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Sleep Outcomes in Neonates with Pierre Robin Sequence Undergoing External Mandibular Distraction: A Longitudinal
Zarmina Ehsan1, K Nicole Weaver1, Brian S Pan1
1From the Division of Pulmonary and Sleep Medicine, Children's Mercy-Kansas City; and the Divisions of Human Genetics, Plastic Surgery, Biostatistics and Epidemiology, and Pulmonary and Sleep Medicine, Cincinnati Children's Hospital Medical Center.
Background:
Robin sequence is a common cause of upper airway obstruction in newborns. Herein, we report sleep outcomes in neonates undergoing external mandibular distraction osteogenesis.
Methods:
In this retrospective, 14-year, single-institution study of neonates with Robin sequence undergoing mandibular distraction osteogenesis, we compare respiratory parameters and sleep architecture before versus after surgery.
Results:
Thirty-one neonates were included; age was 13 days (interquartile range, 5 to 34 days) at preoperative polysomnography and 80 days (interquartile range, 50 to 98 days) at postoperative polysomnography. All neonates had severe obstructive sleep apnea at baseline (defined as pre-operative obstructive apnea hypopnea index ≥ 10). Postoperatively, there was a significant reduction in obstructive apnea hypopnea index [38.3 (interquartile range, 23.4 to 61.8) preoperatively versus 9.4 (interquartile range, 5.3 to 17.1) postoperatively; p < 0.0001], and a significant improvement in sleep efficiency and oxygen saturation nadir. Although 26 neonates (84 percent) had a 50 percent reduction in obstructive apnea hypopnea index postoperatively, all neonates had obstructive sleep apnea, and 15 neonates (48 percent) had persistent severe obstructive sleep apnea following surgery.
Conclusions:
We report the largest cohort of sleep outcomes in neonates with Robin sequence and severe obstructive sleep apnea undergoing external mandibular distraction osteogenesis. Although the severity of obstructive sleep apnea improves postoperatively, the disease persists in all neonates. We propose that neonates undergo polysomnography before and soon after mandibular distraction osteogenesis to objectively assess improvement in obstructive sleep apnea, as they may require additional evaluation for sites of multilevel airway obstruction and treatment.
Clinical Question/Level Of Evidence:
Therapeutic, IV.
