{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,28]],"date-time":"2026-05-28T00:42:32Z","timestamp":1779928952274,"version":"3.53.1"},"reference-count":23,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"3","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["PED"],"published-print":{"date-parts":[[2013,3]]},"abstract":"<jats:sec>\n<jats:title>Object<\/jats:title>\n<jats:p>The association of Chiari malformation Type I (CM-I) with syndromic craniosynostosis (SC) in children is well established. Central sleep apnea (CSA) may subsequently occur. However, sleep studies performed in these patients have been focused mainly on assessing the severity of obstructive sleep apnea. Therefore, the incidence and management of CSA in these patients remains poorly defined. Authors of this study aimed to assess the efficacy of foramen magnum decompression (FMD) in resolving CSA, initially detected incidentally, in a small cohort of patients with CM-I and SC.<\/jats:p><\/jats:sec>\n<jats:sec>\n<jats:title>Methods<\/jats:title>\n<jats:p>The clinical data for 5 children who underwent FMD for CSA at Alder Hey Children's Hospital between December 2007 and December 2009 were retrospectively analyzed. Outcomes were evaluated with respect to FMDs by utilizing pre- and postdecompression sleep studies. Of the 5 patients, 2 had Crouzon syndrome and 3 had Pfeiffer syndrome.<\/jats:p><\/jats:sec>\n<jats:sec>\n<jats:title>Results<\/jats:title>\n<jats:p>Patient age at the time of surgery ranged from 1.1 to 12.6 years (median 4.1 years). The median postoperative follow-up was 3.6 years. Sleep studies revealed that 2 children experienced a &gt; 80% reduction in CSAs at 1.5 and 21 months after decompression. The remaining 3 children experienced a &gt; 60% reduction in CSAs when reevaluated between 2 and 10 months after decompression. The associated central apnea index improved for all patients.<\/jats:p><\/jats:sec>\n<jats:sec>\n<jats:title>Conclusions<\/jats:title>\n<jats:p>Findings suggested that FMD is an effective treatment modality for improving CSA in patients with SC and associated CM-I. The use of multimodal polysomnography technology may improve the evaluation and management of these patients.<\/jats:p><\/jats:sec>","DOI":"10.3171\/2012.11.peds12297","type":"journal-article","created":{"date-parts":[[2012,12,14]],"date-time":"2012-12-14T14:58:09Z","timestamp":1355497089000},"page":"296-301","source":"Crossref","is-referenced-by-count":32,"title":["Central sleep apnea and associated Chiari malformation in children with syndromic craniosynostosis: treatment and outcome data from a supraregional national craniofacial center"],"prefix":"10.3171","volume":"11","author":[{"given":"Nii K.","family":"Addo","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sheila","family":"Javadpour","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jothy","family":"Kandasamy","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul","family":"Sillifant","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul","family":"May","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ajay","family":"Sinha","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1754","reference":[{"key":"b1-peds12297","series-title":"J Craniomaxillofac Surg","doi-asserted-by":"publisher","first-page":"153","DOI":"10.1016\/j.jcms.2010.04.011","article-title":"Sleep-related disordered breathing in children with syndromic craniosynostosis","volume":"39","author":"Al-Saleh","year":"2011"},{"key":"b2-peds12297","series-title":"Int J Oral Maxillofac Surg","doi-asserted-by":"publisher","first-page":"115","DOI":"10.1016\/j.ijom.2009.11.021","article-title":"Obstructive sleep apnea in children with syndromic craniosynostosis: long-term respiratory outcome of midface advancement","volume":"39","author":"Bannink","year":"2010"},{"key":"b3-peds12297","series-title":"Neurosurgery","doi-asserted-by":"publisher","first-page":"66","DOI":"10.1097\/00006123-199801000-00013","article-title":"Occipital remodeling and suboccipital decompression in severe craniosynostosis associated with tonsillar herniation","volume":"42","author":"Cinalli","year":"1998"},{"key":"b4-peds12297","series-title":"Childs Nerv Syst","doi-asserted-by":"publisher","first-page":"889","DOI":"10.1007\/s00381-004-1115-z","article-title":"Chiari malformation in craniosynostosis","volume":"21","author":"Cinalli","year":"2005"},{"key":"b5-peds12297","series-title":"Chest","doi-asserted-by":"publisher","first-page":"595","DOI":"10.1378\/chest.06.2287","article-title":"Central sleep apnea: pathophysiology and treatment","volume":"131","author":"Eckert","year":"2007"},{"key":"b6-peds12297","series-title":"J Craniofac Surg","doi-asserted-by":"publisher","first-page":"8","DOI":"10.1097\/00001665-199207000-00004","article-title":"Aggressive surgical management of sleep apnea syndrome in the syndromal craniosynostoses","volume":"3","author":"Edwards","year":"1992"},{"key":"b7-peds12297","series-title":"Pediatr Neurosurg","first-page":"228","article-title":"Development of symptomatic Chiari malformation in a child with craniofacial dysmorphism","volume":"16","author":"Frim","year":"1991"},{"key":"b8-peds12297","series-title":"Dev Med Child Neurol","first-page":"366","article-title":"Central ventilatory dysfunction in myelodysplasia: an independent determinant of survival","volume":"31","author":"Hays","year":"1989"},{"key":"b9-peds12297","series-title":"J Pediatr Health Care","doi-asserted-by":"publisher","first-page":"190","DOI":"10.1016\/S0891-5245(02)88336-5","article-title":"Arnold-Chiari malformation type I and sleep-disordered breathing: an uncommon manifestation of an important pediatric problem","volume":"17","author":"Hershberger","year":"2003"},{"issue":"Suppl 1","key":"b10-peds12297","series-title":"Int J Pediatr Otorhinolaryngol","doi-asserted-by":"publisher","first-page":"S111","DOI":"10.1016\/j.ijporl.2003.08.007","article-title":"OSAS in craniofacial syndromes: an unsolved problem","volume":"67","author":"Hoeve","year":"2003"},{"key":"b11-peds12297","series-title":"The AASM Manual for the Scoring of Sleep and Associated Events: Rules, Terminology and Technical Specifications","author":"Iber","year":"2007"},{"key":"b12-peds12297","series-title":"Dev Med Child Neurol","doi-asserted-by":"publisher","first-page":"40","DOI":"10.1017\/S0012162299000079","article-title":"Sleep-disordered breathing in patients with myelomeningocele: the missed diagnosis","volume":"41","author":"Kirk","year":"1999"},{"key":"b13-peds12297","series-title":"Childs Nerv Syst","doi-asserted-by":"publisher","first-page":"771","DOI":"10.1007\/s00381-009-1044-y","article-title":"The incidence of Chiari malformation in nonsyndromic, single suture craniosynostosis","volume":"26","author":"Leikola","year":"2010"},{"key":"b14-peds12297","series-title":"Plast Reconstr Surg","doi-asserted-by":"publisher","first-page":"1603","DOI":"10.1097\/00006534-199911000-00001","article-title":"Molecular diagnosis of bilateral coronal synostosis","volume":"104","author":"Mulliken","year":"1999"},{"key":"b15-peds12297","series-title":"J Oral Maxillofac Surg","doi-asserted-by":"publisher","first-page":"2318","DOI":"10.1016\/j.joms.2008.06.063","article-title":"Effect of midfacial distraction on the obstructed airway in patients with syndromic bilateral coronal synostosis","volume":"66","author":"Nelson","year":"2008"},{"key":"b16-peds12297","series-title":"Eur Respir J","doi-asserted-by":"publisher","first-page":"1482","DOI":"10.1183\/09031936.98.12061482","article-title":"Central sleep apnoea in Arnold-Chiari malformation: evidence of pathophysiological heterogeneity","volume":"12","author":"Rabec","year":"1998"},{"issue":"6 Suppl","key":"b17-peds12297","series-title":"J Neurosurg","first-page":"490","article-title":"Anomalous venous drainage preventing safe posterior fossa decompression in patients with chiari malformation type I and multisutural craniosynostosis. Report of two cases and review of the literature","volume":"106","author":"Sandberg","year":"2007"},{"key":"b18-peds12297","series-title":"Pediatr Pulmonol","doi-asserted-by":"publisher","first-page":"1141","DOI":"10.1002\/ppul.21294","article-title":"Isolated central sleep apnea in type I Chiari malformation: improvement after surgery","volume":"45","author":"Spence","year":"2010"},{"key":"b19-peds12297","series-title":"Pediatr Neurosurg","first-page":"288","article-title":"Aetiology of herniation of the hindbrain in craniosynostosis. An investigation incorporating intracranial pressure monitoring and magnetic resonance imaging","volume":"26","author":"Thompson","year":"1997"},{"key":"b20-peds12297","series-title":"Eur J Paediatr Neurol","doi-asserted-by":"publisher","first-page":"463","DOI":"10.1016\/j.ejpn.2008.07.012","article-title":"Chiari type I malformation causing central apnoeas in a 4-month-old boy","volume":"13","author":"Van den Broek","year":"2009"},{"key":"b21-peds12297","series-title":"J Pediatr","doi-asserted-by":"publisher","first-page":"672","DOI":"10.1016\/S0022-3476(98)70359-2","article-title":"Sleep-disordered breathing in children with myelomeningocele","volume":"132","author":"Waters","year":"1998"},{"key":"b22-peds12297","series-title":"Sleep","doi-asserted-by":"publisher","first-page":"389","DOI":"10.1093\/sleep\/34.3.389","article-title":"Executive summary of respiratory indications for polysomnography in children: an evidence-based review","volume":"34","author":"Wise","year":"2011"},{"key":"b23-peds12297","series-title":"Sleep","first-page":"637","article-title":"Chiari malformation and sleep-disordered breathing: a review of diagnostic and management issues","volume":"23","author":"Zolty","year":"2000"}],"container-title":["Journal of Neurosurgery: Pediatrics"],"original-title":[],"link":[{"URL":"https:\/\/thejns.org\/view\/journals\/j-neurosurg-pediatr\/11\/3\/article-p296.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/thejns.org\/downloadpdf\/journals\/j-neurosurg-pediatr\/11\/3\/article-p296.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/thejns.org\/downloadpdf\/journals\/j-neurosurg-pediatr\/11\/3\/article-p296.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,4,15]],"date-time":"2023-04-15T14:26:07Z","timestamp":1681568767000},"score":1,"resource":{"primary":{"URL":"https:\/\/thejns.org\/view\/journals\/j-neurosurg-pediatr\/11\/3\/article-p296.xml"}},"subtitle":["Clinical article"],"short-title":[],"issued":{"date-parts":[[2013,3]]},"references-count":23,"journal-issue":{"issue":"3"},"URL":"https:\/\/doi.org\/10.3171\/2012.11.peds12297","relation":{},"ISSN":["1933-0707","1933-0715"],"issn-type":[{"value":"1933-0707","type":"print"},{"value":"1933-0715","type":"electronic"}],"subject":[],"published":{"date-parts":[[2013,3]]}}}