients (64%) had a family history of VWS\/PPS.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Main Outcome Measures<\/jats:title>\n                    <jats:p>Cleft type, operative procedures, speech, and midfacial growth. Data were compared with historic cohorts of patients with nonsyndromic CL\/P treated at one tertiary care center.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>There were 24 patients (86%) with CP\u00b1L, Veau types I (n = 4,17%), II (n = 4, 17%), III (n = 5, 21%), and IV (n = 11, 46%). Nine patients (38%) had palatal fistula after palatoplasty. Fourteen of 23 (61%) patients with CL\/P age 5 years or older had midfacial retrusion, and 10 (43%) required a pharyngeal flap for velopharyngeal insufficiency. Fisher's exact test demonstrated higher frequencies of Veau type IV CP\u00b1L ( P = .0016), bilateral CL\u00b1P ( P = .0001), and complete CL\u00b1P ( P &lt; .0001) in VWS\/PPS compared with nonsyndromic patients. Incidences of midfacial retrusion ( P = .0001), palatal fistula ( P &lt; .0001), and need for pharyngeal flap ( P = .0014) were significantly greater in patients with VWS\/PPS.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Patients with VWS\/PPS have more severe forms of labiopalatal clefting and higher incidences of midfacial retrusion, palatal fistula, and velopharyngeal insufficiency following primary repair as compared with nonsyndromic CL\/P.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1597\/14-132","type":"journal-article","created":{"date-parts":[[2014,12,8]],"date-time":"2014-12-08T11:03:13Z","timestamp":1418036593000},"page":"676-681","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":12,"title":["Anatomic Severity, Midfacial Growth, and Speech Outcomes in Van der Woude\/Popliteal Pterygium Syndromes Compared to Nonsyndromic Cleft Lip\/Palate"],"prefix":"10.1177","volume":"52","author":[{"given":"Jeffrey B.","family":"Reardon","sequence":"first","affiliation":[{"name":"Harvard School of Dental Medicine, Boston, Massachusetts."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katherine A.","family":"Brustowicz","sequence":"additional","affiliation":[{"name":"Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, Massachusetts."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eileen M.","family":"Marrinan","sequence":"additional","affiliation":[{"name":"Central New York Cleft and Craniofacial Center, Upstate University Hospital, Syracuse, New York."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John B.","family":"Mulliken","sequence":"additional","affiliation":[{"name":"Harvard Medical School, Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, Massachusetts."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bonnie L.","family":"Padwa","sequence":"additional","affiliation":[{"name":"Oral and Maxillofacial Surgery, Harvard School of Dental Medicine, Department of Plastic and Oral Surgery, Boston Children's Hospital, Boston, Massachusetts."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2018,1,3]]},"reference":[{"key":"bibr1-14-132","first-page":"181","volume":"5","author":"Burdick A.B.","year":"1985","journal-title":"J Craniofac Genet Dev Biol."},{"key":"bibr2-14-132","unstructured":"DurdaK.M. SchutteB.C. MurrayJ.C.\n                      IRF6\n                      -related disorders. In: PagonR.A. BirdT.D. DolanC.R., eds. GeneReviews. 2011. Available at http:\/\/www.ncbi.nlm.nih.gov\/books\/NBK1407\/. Accessed December 23, 2011."},{"key":"bibr3-14-132","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.27.5.320"},{"key":"bibr4-14-132","doi-asserted-by":"publisher","DOI":"10.1597\/06-075.1"},{"key":"bibr5-14-132","doi-asserted-by":"publisher","DOI":"10.1597\/06-094.1"},{"key":"bibr6-14-132","doi-asserted-by":"publisher","DOI":"10.1097\/00006534-199803000-00007"},{"key":"bibr7-14-132","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320050203"},{"key":"bibr8-14-132","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0b013e3181ec6aad"},{"key":"bibr9-14-132","doi-asserted-by":"publisher","DOI":"10.1597\/1545-1569_2002_039_0219_acsofg_2.0.co_2"},{"key":"bibr10-14-132","unstructured":"KniffinC.L. O'NeillM.J.F. SobreiraN. WrightM.J. McKusickV.A. Popliteal pterygium syndrome (#119500).\n                      Online Mendelian Inheritance in Man.\n                      Available at: http:\/\/us-east.omim.org\/entry\/119500. Accessed December 23, 2011."},{"key":"bibr11-14-132","doi-asserted-by":"publisher","DOI":"10.1038\/ng985"},{"key":"bibr12-14-132","doi-asserted-by":"publisher","DOI":"10.1111\/j.1834-7819.2009.01178.x"},{"key":"bibr13-14-132","first-page":"888","volume":"36","author":"Lees M.M.","year":"1999","journal-title":"J Med Genet."},{"key":"bibr14-14-132","doi-asserted-by":"publisher","DOI":"10.1597\/06-064"},{"key":"bibr15-14-132","volume-title":"Audio Seminars in Speech Pathology: Velopharyngeal Incompetence.","author":"McWilliams B.J.","year":"1979"},{"key":"bibr16-14-132","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(09)60695-4"},{"key":"bibr17-14-132","doi-asserted-by":"publisher","DOI":"10.1097\/00006534-199910000-00003"},{"key":"bibr18-14-132","doi-asserted-by":"publisher","DOI":"10.1097\/00001665-200309000-00003"},{"key":"bibr19-14-132","doi-asserted-by":"publisher","DOI":"10.1597\/04-028.1"},{"key":"bibr20-14-132","unstructured":"O'NeillM.J.F. SobreiraN. KniffinC.L. WrightM.J. PetersenM.B. RasmussenS.A. LoW.H.Y. SmithM. McKusickV.A. Van der Woude syndrome 1 (#119300).\n                      Online Mendelian Inheritance in Man.\n                      Available at: http:\/\/us-east.omim.org\/entry\/119300. Accessed December 23, 2011."},{"key":"bibr21-14-132","doi-asserted-by":"publisher","DOI":"10.1597\/1545-1569_1997_034_0261_rbllfa_2.3.co_2"},{"key":"bibr22-14-132","doi-asserted-by":"publisher","DOI":"10.1597\/11-220"},{"key":"bibr23-14-132","first-page":"52","volume":"17","author":"Shprintzen R.J.","year":"1980","journal-title":"Cleft Palate J."},{"key":"bibr24-14-132","doi-asserted-by":"publisher","DOI":"10.1597\/06-204.1"},{"key":"bibr25-14-132","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0b013e318192801b"},{"key":"bibr26-14-132","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19980113)75:2<126::AID-AJMG2>3.0.CO;2-R"},{"key":"bibr27-14-132","volume-title":"Division Palatine.","author":"Veau V.","year":"1931"},{"key":"bibr28-14-132","doi-asserted-by":"publisher","DOI":"10.1097\/00006534-199705010-00008"},{"key":"bibr29-14-132","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa032909"}],"container-title":["The Cleft Palate Craniofacial Journal"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1597\/14-132","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1597\/14-132","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1597\/14-132","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,28]],"date-time":"2026-04-28T16:18:39Z","timestamp":1777393119000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1597\/14-132"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,11]]},"references-count":29,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2015,11]]}},"alternative-id":["10.1597\/14-132"],"URL":"https:\/\/doi.org\/10.1597\/14-132","relation":{},"ISSN":["1055-6656","1545-1569"],"issn-type":[{"value":"1055-6656","type":"print"},{"value":"1545-1569","type":"electronic"}],"subject":[],"published":{"date-parts":[[2015,11]]}}}