Unexplained Significant Central Sleep Apnea in Infants: Clinical Presentation and Outcomes

Objective Unexplained significant central sleep apnea in term infants presents as central apneas with associated oxygen desaturations requiring respiratory support and monitoring for prolonged periods. However, there is a paucity of literature describing idiopathic central sleep apnea (ICSA) in term or near-term infants. Our aim was to describe the clinical manifestations, polysomnography data, interventions, and trajectory of ICSA in infants. Design This is a retrospective study of infants (gestational age >= 35 weeks) who presented with significant central apneas and were subsequently diagnosed with ICSA following polysomnography and clinical investigations between January 2011 and April 2021 at a tertiary care hospital in Canada. Polysomnography data, clinical investigations, and treatments were documented. Results Eighteen infants (male, 78%; median gestational age 38 weeks) with ICSA were included. Initial polysomnograms were completed at a median (interquartile range [IQR]) age of 1.2 (0.6-1.6) months (n =18) and follow-up polysomnograms at 12.4 (10.6-14.0) months (n =13). Compared to baseline diagnostic polysomnograms, at follow-up there was a significant reduction in the median (IQR) central apnea-hypopnea index (26.1 [18.2-52.9] versus 4.2 [2.6-7.2] events/hour; p= 0.001), denaturation index (30.9 [12.2-57.4] versus 3.9 [3.0-7.9] events/hour; p = 0.002), average transcutaneous carbon dioxide (41.9 [40.1-47.3 versus 39.4 [37.5-42.7] mmHg; p=0.025), and improved nadir oxygen saturation (79.8 [69.1-83.0] versus 85.5 [83.2-87.8]%; p =0.033), respectively. Prescribed treatments included supplemental oxygen (14/18, 78%), caffeine (5/18, 28%), and noninvasive ventilation (1/18, 6%). Conclusions Infants with significant unexplained ICSA have a favorable clinical trajectory over time. Further research is needed to understand the etiology of this rare disorder.