{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,14]],"date-time":"2026-02-14T01:48:01Z","timestamp":1771033681095,"version":"3.50.1"},"reference-count":25,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2022,1,10]],"date-time":"2022-01-10T00:00:00Z","timestamp":1641772800000},"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":["Orthod Craniofacial Res"],"published-print":{"date-parts":[[2022,11]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Aim<\/jats:title><jats:p>To assess the upper airway (UA) morphology in patients with pycnodysostosis with a 3D analysis, compare results with normative data and investigate the correlation of the total volume (TV) with other UA morphology variables.<\/jats:p><\/jats:sec><jats:sec><jats:title>Materials and methods<\/jats:title><jats:p>Cone beam computed tomography (CBCT) images of eight Danish patients with pycnodysostosis (4 males and 4 females with a mean age of 31.8\u00a0years, SD: 16.3\u00a0years) were analyzed using Mimics<jats:sup>\u00ae<\/jats:sup> (Materialise<jats:sup>\u00ae<\/jats:sup>) and compared with a sex\u2010 and age\u2010matched control group (6 males and 8 females with a mean age of 33.6\u00a0years, SD: 18.6\u00a0years).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The distance from the tip of the epiglottis (E) to the Frankfurt horizontal plane (Fp) was significantly shorter in the pycnodysostosis group (<jats:italic>P<\/jats:italic>\u00a0&lt;\u00a0.042). Regarding the cross\u2010sectional measurements, at the \u2018maximum constriction\u2019 (<jats:italic>P<\/jats:italic>\u00a0&lt;\u00a0.005), the \u2018upper airway limit\u2019 (<jats:italic>P<\/jats:italic>\u00a0&lt;\u00a0.001) and the \u2018lower airway limit\u2019 (<jats:italic>P<\/jats:italic>\u00a0&lt;\u00a0.035) cross\u2010sections were significantly smaller in the pycnodysostosis group. The volumes \u2018nasopharynx\u2019 (<jats:italic>P<\/jats:italic>\u00a0&lt;\u00a0.002) and \u2018total airway\u2019 (TV) (<jats:italic>P<\/jats:italic>\u00a0&lt;\u00a0.01) were also significantly smaller.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Patients with pycnodysostosis have a reduced total airway as well as nasopharyngeal volume compared with matched controls. Additionally, they have a reduced cross\u2010sectional area in the upper and lower borders of the UA, and the area of maximum constriction is also reduced. These factors might explain the high prevalence of obstructive sleep apnoea in pycnodysostosis.<\/jats:p><jats:p>Total airway is positively correlated with total length and cross\u2010sections at all levels including the maximum constriction area as well as the anteroposterior dimension at the upper and lower airway borders.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ocr.12561","type":"journal-article","created":{"date-parts":[[2021,12,28]],"date-time":"2021-12-28T17:30:41Z","timestamp":1640712641000},"page":"494-501","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Obstructive sleep apnoea in pycnodysostosis: A three\u2010dimensional upper airway analysis"],"prefix":"10.1111","volume":"25","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4995-1934","authenticated-orcid":false,"given":"Nikolaos","family":"Ferlias","sequence":"first","affiliation":[{"name":"Section of Orthodontics Department of Dentistry and Oral Health Aarhus University  Aarhus Denmark"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hans","family":"Gj\u00f8rup","sequence":"additional","affiliation":[{"name":"Department of Oral and Maxillofacial Surgery Center for Oral Health in Rare Diseases Aarhus University Hospital  Aarhus Denmark"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mia Aagaard","family":"Doherty","sequence":"additional","affiliation":[{"name":"Department of Paediatrics Nordsj\u00e6llands Hospital  Hillerod Denmark"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Annette","family":"Haagerup","sequence":"additional","affiliation":[{"name":"Faculty of Health NIDO Denmark, Hospital Unit West and Institute of Clinical Medicine Aarhus University  Aarhus Denmark"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Thomas Klit","family":"Pedersen","sequence":"additional","affiliation":[{"name":"Section of Orthodontics Department of Dentistry and Oral Health Aarhus University  Aarhus Denmark"},{"name":"Department of Oral and Maxillofacial Surgery Aarhus University Hospital  Aarhus Denmark"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2022,1,10]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.273.5279.1236"},{"issue":"3","key":"e_1_2_9_3_1","first-page":"204","article-title":"Pycnodysostosis: orofacial manifestations in two pediatric patients","volume":"20","author":"O\u2019connell AC","year":"1998","journal-title":"Pediatr Dent"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.36557"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0278-2391(96)90131-7"},{"issue":"2","key":"e_1_2_9_6_1","first-page":"43","article-title":"Short stature revealing a pycnodysostosis: a case report","volume":"6","author":"Hayat A","year":"2016","journal-title":"J Orthop Case Resports"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/ocr.12479"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.61393"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijom.2004.10.024"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijom.2010.09.007"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1513\/pats.200709\u2010155MG"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1136\/bcr\u20102013\u2010200590"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.5582\/irdr.2014.01014"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF00973678"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2017.02.009"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejmg.2021.104135"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/ocr.12053"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.2319\/083117\u2010587.1"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1601\u20106343.2010.01482.x"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1148\/radiology.171.3.2717750"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1259\/dmfr.20170042"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.sodo.2009.02.001"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tripleo.2009.05.026"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1164\/ajrccm.158.4.9712063"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1017\/S0022215100142136"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/lary.25589"}],"container-title":["Orthodontics &amp; 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