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Volume 15 No. 03
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Accepted Papers





Scientific Investigations

Longitudinal Sleep Outcomes in Neonates With Pierre Robin Sequence Treated Conservatively

Zarmina Ehsan, MD1; Christopher Kurian, MS2; K. Nicole Weaver, MD3; Brian S. Pan, MD4; Guixia Huang, MS5; Md M. Hossain, PhD5; Narong Simakajornboon, MD6
1Division of Pulmonary and Sleep Medicine, Children's Mercy-Kansas City, Kansas City, Missouri; 2University of Missouri Kansas City School of Medicine, Kansas City, Missouri; 3Division of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio; 4Division of Plastic Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio; 5Division of Biostatistics and Epidemiology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio; 6Division of Pulmonary and Sleep Medicine, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio

Study Objectives:

Although Pierre Robin sequence (PRS) is a major cause of neonatal obstructive sleep apnea (OSA), longitudinal studies reporting evolution with age are lacking. This study aimed to describe changes in sleep-related respiratory parameters and sleep architecture in neonates with PRS treated conservatively (defined for this paper as treatment without tracheostomy or mandibular distraction).

Methods:

A retrospective, 14-year, single-institution study of neonates with PRS who underwent diagnostic polysomnography (PSG) and at least one follow-up PSG. Those treated with surgery were excluded. Data were analyzed using a mixed-effects model with subject-specific random effect.

Results:

In a cohort of 21 infants, baseline PSG (mean age 0.9 ± 0.7 months) showed a total apnea-hypopnea index (AHI) of 24.3 ± 3.6 events/h, obstructive apnea-hypopnea index (OAHI) of 13.4 ± 1.6 events/h, central apnea index of 10.2 ± 3.2 events/h, and an arousal index of 28.3 ± 1.3 events/h (variables reported as least square means ± standard error of the mean). There was a significant reduction in AHI, OAHI, arousal index, and percentage of REM sleep with advancing age. Although 71% of infants achieved full oral feeds by one month of age, some infants remained underweight during infancy.

Conclusions:

These neonates with PRS and OSA, treated conservatively, had an improvement in OAHI with advancing age with the median age of OSA resolution at 15 months. Factors potentially responsible include craniofacial growth and maturational changes of respiratory control. Further studies are necessary to determine the long-term effects of conservative management on growth and neurodevelopmental outcomes in these infants.

Citation:

Ehsan Z, Kurian C, Weaver KN, Pan BS, Huang G, Hossain MM, Simakajornboon N. Longitudinal sleep outcomes in neonates with Pierre Robin sequence treated conservatively. J Clin Sleep Med. 2019;15(3):477–482.




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