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Published on: December 6, 2016
Characteristics of sleep apnea in infants with Pierre-Robin sequence: Is there improvement with advancing age?
Jake J Lee1, Prasad J Thottam2, Matthew D Ford3
1University of Pittsburgh School of Medicine, Department of Otolaryngology, 3550 Terrace St, Pittsburgh, PA 15261, United States.
Insights
Infants with Pierre-Robin sequence (PRS) do not show decreased sleep apnea severity with age. Mandibular distraction osteogenesis (MDO) significantly improves both obstructive and central sleep apnea in these infants.
Area of Science:
- Pediatric Otolaryngology
- Sleep Medicine
- Craniofacial Surgery
Background:
- Pierre-Robin sequence (PRS) is associated with upper airway obstruction and sleep-disordered breathing.
- Obstructive sleep apnea (OSA) and central sleep apnea (CSA) are common in infants with PRS.
- The natural course of sleep apnea severity with age in PRS is not well-established.
Purpose of the Study:
- To evaluate changes in OSA and CSA in infants with PRS as they age.
- To assess the impact of mandibular distraction osteogenesis (MDO) on sleep apnea in infants with PRS.
Main Methods:
- Retrospective chart review of 141 infants with PRS.
- Polysomnography (PSG) data analyzed for apnea-hypopnea index (AHI), obstructive apnea-hypopnea index (OAHI), and central apnea index (CAI).
- Comparison of pre- and post-operative PSG data in 9 patients who underwent MDO.
Main Results:
- 80% of infants with PRS had severe sleep apnea (AHI≥10) at initial PSG.
- No significant decrease in AHI, OAHI, or CAI was observed with increasing age up to one year.
- MDO significantly reduced AHI, OAHI, CAI, and improved oxygen saturation (SaO2) nadir and percentage of time with SaO2 <90%.
Conclusions:
- Unlike in non-PRS populations, sleep apnea severity does not naturally decrease with age in infants with PRS.
- MDO is an effective surgical intervention for improving both obstructive and central sleep apnea in infants with PRS.
Objectives:
To investigate changes in obstructive sleep apnea (OSA) and central sleep apnea (CSA) in infants with Pierre-Robin sequence (PRS) with advancing age and after mandibular distraction osteogenesis (MDO).
Methods:
Charts from 141 infants with PRS that presented to our tertiary-care children's hospital between 2005 and 2015 were retrospectively reviewed. Forty-five patients received a polysomnogram (PSG) prior to surgical intervention. Linear regression was utilized to compare age at pre-operative PSG with apnea-hypopnea index (AHI), obstructive apnea-hypopnea index (OAHI), and central apnea index (CAI). We then analyzed a subset of 9 patients who underwent MDO with pre- and post-operative PSGs. Wilcoxon signed-rank test was utilized to examine differences in pre- and post-operative OSA and CSA scores.
Results:
Forty-five patients received pre-operative PSGs. Of these, 80.0% demonstrated severe sleep apnea (AHI≥10), 68.9% demonstrated severe obstructive sleep apnea (OAHI≥10), and 55.6% demonstrated central sleep apnea (CAI≥1). There was no significant pattern of decrease in AHI, OAHI, and CAI with increased age up to 1 year. Among the 9 patients who underwent MDO with pre- and post-operative PSGs, significant reductions in AHI, OAHI, CAI, and percentage of total sleep time with arterial oxygen saturation (SaO2) <90% and significant increases in SaO2 nadir were identified after MDO.
Conclusions:
Contrary to previously examined literature in non-PRS patients, we did not find a decreased severity of central or obstructive sleep apnea with advancing age. Infants with PRS who underwent MDO demonstrated significant decreases in both obstructive and central apnea indices.
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