Sleep architecture in Pierre-Robin sequence: The effect of mandibular distraction osteogenesis

Abstract

Introduction: Pierre-Robin Sequence (PRS), a triad of micro/retrognathia, glossoptosis, and upper airway obstruction, usually in conjunction with a cleft palate is frequently associated with significant morbidity. Mandibular distraction osteogenesis (MDO) is an effective treatment modality to address retroglossal upper airway obstruction by increasing the anterior-posterior diameter of the infant airway. Although MDO has been shown to improve the apnea-hypopnea index (AHI) in children with PRS, the consequences of MDO on other aspects of infant sleep, including hypercapnea, hypoxia, the REM to Non-REM ratio, as well as its effect on central and mixed apneas has not been investigated with an adequate sample size.

Objective: To characterize the effect of MDO on key components of sleep architecture in infants with PRS.

Methods: Charts from 32 infants with PRS that were addressed with MDO at our tertiary-care children's hospital were retrospectively reviewed. Of these, 26 infants (57.7% male; mean age = 4.1 weeks, SD = 5.0) had pre- and post-operative polysomnograms (PSG). Paired samples t-tests were used to compare pre- and post- MDO sleep architecture mean score differences.

Results: Among the 26 infants, 73.1% demonstrated severe pre-MDO sleep apnea (AHI > 10). Several aspects of sleep architecture were found to improve post-operatively. Significant improvements were found in AHI (30.3 vs. 8.7; t = 4.1, p < 0.001), obstructive apneas (79.3 vs. 5.8; t = 4.0, p < 0.001), hypopneas (48.1 vs. 22.1; t = 2.2, p = 0.040), time spent below 90% SpO2 (3.9% vs. 0.7%; t = 3.3, p = 0.003), and lowest SpO2 nadir (75.4% vs. 82.9%; 3.4, p = 0.002). In addition, a marginally significant improvement was found for mixed apnea (6.3 vs. 1.6; t = 1.99, p = 0.058).

Conclusion: MDO improves several sleep architecture parameters in this sample of infants with PRS. Statistically significant improvement was seen in obstructive apneas, hypopneas, AHI, obstructive AHI, and several indicators of hypoxia during sleep.

Keywords: Central sleep apnea; Glossoptosis; Mandibular distraction osteogenesis; Obstructive sleep apnea; Pierre-Robin sequence; Polysomnography; Sleep-disordered breathing; Upper airway obstruction.