Airway management in children with major craniofacial anomalies

Sculerati, Nancy; Gottlieb, Marc D.; Zimbler, Marc S.; Chibbaro, Patricia D.; McCarthy, Joseph G.

doi:10.1097/00005537-199812000-00008

Cited by 159 publications

(88 citation statements)

References 11 publications

(17 reference statements)

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“…Early tracheotomy is advocated for these infants. Approximately 20% of children with syndromes involving bony deformities of the face and skull unambiguously required tracheotomy, as reported by Sculerati et al [29]. …”

Section: Discussionmentioning

confidence: 97%

Antley-Bixler Syndrome, Description of Two New Cases and Review of the Literature

2001

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Objective: Antley-Bixler syndrome (ABS) is a rare disorder characterized by multiple malformations of cartilage and bone including multisynostotic osteodysgenesis, midface hypoplasia, choanal atresia or stenosis, femoral bowing, neonatal fractures and multiple joint contractures and, occasionally, urogenital, gastrointestinal or cardiac defects. Since the first report in 1975, at least 34 cases (including this report) have been described. We present 2 cases of congenital hydrocephalus, suggesting the cause of craniosynostosis and midface hypoplasia is associated with ABS. Clinical Presentation: The first case was a 1-day-old female with Arnold-Chiari malformation, multiple cranial synostosis, obstructive hydrocephalus and radioulnahumeral synostosis. Ventriculoperitoneal (V-P) shunting was performed when she was 7 days old. She died 42 days later due to cardiopulmonary failure. The second case was a 2-month-old female with bilateral coronal synostosis, obstructive hydrocephalus and brachycephaly. V-P shunting was done soon after her admission, and bilateral lateral canthal advancement with the floating forehead procedure was performed 1 month later. She is still alive with good development. Discussion: The main anomalies of the ABS can be divided into 4 categories: craniofacial, skeletal, extremity and urogenital anomalies. They may be the result of mutation in the fibroblast growth factor receptor 2 (Ser351Cys) gene, which was confirmed in our case 2. Craniosynostosis combined with hydrocephalus created congenital increased intracranial pressure (IICP). Early V-P shunt implantation and surgical release of the closed suture and lateral canthal advancement should be done as soon as possible, ideally when the patient is younger than 3 months. Conclusion: Early correction of craniosynostosis was feasible and safe for one of our patients. We offer our experience in the treatment of hydrocephalus and correction of craniosynostosis to relieve IICP.

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Section: Discussionmentioning

confidence: 97%

Antley-Bixler Syndrome, Description of Two New Cases and Review of the Literature

2001

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“…Most of the patients with craniofacial anomalies require airway management ranging from prone positioning to tracheotomy and DO 11 . DO is accepted as a reliable surgical technique and an alternative to tracheotomy for long-term management of the airway 11,12,13 . It has been observed that DO reduces the need for tracheotomy especially in pediatric patients and allows for early decannulation in patients who previously underwent tracheotomy 14,15 .…”

Section: Discussionmentioning

confidence: 99%

Evaluation of airway volume changes in patients treated with distraction osteogenesis: a pilot study

Valiathan¹,

Metzgera²,

El³

et al. 2010

J Dentofacial Anom Orthod

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The use of distraction osteogenesis to improve the airway volume of individuals affected by craniofacial anomalies is common practice today. The methods of assessing the outcomes of such surgical procedures have changed over the last several years.Objective : The objective of the present pilot study was to determine if the CBCT imaging modality may serve as a simple and reliable method for craniofacial practitioners to evaluate airway volume changes following distraction osteogenesis.Materials and Methods : Twelve patients who had previously undergone distraction osteogenesis for the primary purpose of improving their airways were enrolled in the current study. Pre (T0) and post-surgical (T1) CBCT's volumes were analyzed to measure the nasopharyngeal (NP) and oropharyngeal (OP) volume changes. InVivoDental 4.0 (Anatomage Inc., San Jose, CA) (IVD) program was used to visualize and render the oropharyngeal (OP) and nasal passage (NP) volumes, separately. Means and standard deviations were calculated.Results : Of the 7 males and 5 females in the study, 4 patients underwent mandibular distraction (MandDO) and 8 underwent maxillary distraction (MaxDO). Four in the MaxDO group were treated with internal distractors and 4 were treated with external distractors. Individuals who underwent internalArticle available at

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“…(d) Tracheostomy may be necessary in some infants with bilateral CA. 10 However, this is an invasive procedure.…”

Section: Discussionmentioning

confidence: 99%

Preoperative airway management in a preterm with bilateral choanal atresia

et al. 2007

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Airway management in children with major craniofacial anomalies

Cited by 159 publications

References 11 publications

Antley-Bixler Syndrome, Description of Two New Cases and Review of the Literature

Antley-Bixler Syndrome, Description of Two New Cases and Review of the Literature

Evaluation of airway volume changes in patients treated with distraction osteogenesis: a pilot study

Preoperative airway management in a preterm with bilateral choanal atresia

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