Tracheobronchial Anomalies in Syndromic Craniosynostosis With 3-Dimensional CT Image and Bronchoscopy

Fujimoto, Takuya; Imai, Keisuke; Matsumoto, Hiroshi; Sakamoto, Hiroaki; Nakano, Tomoaki

doi:10.1097/scs.0b013e31822e5d15

Cited by 13 publications

(15 citation statements)

References 16 publications

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“…Almost half of these patients have morphologic airway abnormalities . Upper airway complications reported in these studies are choanal and upper airway stenosis, adenotonsillar hypertrophy (ATH), and nasal narrowing; notable lower airway complications are tracheomalacia, stenosis of the main bronchus, and tracheal cartilaginous sleeve (TCS) . It has also been reported that 40% to 50% of patients with SCS require tracheostomy and that most of these patients later present with severe upper airway stenosis .…”

Section: Introductionmentioning

confidence: 96%

“…Upper airway complications reported in these studies are choanal and upper airway stenosis, adenotonsillar hypertrophy (ATH), and nasal narrowing; notable lower airway complications are tracheomalacia, stenosis of the main bronchus, and tracheal cartilaginous sleeve (TCS) . It has also been reported that 40% to 50% of patients with SCS require tracheostomy and that most of these patients later present with severe upper airway stenosis . Midfacial hypoplasia can additionally result in stenosis of the upper airway tract, which in patients with SCS has been linked to obstructive sleep apnea (OSA), a disorder observed in up to 70% of patients with SCS .…”

Section: Introductionmentioning

confidence: 98%

“…There have been many studies demonstrating airway obstruction in patients with SCS. Almost half of these patients have morphologic airway abnormalities . Upper airway complications reported in these studies are choanal and upper airway stenosis, adenotonsillar hypertrophy (ATH), and nasal narrowing; notable lower airway complications are tracheomalacia, stenosis of the main bronchus, and tracheal cartilaginous sleeve (TCS) .…”

Section: Introductionmentioning

confidence: 99%

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Airway anomalies in patients with craniosynostosis

et al. 2018

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Objectives: 1) Characterize the spectrum of airway anomalies in patients with craniosynostosis, and 2) identify clinical characteristics of these patients that may be associated with the development of airway anomalies.Methods: This study is a retrospective case series assessing the type and frequency of airway anomalies in all patients with craniosynostosis seen at a tertiary-care children's hospital between 2000 and 2016. Cohort analyses were then performed to identify differences in airway anomalies dependent on syndromic associations, multisutural fusion, and location of suture fusion. Clinical characteristics examined included demographics and additional neurologic and craniofacial abnormalities.Results: Four hundred and ninety-six patients with craniosynostosis (83.5% white, 64.5% male; 33.9% sagittal, 28.8% metopic, 11.5% coronal, 1.2% lambdoid, and 24.6% multisutural) were included. Notable airway anomalies included the following: 13.3% adenotonsillar hypertrophy, 8.9% laryngomalacia, 7.3% tracheomalacia, 7.1% subglottic stenosis, 4.0% bronchomalacia, 3.8% laryngeal cleft, and 1.2% vocal fold paresis. Multisutural craniosynostosis patients (n = 122) were more likely to have obstructive sleep apnea (P = 0.005), adenotonsillar hypertrophy (P = 0.014), tracheomalacia (P = 0.011), subglottic stenosis (P < 0.001), and epiglottic/base of tongue collapse (P = 0.003) and require tracheostomy (P = 0.001) and mechanical ventilation (P = 0.017) compared with single suture craniosynostosis. Syndromic craniosynostosis patients (n = 33) were more likely to have obstructive sleep apnea (P < 0.001), laryngomalacia (P = 0.047), and subglottic stenosis (P = 0.009) compared with nonsyndromic patients.Conclusion: Airway anomalies are prevalent in patients with craniosynostosis; patients with multisutural or syndromic types have an increased risk of developing certain abnormalities. There should be a lower threshold for referral for airway evaluation in these populations.

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Section: Introductionmentioning

confidence: 96%

Section: Introductionmentioning

confidence: 98%

Section: Introductionmentioning

confidence: 99%

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Airway anomalies in patients with craniosynostosis

et al. 2018

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“…3,4 Careful planning of the delivery method and perinatal care is crucial because of the potential for complex anomalies of the upper airway due to midfacial hypoplasia. 5 Moreover, multiple reconstructive surgeries requiring the coordination of dental and orthodontic specialists may be necessary after the infancy period. 6,7 Various types of neurological pathophysiology, including elevated intracranial pressure, hydrocephalus and Chiari malformations in growing children can cause brain damage associated with neurologic and cognitive dysfunction.…”

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confidence: 99%

Prenatal sonographic findings and prognosis of craniosynostosis diagnosed during the fetal and neonatal periods

Aya

Miyashita

Nagai³

et al. 2018

Congenital Anomalies

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The aim of the study was to explore the sonographic findings of fetuses with craniosynostosis and investigate their prognosis. We conducted a 5-year, multicenter retrospective study and collected data on patients with craniosynostosis diagnosed in the perinatal period. Of 41 cases, 30 cases (73%) were syndromic craniosynostosis, eight cases (20%) were non-syndromic craniosynostosis and the other three cases (7%) were secondary craniosynostosis of chromosomal deletion syndromes. The prenatal ultrasound detection rate was 61%. Half of the cases of syndromic craniosynostosis detected during the perinatal period were Pfeiffer syndrome; there were also six cases of Apert syndrome, three cases of Crouzon syndrome and other rare form of syndromic craniosynostosis (Beare-Stevenson syndrome, Saethre-Chotzen syndrome, cranioectodermal dysplasia, and thanatophoric dysplasia). Abnormal shape of the skull was the most common finding leading to prenatal diagnosis of craniosynostosis. Abnormal head biometry, which was the second most frequent finding, was closely correlated with deformation of the cranial shape. Three cases presented with ventriculomegaly and exophthalmos but normal cranial shape and size. The overall survival rate of infants with syndromic craniosynostosis was 79%, while all of the infants with non-syndromic craniosynostosis survived. In conclusion, prenatal diagnosis of craniosynostosis is difficult, especially when dysmorphic change of the fetal cranium is not evident. Abnormal head biometry and ventriculomegaly could potentially be additional markers of fetal craniosynostosis and consequently increase the prenatal detection rate.

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“…Virtual 3D reconstructions have been used before in otorhinolaryngology, using stereophotogrammetrical analyses or CT scans [5][6][7][8]. Applications vary from laryngectomy stoma assessment to 3D planning of operations.…”

Section: Discussionmentioning

confidence: 99%

Virtual 3D planning of tracheostomy placement and clinical applicability of 3D cannula design: a three-step study

Kleijn

Kraeima

Wachters

et al. 2017

Eur Arch Otorhinolaryngol

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Aim We aimed to investigate the potential of 3D virtual planning of tracheostomy tube placement and 3D cannula design to prevent tracheostomy complications due to inadequate cannula position. Materials and methods 3D models of commercially available cannula were positioned in 3D models of the airway. In study (1), a cohort that underwent tracheostomy between 2013 and 2015 was selected (n = 26). The cannula was virtually placed in the airway in the pre-operative CT scan and its position was compared to the cannula position on post-operative CT scans. In study (2), a cohort with neuromuscular disease (n = 14) was analyzed. Virtual cannula placing was performed in CT scans and tested if problems could be anticipated. Finally (3), for a patient with Duchenne muscular dystrophy and complications of conventional tracheostomy cannula, a patient-specific cannula was 3D designed, fabricated, and placed. Results (1) The 3D planned and post-operative tracheostomy position differed significantly. (2) Three groups of patients were identified: (A) normal anatomy; (B) abnormal anatomy, commercially available cannula fits; and (C) abnormal anatomy, custom-made cannula, may be necessary. (3) The position of the custom-designed cannula was optimal and the trachea healed. Conclusions Virtual planning of the tracheostomy did not correlate with actual cannula position. Identifying patients with abnormal airway anatomy in whom commercially available cannula cannot be optimally positioned is advantageous. Patientspecific cannula design based on 3D virtualization of the airway was beneficial in a patient with abnormal airway anatomy.

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Tracheobronchial Anomalies in Syndromic Craniosynostosis With 3-Dimensional CT Image and Bronchoscopy

Cited by 13 publications

References 16 publications

Airway anomalies in patients with craniosynostosis

Airway anomalies in patients with craniosynostosis

Prenatal sonographic findings and prognosis of craniosynostosis diagnosed during the fetal and neonatal periods

Virtual 3D planning of tracheostomy placement and clinical applicability of 3D cannula design: a three-step study

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