Objective: To characterize the clinical presentation of sleep-disordered breathing and respiratory patterns at rest and during a 6-min walk test (6MWT) in children with rapid-onset obesity, hypothalamic dysfunction, hypoventilation, and autonomic dysregulation (ROHHAD) syndrome. Methods: Retrospective study of children with ROHHAD who had a diagnostic baseline polysomnography, daytime cardiorespiratory monitoring at rest and a 6MWT. Polysomnography data were also compared with body mass index-, age-, and sex-matched controls. Results: Of the eight children with ROHHAD, all eight (100%) had obstructive sleep apnea (OSA) and 2/8 (25%) had nocturnal hypoventilation (NH) on their baseline polysomnography. Comparing the ROHHAD group to the control group, there were no significant differences in the median (interquartile range [IQR]) obstructive apnea
Study Objectives: Positional obstructive sleep apnea (POSA) is a phenotype of obstructive sleep apnea (OSA) where sleep-related obstructive events occur predominantly in the supine position. Limited knowledge exists regarding the presence of POSA in children with obesity. The study objective was to determine the prevalence of POSA while identifying factors associated with POSA in children with obesity. Methods: This was a cross-sectional study of children with obesity, aged 8 to 18 years, with a diagnostic polysomnogram (PSG) between 2012 to 2019, who were referred for the evaluation of sleep-related breathing. POSA was defined as an overall obstructive apnea-hypopnea index (OAHI) ≥5 events/h and a supine OAHI to nonsupine OAHI ratio of ≥2. Patient demographics, anthropometrics, and PSG data were recorded. Results: Of the 112 children with obesity with a diagnostic PSG, 43 (38%) had OSA. Among those with OSA, 25 of 43 (58%) had POSA (mean age: 14.6 ± 2.3 years; mean body mass index: 37.7 ± 7.6 kg/m 2 ; 68% male) and 18 of 43 (42%) had non-POSA (mean age: 13.9 ± 2.8 years; mean body mass index: 37.9 ± 7.2 kg/m 2 ; 78% male). Among those with POSA, 13 of 25 (52%) had mild OSA, 7 of 25 (28%) had moderate OSA, and 5 of 25 (20%) had severe OSA. No significant differences were found in age, sex, and anthropometric measures between POSA and non-POSA groups. Time spent in supine and nonsupine sleep did not differ significantly between groups. Conclusions: In children with obesity and OSA, POSA occurs frequently. Identifying POSA allows for potential targeted positional therapy for children with obesity.
OBJECTIVE Chiari malformation type I (CM-I) involves the herniation of the cerebellar tonsils through the foramen magnum. CM-I is associated with both obstructive sleep apnea (OSA) and central sleep apnea (CSA) in children. The primary management of symptomatic CM-I remains surgical decompression. There is, however, a paucity of data evaluating the efficacy of decompression surgery on outcomes related to sleep-disordered breathing (SDB). The objective of this study was to evaluate SDB outcomes, specifically the need for respiratory support following decompression in pediatric patients with CM-I. METHODS This was a retrospective chart review of all children diagnosed with CM-I when younger than 18 years of age who had polysomnography (PSG) studies pre- and postsurgery, between January 2008 and October 2018 at the Hospital for Sick Children in Toronto. Patient demographics, symptoms, PSG data, ongoing respiratory support, and surgical notes were recorded. Differences in PSG studies obtained pre- and postsurgery were compared using the Wilcoxon test for paired samples. RESULTS A total of 15 children with 15 interventions met inclusion criteria with pre- and postsurgery PSG studies and were considered for statistical analysis. Of the 15 subjects included for analysis, preoperative OSA was present in 2 (13.3%), CSA in 5 (33.3%), mixed SDB (both OSA and CSA) in 4 (26.7%), and no significant SDB in 4 (26.7%). Postoperatively, OSA was present in 3 (20.0%), CSA in 4 (26.7%), mixed SDB in 0 (0%), and no significant SDB in 8 (53.3%). The presence of severe OSA decreased from 4/15 (26.7%) to 2/15 (13.3%) postoperatively, and severe CSA decreased from 5/15 (33.3%) to 2/15 (13.3%) postoperatively. Following decompression surgery, 7/15 subjects (46.7%) required positive airway pressure for management of their SDB. Overall, significant improvements were observed in a number of respiratory parameters following decompression including the following: the total apnea-hypopnea index (AHI) (17.5 ± 48.2 vs 6.1 ± 32.7 events/hour; p = 0.001), obstructive AHI (2.1 ± 16.1 vs 1.0 ± 6.6 events/hour; p = 0.005), central AHI (6.3 ± 48.9 vs 2.7 ± 33.0 events/hour; p = 0.005), and the desaturation index (16.7 ± 49.6 vs 3.8 ± 25.3; p = 0.001). CONCLUSIONS Although decompression surgery led to a significant reduction in obstructive and central events, many children continued to have persistent SDB and required additional positive airway pressure therapy. This information is important and relevant for anticipatory guidance around decompression surgery and the necessity for respiratory support for the management of SDB in pediatric patients with CM-I.
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