{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,28]],"date-time":"2026-07-28T04:15:59Z","timestamp":1785212159147,"version":"3.55.0"},"reference-count":29,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2023,1,25]],"date-time":"2023-01-25T00:00:00Z","timestamp":1674604800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["The Laryngoscope"],"published-print":{"date-parts":[[2023,10]]},"abstract":"<jats:sec><jats:title>Objective<\/jats:title><jats:p>Patients with 22q11.2 deletion syndrome (22q11DelS) often present with velopharyngeal dysfunction (VPD). VPD in patients with 22q11DelS is multifactorial beyond velopharyngeal insufficiency (VPI) alone, and differences in surgical outcomes are poorly understood. Our objective was to determine whether patients with 22q11DelS have an increased risk for persistent VPI after sphincter pharyngoplasty compared to patients without 22q11DelS.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We completed a retrospective cohort study of patients with 22q11DelS undergoing sphincter pharyngoplasty between 1995 and 2019 using a VPD clinic database. Patients with 22q11DelS were compared to a cohort of 2:1 frequency\u2010matched (age, degree of velopharyngeal closure) patients without 22q11DelS. Variables included patient characteristics, surgical history, perceptual speech evaluation, and degree of closure on nasopharyngoscopic evaluations. Primary outcomes included postoperative VPI severity and hypernasality. Speech and nasopharyngoscopic characteristics were compared using Fisher's exact test. Postoperative VPI severity and hypernasality were compared between groups via relative risks (RR) from mixed effects Poisson regression models, with random effects of age and velopharyngeal closure.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>134 patients (51 22q11DelS, 83 matched) were included, with mean age of 7.3\u2009years (standard deviation 3.0) and 50% male. Cohorts had similar preoperative speech characteristics and nasopharyngoscopic findings. Patients with 22q11DelS had similar postoperative VP function as patients without 22q11DelS (RR 0.85, CI 0.46\u20131.57 for VPI severity, RR 0.83, CI 0.45\u20131.53 for hypernasality). Even after adjusting by preoperative variables, no differences were seen between both groups.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Matched for age and pre\u2010operative velopharyngeal closure, patients with and without 22q11DelS and VPI had similar benefits after sphincter pharyngoplasty.<\/jats:p><\/jats:sec><jats:sec><jats:title>Level of Evidence<\/jats:title><jats:p>Non\u2010randomized controlled cohort study, 3 <jats:italic>Laryngoscope<\/jats:italic>, 133:2813\u20132820, 2023<\/jats:p><\/jats:sec>","DOI":"10.1002\/lary.30579","type":"journal-article","created":{"date-parts":[[2023,1,25]],"date-time":"2023-01-25T09:28:49Z","timestamp":1674638929000},"page":"2813-2820","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Sphincter Pharyngoplasty for Velopharyngeal Dysfunction: Impact of 22q11.2 Deletion Syndrome"],"prefix":"10.1002","volume":"133","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6857-2086","authenticated-orcid":false,"given":"Prasanth","family":"Pattisapu","sequence":"first","affiliation":[{"name":"Department of Otolaryngology\u2013Head &amp; Neck Surgery Nationwide Children's Hospital and The Ohio State University  Columbus Ohio USA"},{"name":"Center for Surgical Outcomes Research and Center for Health Equity and Outcomes Research Abigail Wexner Research Institute, Nationwide Children's Hospital  Columbus Ohio USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6017-4529","authenticated-orcid":false,"given":"Sara","family":"Kinter","sequence":"additional","affiliation":[{"name":"Childhood Communication Center Seattle Children's Hospital  Seattle Washington USA"},{"name":"Division of Craniofacial Medicine, Department of Pediatrics University of Washington  Seattle Washington USA"},{"name":"Center for Child Health, Behavior &amp; Development Seattle Children's Hospital  Seattle Washington USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5308-527X","authenticated-orcid":false,"given":"Randall A.","family":"Bly","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery University of Washington  Seattle Washington USA"},{"name":"Division of Pediatric Otolaryngology Seattle Children's Hospital  Seattle Washington USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2728-3576","authenticated-orcid":false,"given":"John P.","family":"Dahl","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery University of Washington  Seattle Washington USA"},{"name":"Division of Pediatric Otolaryngology Seattle Children's Hospital  Seattle Washington USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0181-3997","authenticated-orcid":false,"given":"Jonathan A.","family":"Perkins","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery University of Washington  Seattle Washington USA"},{"name":"Division of Pediatric Otolaryngology Seattle Children's Hospital  Seattle Washington USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xing","family":"Wang","sequence":"additional","affiliation":[{"name":"Center for Clinical and Translational Research Seattle Children's Hospital  Seattle Washington USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathleen C. Y.","family":"Sie","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery University of Washington  Seattle Washington USA"},{"name":"Division of Pediatric Otolaryngology Seattle Children's Hospital  Seattle Washington USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,1,25]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrdp.2015.71"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2011.02.039"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2014.175"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1044\/2018_AJSLP-18-0037"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1080\/02699206.2019.1595732"},{"issue":"1","key":"e_1_2_7_7_1","first-page":"68","article-title":"Coming to terms with VPI: a response to Loney and Bloem","volume":"26","author":"Trost\u2010Cardamone JE","year":"1989","journal-title":"Cleft Palate J"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.61152"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bjps.2008.02.007"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1044\/2019_AJSLP-16-0147"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1597\/1545-1569_1995_032_0179_mothv_2.3.co_2"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1177\/0003489414554943"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2018.10.016"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1001\/archotol.134.11.1159"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1001\/archoto.2009.64"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2015.10.007"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2016.06.044"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.3389\/fpsyg.2017.01413"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/pds.3263"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-2007-979282"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1597\/14-027"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1597\/1545-1569(1990)027<0337:sftron>2.3.co;2"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.prs.0000169694.29082.69"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0b013e31822e624f"},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1054\/bjps.1999.3168"},{"key":"e_1_2_7_26_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.prs.0000206377.14083.ce"},{"key":"e_1_2_7_27_1","doi-asserted-by":"publisher","DOI":"10.1597\/05-136.1"},{"key":"e_1_2_7_28_1","doi-asserted-by":"publisher","DOI":"10.1159\/000200768"},{"key":"e_1_2_7_29_1","doi-asserted-by":"publisher","DOI":"10.1597\/04-141.1"},{"key":"e_1_2_7_30_1","doi-asserted-by":"publisher","DOI":"10.1044\/jslhr.4304.989"}],"container-title":["The Laryngoscope"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/lary.30579","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/lary.30579","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/lary.30579","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,19]],"date-time":"2023-09-19T18:58:16Z","timestamp":1695149896000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/lary.30579"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,1,25]]},"references-count":29,"journal-issue":{"issue":"10","published-print":{"date-parts":[[2023,10]]}},"alternative-id":["10.1002\/lary.30579"],"URL":"https:\/\/doi.org\/10.1002\/lary.30579","archive":["Portico"],"relation":{},"ISSN":["0023-852X","1531-4995"],"issn-type":[{"value":"0023-852X","type":"print"},{"value":"1531-4995","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,1,25]]},"assertion":[{"value":"2022-06-23","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-01-05","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-01-25","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}