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

Purpose To evaluate the impact of the COVID-19 pandemic on non-invasive positive airway pressure (PAP) usage among children with sleep-disordered breathing (SDB). Methods PAP usage data in children with SDB aged 1 to 18 years old at The Hospital for Sick Children, Canada, were analyzed. The PAP usage data were recorded for 3 months prior to and 3 months following the COVID-19 lockdown in Ontario, Canada. The primary outcomes of interest were (i) percentage of days that PAP was used for ≥ 4 h and (ii) average daily usage of PAP based on days when PAP was used. Results A total of 151 children were included. The mean (± SD) age and BMI were 12.6 ± 4.1 years and 28.7 ± 12.4 kg/m 2 , respectively. The median (IQR) percentage of days of PAP usage for ≥ 4 h and average nightly PAP usage was significantly higher during compared with prior to the pandemic (76.7 [19.0–94.0] vs 62.0 [15.5–89.0]%, p = 0.02, and 406.0 [244.0–525.0] vs 367.0 [218.0–496.0] min, p = 0.006, respectively). Within this cohort, 95/151 (63%) children with SDB showed increased PAP usage and 56/151 (37%) either decreased the amount of time they used PAP or stopped PAP use altogether. Conclusions COVID-19 pandemic has provided opportunities for increased PAP usage in a significant number of children with SDB. A subset of children with prior evidence for suboptimal PAP usage showed further decreases in PAP usage during the pandemic. This information is critical for clinicians to provide anticipatory guidance to encourage PAP usage both during the pandemic and beyond. Supplementary Information The online version contains supplementary material available at 10.1007/s11325-021-02409-w.

Positive airway pressure (PAP) therapy is an effective treatment prescribed to children with sleep disordered breathing (SDB); however, PAP adherence remains challenging. Given that COVID-19 pandemic continues to impact sleep and daily life, the aim of this study was to evaluate longitudinal trajectory of PAP usage in children during the COVID-19 pandemic. Patients and Methods: This was a retrospective study. Children aged 1-18 years with SDB prescribed PAP at The Hospital for Sick Children (Toronto, Canada) were evaluated for PAP adherence. Demographics, medical history and PAP adherence data during four consecutive 3-month time periods from December 2019 to December 2020 were collected. These four time periods included i) prior to COVID-19 lockdown, ii) during the first three months of lockdown, iii) summer and iv) return to school period. Percentage of days where PAP was used for ≥4 hours and average nightly usage of PAP were primary outcomes. Results: A total of 149 children (61.7% male, mean (±SD) age=12.8 ± 4.1 years, BMI (±SD) z-score=1.45±1.43) were enrolled. Compared to prior to lockdown, the median (IQR) of percentage of PAP usage ≥4 hours and average nightly usage of PAP declined significantly during the summer and return to school periods (p<0.001 for all). By the end of the return to school period, only 69/149 (46%) showed sustained PAP usage and 80/149 (54%) had decreased PAP usage. Obesity was a risk factor for a decline in PAP usage after returning to school (β=−15.36, p=0.03). Conclusion: Compared to COVID-19 pre-pandemic PAP usage, there was a significant decline in PAP usage across COVID-19 pandemic. There is critical under usage of PAP in children diagnosed with SDB, resulting in an urgent need to address barriers to mitigate poor adherence to PAP long-term. Targeted strategies are required to optimize PAP adherence in children with SDB.

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