Zarmina Ehsan1, Christopher Kurian2, K Nicole Weaver3, Brian S Pan4, Guixia Huang5, Md M Hossain5, Narong Simakajornboon6. 1. Division of Pulmonary and Sleep Medicine, Children's Mercy-Kansas City, Kansas City, Missouri. 2. University of Missouri Kansas City School of Medicine, Kansas City, Missouri. 3. Division of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio. 4. Division of Plastic Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio. 5. Division of Biostatistics and Epidemiology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio. 6. Division of Pulmonary and Sleep Medicine, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
Abstract
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.
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.
Authors: Iee Ching W Anderson; Ahmad R Sedaghat; Brian M McGinley; Richard J Redett; Emily F Boss; Stacey L Ishman Journal: Cleft Palate Craniofac J Date: 2010-08-11
Authors: Hanneke Basart; Frea H Kruisinga; Corstiaan C Breugem; J Peter W Don Griot; Raoul C Hennekam; Chantal M A M Van der Horst Journal: J Craniomaxillofac Surg Date: 2014-11-05 Impact factor: 2.078
Authors: M Rathé; M Rayyan; J Schoenaers; J T Dormaar; M Breuls; A Verdonck; K Devriendt; V Vander Poorten; G Hens Journal: Int J Pediatr Otorhinolaryngol Date: 2015-06-06 Impact factor: 1.675
Authors: Adele Karen Evans; Reza Rahbar; Gary F Rogers; John B Mulliken; Mark S Volk Journal: Int J Pediatr Otorhinolaryngol Date: 2006-01-26 Impact factor: 1.675
Authors: Edgar Soto; Shivani Ananthasekar; Srikanth Kurapati; Nathaniel H Robin; Cassi Smola; Mary Halsey Maddox; Carter J Boyd; René P Myers Journal: Ann Plast Surg Date: 2021-06-01 Impact factor: 1.763
Authors: Robrecht J H Logjes; Joanna E MacLean; Noor W de Cort; Christian F Poets; Véronique Abadie; Koen F M Joosten; Cory M Resnick; Ivy K Trindade-Suedam; Carlton J Zdanski; Christopher R Forrest; Frea H Kruisinga; Roberto L Flores; Kelly N Evans; Corstiaan C Breugem Journal: J Clin Sleep Med Date: 2021-08-01 Impact factor: 4.324