Airway anomalies in patients with craniosynostosis

Mathews, Fasil; Shaffer, Amber D.; Georg, Matthew W.; Ford, Matthew; Goldstein, Jesse A.; Jabbour, Noel; Simons, Jeffrey P.

doi:10.1002/lary.27589

Cited by 12 publications

(15 citation statements)

References 47 publications

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“…This present study determined that patients with craniosynostosis, especially syndromic patients, had a significantly higher incidence of not only choanal atresia but also other various airway anomalies. These results are consistent with a single institution retrospective review of syndromic craniosynostosis patients by Matthews et al 3 which found that patients with syndromic types were at a much higher risk of developing these anomalies. For example, they similarly discovered higher rates of subglottic stenosis and tracheomalacia in those with syndromic craniosynostosis 3 …”

Section: Discussionsupporting

confidence: 92%

“…These results are consistent with a single institution retrospective review of syndromic craniosynostosis patients by Matthews et al 3 which found that patients with syndromic types were at a much higher risk of developing these anomalies. For example, they similarly discovered higher rates of subglottic stenosis and tracheomalacia in those with syndromic craniosynostosis 3 …”

Section: Discussionsupporting

confidence: 92%

“…Although there have been other research studies investigating the relationship between craniosynostosis and airway abnormalities, the incidence of these abnormalities has not been investigated based on a large-scale database study 3,5 . This present study determined that patients with craniosynostosis, especially syndromic patients, had a significantly higher incidence of not only choanal atresia but also other various airway anomalies.…”

Section: Discussionmentioning

confidence: 64%

“…FGFR has a significant role in not only the formation of cranial sutures and bones of the skull but also in the differentiation and proliferation of many embryologic tissues 2 . Therefore, in addition to craniofacial abnormalities, these syndromes can feature airway complications such as laryngomalacia, tracheal stenosis, tracheomalacia, subglottic stenosis, and choanal atresia 3 …”

mentioning

confidence: 99%

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Incidence of Airway Abnormalities in Children With Craniosynostosis

Eitan,

Bhuskute,

Scheffler

2023

Journal of Craniofacial Surgery

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Introduction: Children with syndromic craniosynostosis are known to have a high propensity for associated airway abnormalities. However, this has not been investigated using a large-scale national database. Methods: For this retrospective cohort study, the 2016 Healthcare Cost and Utilization Project Kid’s Inpatient Database was queried for craniosynostosis patients. Data on demographics, airway diagnoses, and comorbidities were analyzed. Results: Four thousand nine hundred fourteen children with craniosynostosis with a mean age of 1.7±3.6 years were identified. Of these, 51% were female and 136 children had an associated syndrome. Choanal atresia was present in 31% of patients with an associated syndrome versus 2.5% without. Syndromic patients are 4.59 times more likely (95% CI 2.65–7.94) to have airway anomalies than nonsyndromic patients. After age and sex adjustment, craniosynostosis patients have higher likelihoods of presenting with other anomalies, with syndromic having higher incidences: 5.23 times (95% CI 2.63–10.39) more likely to have laryngomalacia, 18.30 times (95% CI 3.27–102.36) more likely to have tracheal stenosis, and 4.58 times (95% CI 1.36– 15.43) more likely to have tracheomalacia. Incidence of tracheostomy was 5.84 times (95% CI 3.77–9.04) higher in syndromic patients with craniosynostosis. Tracheostomy rates were 28.4% and 4.6% in craniosynostosis patients with and without associated syndrome, respectively. Conclusion: Syndromic craniosynostosis patients had significantly higher incidences of choanal atresia and other airway anomalies. Given a high incidence of airway anomalies, syndromic craniosynostosis patients likely warrant routine airway evaluation. Providers should also be vigilant about airway evaluation in patients with nonsyndromic craniosynostosis when aerodigestive symptoms arise.

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Section: Discussionsupporting

confidence: 92%

Section: Discussionsupporting

confidence: 92%

Section: Discussionmentioning

confidence: 64%

mentioning

confidence: 99%

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Incidence of Airway Abnormalities in Children With Craniosynostosis

Eitan,

Bhuskute,

Scheffler

2023

Journal of Craniofacial Surgery

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“…(10) Some of the causes are associated with some syndromes. (11) There are very few case series in Indian population reporting such cases. In our tertiary care centre, we come across many cases with stridor.…”

Section: B a C K G R O U N Dmentioning

confidence: 99%

Management of Stridor in Paediatric Population- Our Experience

Thakur¹,

Singh²,

Roy³

et al. 2019

jemds

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BACKGROUNDStridor is a common presentation in paediatric ENT OPDs. (1) There can be numerous causes of stridor in this age group-congenital and acquired. (2) There are various methods available for diagnosis of such cases-radiological, rigid laryngotracheobronchoscopy and fibreoptic laryngoscopy. (3) The approach to such a case and its subsequent management varies from case to case. (4) Treatment strategy for each possible cause varies. There are very few case series in Indian population reporting such cases. In our tertiary care centre, we come across many cases with stridor. Their subsequent evaluation leads us to diagnosis. At times, upper airway evaluation is normal. The management then depends on specific aetiology. We are describing our case series comprising 15 patients with various diagnosis and subsequent management. METHODSThis is a case series comprising 15 patients. This is a prospective study carried out at our tertiary care centre. The variety of cases included laryngomalacia, retropharyngeal abscess and subglottic stenosis which were managed as per existing guidelines. RESULTSThe outcomes of our series were quite promising with 93.33 % children successfully decannulated. CONCLUSIONSThe management of a child with stridor is a challenge for the treating team. We should keep in mind that every paediatric case of stridor does not imply an underlying pathology. A systematic approach and keeping all the possible differential diagnoses in mind are the most important steps in achieving optimum results.

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Measurement of Upper Airway Volume in Children with Craniofacial Abnormalities

Gordon,

Ben‐Dov,

Asfour

et al. 2023

The Laryngoscope

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Objective(s)Previous literature has established a high prevalence of upper airway obstruction in children with craniofacial abnormalities. This study aims to perform quantitative airway volume measurements in patients with craniofacial abnormalities and compare them to age and sex‐matched controls.MethodsWe performed a retrospective review of the records of all children with craniofacial abnormalities who underwent head‐and‐neck computed tomography (CT) imaging at a single tertiary‐care center between 1/1/13 and 12/31/20 using the ICD‐10 codes Q75.1, Q75.4, and Q87.0. These patients were then matched by age and sex to patients with isolated craniosynostosis (Q75.0). CT scans were imported into Dolphin Imaging software, and airway volumes were measured for the nasal cavity, nasopharynx, oropharynx, and hypopharynx. The primary outcome was the total airway volume, defined as the sum of these measurements.ResultsThirty subjects with craniofacial syndromes were matched to 30 patients with isolated craniosynostosis (controls). In both groups, 18 subjects (60%) were male (p = 0.99). The average ages for syndromic patients and controls were 12.1 and 12.9 months, respectively (p = 0.84). On average, the total airway volumes of syndromic patients were 25% lower than those of controls (p = 0.02). Syndromic patients had 39% smaller nasal cavity volumes (p < 0.001) and 32% smaller nasopharyngeal volumes (p < 0.01). Significant volume differences were not observed for the oropharynx or hypopharynx.ConclusionWe present a unique technique to measure airway volumes in patients with craniofacial abnormalities. These findings will help practitioners to further understand the anatomy and pathophysiology of disturbed breathing in children with craniofacial syndromes.Level of EvidenceIII Laryngoscope, 2023

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Airway anomalies in patients with craniosynostosis

Cited by 12 publications

References 47 publications

Incidence of Airway Abnormalities in Children With Craniosynostosis

Incidence of Airway Abnormalities in Children With Craniosynostosis

Management of Stridor in Paediatric Population- Our Experience

Measurement of Upper Airway Volume in Children with Craniofacial Abnormalities

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