{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,28]],"date-time":"2026-05-28T00:29:02Z","timestamp":1779928142239,"version":"3.53.1"},"reference-count":25,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2021,6,16]],"date-time":"2021-06-16T00:00:00Z","timestamp":1623801600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["The Laryngoscope"],"published-print":{"date-parts":[[2022,1]]},"abstract":"<jats:sec>\n                    <jats:title>Objectives\/Hypothesis<\/jats:title>\n                    <jats:p>\n                      A tracheal cartilaginous sleeve (TCS) is a rare anomaly characterized by anterior fusion of tracheal cartilages. TCS is associated with syndromic craniosynostoses including Apert, Crouzon and Pfeiffer syndromes and\n                      <jats:italic>FGFR2<\/jats:italic>\n                      ,\n                      <jats:italic>FGFR3<\/jats:italic>\n                      , and\n                      <jats:italic>TWIST1<\/jats:italic>\n                      variants. This study presents a 30\u2010year review of patients with syndromic craniosynostosis and TCS and describes diagnostic methods, genetic variants, surgical interventions, and long\u2010term outcomes.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Study Design<\/jats:title>\n                    <jats:p>Retrospective, single\u2010institution review.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      This review included patients with syndromic craniosynostosis and TCS treated at Seattle Children's Hospital from 1990 to 2020. Tracheostomy, genetic variants, and additional surgery were primary measures. Fisher's exact test compared need for tracheostomy in patients with proposed high\u2010risk (\n                      <jats:italic>FGFR2<\/jats:italic>\n                      p.W290 or\n                      <jats:italic>FGFR2<\/jats:italic>\n                      p.C342) versus low\u2010risk genetic variants.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Thirty patients with TCS were identified. Average age at diagnosis was 12\u2009months (range 2\u2010weeks to 7.9\u2010years; standard deviation 19.8\u2009months). Syndromes included Pfeiffer (37%), Apert (37%), and Crouzon (26%). Severe obstructive sleep apnea was present in 76% of patients. Tracheostomy was performed in 17 patients (57%); five were successfully decannulated. Additional interventions included adenotonsillectomy (57%), nasal (20%), laryngeal (17%), and craniofacial skeletal surgery (87%). All patients with Pfeiffer syndrome and\n                      <jats:italic>FGFR2<\/jats:italic>\n                      p.W290C variants and 83% of patients with\n                      <jats:italic>FGFR2<\/jats:italic>\n                      p.C342 variants required tracheostomy, differing from other variants (\n                      <jats:italic>P<\/jats:italic>\n                      \u00a0=\u00a0.02, odds ratio 33, 95% confidence interval 1.56\u2013697.96). One patient (3%) died.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      TCS contributes to multilevel airway obstruction in patients with syndromic craniosynostosis. Genetic testing in patients with\n                      <jats:italic>FGFR2<\/jats:italic>\n                      \u2010related syndromic craniosynostoses may identify those at risk of TCS and facilitate early intervention. A better understanding of this patient population may foster individualized airway management strategies and improve outcomes.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Level of Evidence<\/jats:title>\n                    <jats:p>\n                      4\n                      <jats:italic>Laryngoscope<\/jats:italic>\n                      , 132:215\u2013221, 2022\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/lary.29692","type":"journal-article","created":{"date-parts":[[2021,6,16]],"date-time":"2021-06-16T13:18:58Z","timestamp":1623849538000},"page":"215-221","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Complex Airway Management in Patients with Tracheal Cartilaginous Sleeves"],"prefix":"10.1002","volume":"132","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1442-3819","authenticated-orcid":false,"given":"Anisha R.","family":"Noble","sequence":"first","affiliation":[{"name":"Department of Otolaryngology \u2013 Head and Neck Surgery University of Washington School of Medicine  Seattle Washington U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael L.","family":"Cunningham","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Craniofacial Center Seattle Children's Hospital  Seattle Washington U.S.A."},{"name":"Department of Pediatrics, Division of Craniofacial Medicine University of Washington School of Medicine  Seattle Washington U.S.A."},{"name":"Seattle Children's Research Division Seattle Children's Research Institute  Seattle Washington U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2297-6376","authenticated-orcid":false,"given":"Austin","family":"Lam","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology \u2013 Head and Neck Surgery University of Washington School of Medicine  Seattle Washington U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tara L.","family":"Wenger","sequence":"additional","affiliation":[{"name":"Division of Genetic Medicine University of Washington School of Medicine  Seattle Washington U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathleen C.","family":"Sie","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology \u2013 Head and Neck Surgery University of Washington School of Medicine  Seattle Washington U.S.A."},{"name":"Department of Pediatrics, Craniofacial Center Seattle Children's Hospital  Seattle Washington U.S.A."},{"name":"Seattle Children's Research Division Seattle Children's Research Institute  Seattle Washington U.S.A."},{"name":"Division of Pediatric Otolaryngology \u2013 Head and Neck Surgery Seattle Children's Hospital  Seattle Washington U.S.A."}],"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 \u2013 Head and Neck Surgery University of Washington School of Medicine  Seattle Washington U.S.A."},{"name":"Department of Pediatrics, Craniofacial Center Seattle Children's Hospital  Seattle Washington U.S.A."},{"name":"Seattle Children's Research Division Seattle Children's Research Institute  Seattle Washington U.S.A."},{"name":"Division of Pediatric Otolaryngology \u2013 Head and Neck Surgery Seattle Children's Hospital  Seattle Washington U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John P.","family":"Dahl","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology \u2013 Head and Neck Surgery University of Washington School of Medicine  Seattle Washington U.S.A."},{"name":"Department of Pediatrics, Craniofacial Center Seattle Children's Hospital  Seattle Washington U.S.A."},{"name":"Seattle Children's Research Division Seattle Children's Research Institute  Seattle Washington U.S.A."},{"name":"Division of Pediatric Otolaryngology \u2013 Head and Neck Surgery Seattle Children's Hospital  Seattle Washington U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2021,6,16]]},"reference":[{"key":"e_1_2_6_2_1","volume-title":"GeneReviews(\u00ae)","author":"Wenger 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