{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T17:26:23Z","timestamp":1787765183454,"version":"build-2784847793"},"reference-count":36,"publisher":"Oxford University Press (OUP)","issue":"5","license":[{"start":{"date-parts":[[2022,12,25]],"date-time":"2022-12-25T00:00:00Z","timestamp":1671926400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,5,10]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Study objectives<\/jats:title>\n                  <jats:p>To investigate neurocognitive and behavioral outcomes at primary school age in relation to obstructive sleep apnea (OSA) in children with Robin sequence (RS) treated with the Tuebingen palatal plate in infancy and to assess the impact of OSA in these patients.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>Forty-two primary school-aged children (n = 21 with RS, n = 21 age- and sex-matched controls) underwent polysomnography, intelligence testing (\u201cWechsler Intelligence Scale for Children\u2014Fifth Edition\u201d [WISC-V]), and anthropometrics. Families completed a 7-day sleep diary and questionnaires on sleep and behavior (Children\u2019s Sleep Habits Questionnaire [CSHQ] and the Child Behavior Checklist [CBCL]).<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>In children with RS (17 non-syndromic, four syndromic; median age 9.7 [8.5\u201310.8] years), the obstructive apnea-hypopnea index (OAHI) was significantly higher than in controls (1.3 [0.4\u20132.7]\/h vs. 0.4 [0.1\u20130.6]\/h). Two syndromic children with RS were already on nocturnal respiratory support for OSA prior to our study, and one non-syndromic child was diagnosed with severe OSA (OAHI 57\/h) despite an unremarkable medical history and questionnaire. The overall intelligence quotient in children with RS was within the normal range and did not differ between children with RS and healthy peers (102 vs. 108, p = .05). However, children with RS had values in the at-risk clinical range for externalizing behavior.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>These children with RS showed an increased risk of OSA and behavioral problems, suggesting regular screening for OSA throughout childhood. Neurocognitive scores in children with RS were within the normal range after adequate treatment of OSA during infancy.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1093\/sleep\/zsac317","type":"journal-article","created":{"date-parts":[[2022,12,25]],"date-time":"2022-12-25T20:51:31Z","timestamp":1672001491000},"source":"Crossref","is-referenced-by-count":20,"title":["Sleep and neurocognitive outcome in primary school children with Robin Sequence"],"prefix":"10.1093","volume":"46","author":[{"given":"Cornelia","family":"Wiechers","sequence":"first","affiliation":[{"name":"Department of Neonatology, Tuebingen University Hospital , Tuebingen , Germany"},{"name":"Interdisciplinary Center for Craniofacial Malformations, Tuebingen University Hospital , Tuebingen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Simone","family":"Uhlig","sequence":"additional","affiliation":[{"name":"Department of Neonatology, Tuebingen University Hospital , Tuebingen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anette","family":"Poets","sequence":"additional","affiliation":[{"name":"Department of Neonatology, Tuebingen University Hospital , Tuebingen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christina","family":"Weise","sequence":"additional","affiliation":[{"name":"Interdisciplinary Center for Craniofacial Malformations, Tuebingen University Hospital , Tuebingen , Germany"},{"name":"Department of Orthodontics, Tuebingen University Hospital , Tuebingen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andreas","family":"Naros","sequence":"additional","affiliation":[{"name":"Interdisciplinary Center for Craniofacial Malformations, Tuebingen University Hospital , Tuebingen , Germany"},{"name":"Department of Craniofacial Surgery, Tuebingen University Hospital , Tuebingen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Krimmel","sequence":"additional","affiliation":[{"name":"Interdisciplinary Center for Craniofacial Malformations, Tuebingen University Hospital , Tuebingen , Germany"},{"name":"Department of Craniofacial Surgery, Tuebingen University Hospital , Tuebingen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bernd","family":"Koos","sequence":"additional","affiliation":[{"name":"Interdisciplinary Center for Craniofacial Malformations, Tuebingen University Hospital , Tuebingen , Germany"},{"name":"Department of Orthodontics, Tuebingen University Hospital , Tuebingen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christian F","family":"Poets","sequence":"additional","affiliation":[{"name":"Department of Neonatology, Tuebingen University Hospital , Tuebingen , Germany"},{"name":"Interdisciplinary Center for Craniofacial Malformations, Tuebingen University Hospital , Tuebingen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mirja","family":"Quante","sequence":"additional","affiliation":[{"name":"Department of Neonatology, Tuebingen University Hospital , Tuebingen , Germany"},{"name":"Interdisciplinary Center for Craniofacial Malformations, Tuebingen University Hospital , Tuebingen , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2022,12,25]]},"reference":[{"issue":"9","key":"2023051017080300300_CIT0001","doi-asserted-by":"crossref","first-page":"894","DOI":"10.1001\/jamapediatrics.2016.0796","article-title":"Best practices for the diagnosis and evaluation of infants with Robin sequence: a clinical consensus report","volume":"170","author":"Breugem","year":"2016","journal-title":"JAMA Pediatr"},{"issue":"6","key":"2023051017080300300_CIT0002","doi-asserted-by":"crossref","first-page":"887","DOI":"10.1016\/j.jpeds.2011.07.033","article-title":"Treatment of upper airway obstruction and feeding problems in Robin-like phenotype","volume":"159","author":"Poets","year":"2011","journal-title":"J Pediatr."},{"issue":"5","key":"2023051017080300300_CIT0003","doi-asserted-by":"crossref","first-page":"936","DOI":"10.1542\/peds.2010-2615","article-title":"Robin sequence: from diagnosis to development of an effective management plan","volume":"127","author":"Evans","year":"2011","journal-title":"Pediatrics."},{"key":"2023051017080300300_CIT0004","doi-asserted-by":"crossref","first-page":"9","DOI":"10.1186\/1750-1172-9-9","article-title":"Birth prevalence and initial treatment of Robin sequence in Germany: a prospective epidemiologic study","volume":"9","author":"Vatlach","year":"2014","journal-title":"Orphanet J Rare Dis."},{"issue":"3","key":"2023051017080300300_CIT0005","doi-asserted-by":"crossref","first-page":"477","DOI":"10.5664\/jcsm.7680","article-title":"Longitudinal sleep outcomes in neonates with Pierre Robin sequence treated conservatively","volume":"15","author":"Ehsan","year":"2019","journal-title":"J Clin Sleep Med."},{"issue":"5","key":"2023051017080300300_CIT0006","doi-asserted-by":"crossref","first-page":"919","DOI":"10.5665\/sleep.3660","article-title":"Impact of sleep and breathing in infancy on outcomes at three years of age for children with cleft lip and\/or palate","volume":"37","author":"Smith","year":"2014","journal-title":"Sleep."},{"issue":"4","key":"2023051017080300300_CIT0007","doi-asserted-by":"crossref","first-page":"298","DOI":"10.1016\/j.jcms.2021.02.001","article-title":"Perioperative complications in cleft palate repair with Robin sequence following Tuebingen palatal plate treatment","volume":"49","author":"Naros","year":"2021","journal-title":"J Craniomaxillofac Surg."},{"issue":"7","key":"2023051017080300300_CIT0008","doi-asserted-by":"crossref","first-page":"875","DOI":"10.5665\/SLEEP.1118","article-title":"Prevalence and persistence of sleep disordered breathing symptoms in young children: a 6-year population-based cohort study","volume":"34","author":"Bonuck","year":"2011","journal-title":"Sleep."},{"issue":"6","key":"2023051017080300300_CIT0009","doi-asserted-by":"crossref","DOI":"10.1016\/j.siny.2021.101281","article-title":"Evidence and practical aspects of treatment with the Tubingen palatal plate","volume":"26","author":"Wiechers","year":"2021","journal-title":"Semin Fetal Neonatal Med."},{"issue":"6","key":"2023051017080300300_CIT0010","doi-asserted-by":"crossref","first-page":"101281","DOI":"10.1016\/j.siny.2021.101281","article-title":"Evidence and practical aspects of treatment with the Tubingen palatal plate","volume":"26","author":"Wiechers","year":"2021","journal-title":"Semin Fetal Neonatal Med."},{"key":"2023051017080300300_CIT0011","volume-title":"The WHO Child Growth Standards","author":"World Health Organization."},{"issue":"5","key":"2023051017080300300_CIT0012","doi-asserted-by":"crossref","first-page":"597","DOI":"10.5664\/jcsm.2172","article-title":"Rules for scoring respiratory events in sleep: update of the 2007 AASM Manual for the Scoring of Sleep and Associated Events. Deliberations of the Sleep Apnea Definitions Task Force of the American Academy of Sleep Medicine","volume":"8","author":"Berry","year":"2012","journal-title":"J Clin Sleep Med."},{"issue":"1","key":"2023051017080300300_CIT0013","doi-asserted-by":"crossref","first-page":"69","DOI":"10.1183\/13993003.00385-2015","article-title":"Obstructive sleep disordered breathing in 2- to 18-year-old children: diagnosis and management","volume":"47","author":"Kaditis","year":"2016","journal-title":"Eur Respir J."},{"key":"2023051017080300300_CIT0014","volume-title":"Wechsler Intelligence Scale for Children","author":"D W.","year":"2014","edition":"5"},{"key":"2023051017080300300_CIT0015","doi-asserted-by":"crossref","first-page":"260","DOI":"10.1007\/s11818-010-0495-4","article-title":"Validation and psychometric properties of the German version of the Children\u2019s Sleep Habits Questionnaire (CSHQ-DE)","volume":"14","author":"Schlarb","year":"2010","journal-title":"Somnologie"},{"issue":"4","key":"2023051017080300300_CIT0016","doi-asserted-by":"crossref","first-page":"240","DOI":"10.1007\/s007870170013","article-title":"Discriminant validity of the child behaviour checklist CBCL-4\/18 in German samples","volume":"10","author":"Schmeck","year":"2001","journal-title":"Eur Child Adolesc Psychiatry."},{"key":"2023051017080300300_CIT0017","doi-asserted-by":"crossref","first-page":"98","DOI":"10.1016\/j.ijporl.2016.04.030","article-title":"Quality of life in children with Robin Sequence","volume":"86","author":"Dulfer","year":"2016","journal-title":"Int J Pediatr Otorhinolaryngol."},{"issue":"4","key":"2023051017080300300_CIT0018","doi-asserted-by":"crossref","first-page":"313","DOI":"10.1001\/archpedi.162.4.313","article-title":"Effect of weight, sleep duration, and comorbid sleep disorders on behavioral outcomes in children with sleep-disordered breathing","volume":"162","author":"Owens","year":"2008","journal-title":"Arch Pediatr Adolesc Med."},{"issue":"3","key":"2023051017080300300_CIT0019","doi-asserted-by":"crossref","first-page":"307","DOI":"10.5664\/jcsm.3536","article-title":"Symptoms of sleep disordered breathing in children with craniofacial malformations","volume":"10","author":"Moraleda-Cibrian","year":"2014","journal-title":"J Clin Sleep Med."},{"issue":"3","key":"2023051017080300300_CIT0020","doi-asserted-by":"crossref","first-page":"269","DOI":"10.1001\/archoto.2011.8","article-title":"Increased prevalence of obstructive sleep apnea in patients with cleft palate","volume":"137","author":"Robison","year":"2011","journal-title":"Arch Otolaryngol Head Neck Surg."},{"issue":"2","key":"2023051017080300300_CIT0021","doi-asserted-by":"crossref","DOI":"10.1177\/1055665618774025","article-title":"Mandibular catch-up growth in Pierre Robin sequence: a systematic review","volume":"56","author":"Purnell","year":"2018","journal-title":"Cleft Palate Craniofac J."},{"issue":"4","key":"2023051017080300300_CIT0022","doi-asserted-by":"crossref","first-page":"425","DOI":"10.1597\/1545-1569_1991_028_0425_mtaaip_2.3.co_2","article-title":"Mandible, tongue, and airway in Pierre Robin sequence: a longitudinal cephalometric study","volume":"28","author":"Figueroa","year":"1991","journal-title":"Cleft Palate Craniofac J."},{"issue":"5","key":"2023051017080300300_CIT0023","first-page":"898","article-title":"Skeletal characteristics and treatment outcome of five patients with Robin sequence","volume":"76","author":"Matsuda","year":"2006","journal-title":"Angle Orthod."},{"issue":"1","key":"2023051017080300300_CIT0024","doi-asserted-by":"crossref","first-page":"17","DOI":"10.1186\/s13005-019-0200-1","article-title":"Mandibular growth in infants with Robin sequence treated with the Tubingen palatal plate","volume":"15","author":"Wiechers","year":"2019","journal-title":"Head Face Med"},{"issue":"6","key":"2023051017080300300_CIT0025","doi-asserted-by":"crossref","first-page":"2055","DOI":"10.1097\/SCS.0b013e3181be87db","article-title":"Three-dimensional assessment of facial development in children with Pierre Robin sequence","volume":"20","author":"Krimmel","year":"2009","journal-title":"J Craniofac Surg."},{"issue":"4","key":"2023051017080300300_CIT0026","doi-asserted-by":"crossref","first-page":"1459","DOI":"10.1097\/SCS.0000000000007339","article-title":"Effects of mandibular distraction osteogenesis on three-dimensional upper airway anatomy in newborns affected by isolated Pierre Robin sequence","volume":"32","author":"Mao","year":"2021","journal-title":"J Craniofac Surg."},{"issue":"3","key":"2023051017080300300_CIT0027","doi-asserted-by":"crossref","first-page":"377","DOI":"10.1016\/j.jcms.2016.12.007","article-title":"Three-dimensional airways volumetric analysis before and after fast and early mandibular osteodistraction","volume":"45","author":"Ramieri","year":"2017","journal-title":"J Craniomaxillofac Surg."},{"issue":"1","key":"2023051017080300300_CIT0028","doi-asserted-by":"crossref","first-page":"90","DOI":"10.1016\/j.joms.2012.03.014","article-title":"Effects of mandibular distraction osteogenesis on three-dimensional airway anatomy in children with congenital micrognathia","volume":"71","author":"Abramson","year":"2013","journal-title":"J Oral Maxillofac Surg."},{"issue":"3","key":"2023051017080300300_CIT0029","doi-asserted-by":"crossref","first-page":"576","DOI":"10.1542\/peds.2012-1671","article-title":"Diagnosis and management of childhood obstructive sleep apnea syndrome","volume":"130","author":"Marcus","year":"2012","journal-title":"Pediatrics."},{"issue":"5","key":"2023051017080300300_CIT0030","doi-asserted-by":"crossref","first-page":"934","DOI":"10.1097\/00006534-199404001-00006","article-title":"The Pierre Robin sequence: review of 125 cases and evolution of treatment modalities","volume":"93","author":"Caouette-Laberge","year":"1994","journal-title":"Plast Reconstr Surg."},{"issue":"5","key":"2023051017080300300_CIT0031","doi-asserted-by":"crossref","first-page":"653","DOI":"10.1111\/j.1651-2227.2008.00742.x","article-title":"Cognitive and psychosocial development of children with Pierre Robin sequence","volume":"97","author":"Drescher","year":"2008","journal-title":"Acta Paediatr."},{"key":"2023051017080300300_CIT0032","doi-asserted-by":"crossref","DOI":"10.1007\/s00784-022-04448-3","article-title":"Neurocognitive development in isolated Robin sequence treated with the Tuebingen palatal plate","author":"Naros","year":"2022","journal-title":"Clin Oral Investig."},{"issue":"3","key":"2023051017080300300_CIT0033","doi-asserted-by":"crossref","first-page":"366","DOI":"10.1080\/15374416.2015.1079782","article-title":"Sleeping toward behavioral regulation: relations between sleep and externalizing symptoms in toddlers and preschoolers","volume":"47","author":"Belanger","year":"2018","journal-title":"J Clin Child Adolesc Psychol."},{"issue":"8","key":"2023051017080300300_CIT0034","doi-asserted-by":"crossref","first-page":"897","DOI":"10.1007\/s00787-014-0636-6","article-title":"Sleep problems predict comorbid externalizing behaviors and depression in young adolescents with attention-deficit\/hyperactivity disorder","volume":"24","author":"Becker","year":"2015","journal-title":"Eur Child Adolesc Psychiatry."},{"issue":"10","key":"2023051017080300300_CIT0035","doi-asserted-by":"crossref","first-page":"3358","DOI":"10.1002\/ppul.25611","article-title":"Impact of sleep-disordered breathing on behavior and quality of life in children aged 2 to 7 years with non-syndromic cleft lip and\/or palate","volume":"56","author":"Moraleda-Cibrian","year":"2021","journal-title":"Pediatr Pulmonol."},{"issue":"1","key":"2023051017080300300_CIT0036","doi-asserted-by":"crossref","first-page":"36","DOI":"10.1111\/apa.15485","article-title":"Current ideas about the roles of rapid eye movement and non\u2013rapid eye movement sleep in brain development","volume":"110","author":"Knoop","year":"2021","journal-title":"Acta Paediatr."}],"container-title":["SLEEP"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/sleep\/advance-article-pdf\/doi\/10.1093\/sleep\/zsac317\/49628480\/zsac317.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/sleep\/article-pdf\/46\/5\/zsac317\/50267877\/zsac317.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/sleep\/article-pdf\/46\/5\/zsac317\/50267877\/zsac317.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,5,11]],"date-time":"2023-05-11T07:21:24Z","timestamp":1683789684000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/sleep\/article\/doi\/10.1093\/sleep\/zsac317\/6960708"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,12,25]]},"references-count":36,"journal-issue":{"issue":"5","published-online":{"date-parts":[[2022,12,25]]},"published-print":{"date-parts":[[2023,5,10]]}},"URL":"https:\/\/doi.org\/10.1093\/sleep\/zsac317","relation":{},"ISSN":["0161-8105","1550-9109"],"issn-type":[{"value":"0161-8105","type":"print"},{"value":"1550-9109","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2023,5,1]]},"published":{"date-parts":[[2022,12,25]]},"article-number":"zsac317"}}