Bedside Polysomnography as an Adjunct in the Management of Infants with Robin Sequence

Gilhooly, Joseph; Smith, James D.; Howell, Laurene L.; Deschaine, Barbara L.; Richey, Susan L.

doi:10.1097/00006534-199307000-00003

Cited by 48 publications

(31 citation statements)

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“…Bull et al have noted that significant obstruction can appear over the first one to four weeks of life 5. Furthermore, Gilhooly et al have reported two patients presenting with apparent life threatening events at about 2 months of age as their first evidence of upper airway obstruction 8. Again the vast majority of patients in this series (13 of 15), had clinically apparent airway obstruction on day 1.…”

Section: Discussionmentioning

confidence: 55%

“…There is polysomnographic evidence that this can occur. Gilhooly et al reported that one infant had a normal initial polysomnogram but subsequently an abnormal study at 13 days of age with significant upper airway obstruction 8. In support of this, Smith also reported that respiratory obstruction in Pierre Robin sequence may be present at birth or may become progressively worse at 6 to 8 weeks of age 9.…”

Section: Discussionmentioning

confidence: 91%

“…There was also considerable disagreement about whether single or serial studies were required. Serial polysomnography may be necessary in view of the findings of Gilhooly et al 8. The notable phenotypic and pathogenetic variability of Pierre Robin sequence, which has recently been highlighted by Cohen,15 provides further support for the use of objective diagnostic techniques to detect upper airway obstruction.…”

Section: Discussionmentioning

confidence: 93%

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Late presentation of upper airway obstruction in Pierre Robin sequence

Wilson

Moore²,

Moore³

et al. 2000

Archives of Disease in Childhood

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A retrospective review was carried out of 11 consecutive patients with the Pierre Robin sequence referred to a tertiary paediatric referral centre over a five year period from 1993 to 1998. Ten patients were diagnosed with significant upper airway obstruction; seven of these presented late at between 24 and 51 days of age. Failure to thrive occured in six of these seven infants at the time of presentation, and was a strong indicator of the severity of upper airway obstruction. Growth normalised on treatment of the upper airway obstruction with nasopharyngeal tube placement. All children had been reviewed by either an experienced general paediatrician or a neonatologist in the first week of life, suggesting that clinical signs alone are insuYcent to alert the physician to the degree of upper airway obstruction or that obstruction developed gradually after discharge home. The use of polysomnography greatly improved the diagnostic accuracy in assesssing the severity of upper airway obstruction and monitoring the response to treatment. This report highlights the prevalence of late presentation of upper airway obstruction in the Pierre Robin sequence and emphasises the need for close prospective respiratory monitoring in this condition. Objective measures such as polysomnography should be used, as clinical signs alone may be an inadequate guide to the degree of upper airway obstruction. (Arch Dis Child 2000;83:435-438)

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Section: Discussionmentioning

confidence: 55%

Section: Discussionmentioning

confidence: 91%

Section: Discussionmentioning

confidence: 93%

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Late presentation of upper airway obstruction in Pierre Robin sequence

Wilson

Moore²,

Moore³

et al. 2000

Archives of Disease in Childhood

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“…When oxygenation is a problem, monitoring in the neonatal intensive care unit is necessary. Polysomnography and monitoring for carbon dioxide retention are necessary to evaluate for proper oxygenation and ventilation [109][110][111] (Fig. 10).…”

Section: Airway: Pierre Robin Sequencementioning

confidence: 99%

Craniofacial Syndromes

Buchanan

Xue

Hollier

2014

Plastic and Reconstructive Surgery

170

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Craniofacial syndromes fall into two major categories-those associated with craniosynostosis, and those associated with clefts. Each has a different set of potential complications requiring a unique approach for surgical management. Craniosynostosis is a congenital disorder in which one or more of the cranial sutures fuses prematurely. The most common syndromes associated with this condition include Crouzon, Apert, Pfeiffer, Muenke, and Saethre-Chotzen syndromes. Surgical management of these children requires a multidisciplinary approach and close involvement of the family. Operations must take into consideration the growing potential of the bony structures. Common syndromes associated with clefts include Pierre Robin, Treacher Collins, Nager, Binder, and Stickler syndromes. Many of these children have severe airway issues requiring immediate address before operative reconstruction. As with syndromes associated with craniosynostosis, the key to management is a multidisciplinary approach focused on the right timing. The purpose of this article is to review the clinical presentation, care, and treatment of these patients.

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“…However, most of these studies are based on clinical assessment. Few of these studies described the prevalence using PSG[19202122232425] with only two cross-sectional studies that described the OSA in PRS infants using the American Academy of Sleep Medicine (AASM) scoring criteria. [2425]…”

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confidence: 99%

Polysomnographic findings in infants with Pierre Robin sequence

2017

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INTRODUCTION:Pierre Robin sequence (PRS) is characterized by the triad of micrognathia, glossoptosis, and upper airway obstruction. It is commonly associated with the secondary cleft palate. Infants with PRS commonly have sleep-disordered breathing (SDB); including obstructive sleep apnea (OSA) as well as central sleep breathing abnormalities that are present from infancy.AIM OF THE STUDY:Evaluate the prevalence and severity of SDB in infants with PRS using polysomnography (PSG).SETTINGS AND DESIGN:We retrospectively reviewed the sleep laboratory database at The Hospital for Sick Children, Toronto, during the period of May 2007 to March 2016.STATISTICAL ANALYSIS:Comparisons of PSG data were made between the OSA and non-OSA group using the Student's t-test for age and body mass index, Wilcoxon signed ranks test for the continuous PSG data and Chi-squared test for the categorical variables.METHODS:Patients with PRS were identified and their initial PSG was selected for this study. The main indication for referral was ongoing concerns regarding OSA symptoms.RESULTS:A total of 46 patients (28 females) were included with a mean age (±standard deviation) of 0.8 (±0.3) year. Twenty-two out of 46 (47%) had evidence of OSA of which 10 had mild, 3 had moderate, and 9 had severe OSA. The PRS infants with OSA were younger than the non-OSA group. Significant correlations were found between desaturation and arousal indices with obstructive apnea–hypopnea index.CONCLUSION:This retrospective chart review confirms a high prevalence of OSA in this population. Prospective longitudinal studies are needed to evaluate the outcomes of OSA in PRS population.

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Bedside Polysomnography as an Adjunct in the Management of Infants with Robin Sequence

Cited by 48 publications

References 0 publications

Late presentation of upper airway obstruction in Pierre Robin sequence

Late presentation of upper airway obstruction in Pierre Robin sequence

Craniofacial Syndromes

Polysomnographic findings in infants with Pierre Robin sequence

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