Comparison of high flow nasal cannula therapy to nasal oxygen as a treatment for obstructive sleep apnea in infants.

INTRODUCTION: Obstructive sleep apnea (OSA) in infants is treated with low flow oxygen via nasal cannula (NC), CPAP (continous positive airway pressure), or surgery. Literature supports the use of high flow NC (HFNC) in children in the outpatient setting, however there is limited data on the use of HFNC in infants. OBJECTIVE: The purpose of this study was to compare HFNC and low-flow oxygen as treatments for OSA in infants. METHODS: A prospective pilot study was performed at two institutions. Infants with primarily OSA underwent a 3-4 h sleep study with HFNC titration at 6-14 lpm for OSA, followed by clinical polysomnography (PSG) for oxygen titration (1/8-1 lpm). Infants with primarily central apnea were excluded. RESULTS: Nine infants were enrolled, with a mean age of 1.3 ± 1.7 months. Average apnea hypopnea index (AHI), average obstructive apnea hypopnea index (OAHI) and average central apnea index during the diagnostic PSG was 17.2 ± 7/h, 13.4 ± 5.4/h and 3.7 ± 4.8/h respectively. OSA improved in 44.4% of subjects with HFNC; the mean AHI and OAHI decreased from 15.6 ± 5.65/h and 12.8 ± 4.4/h on diagnostic PSG to 5.12 ± 2.5/h and 4.25 ± 2.5/h on titration PSG. OSA improved universally with low flow oxygen; the mean AHI decreased from 17.2 ± 7/h on diagnostic PSG to 4.44 ± 3.6/h on titration PSG. CONCLUSION: HFNC reduced OSA in some infants, though low flow oxygen reduced OSA in all subjects. Respiratory instability (high loop gain) in infants may explain why infants responded to low flow oxygen. More studies are needed to determine if HFNC is beneficial in selected groups of infants with OSA.

Melatonin use in managing insomnia in children with autism and other neurogenetic disorders - An assessment by the international pediatric sleep association (IPSA).

Though it is widely prescribed for improving sleep of children with autism and other neurogenetic disorders, there is a need for practical guidance to clinicians on the use of melatonin for managing insomnia in this population. Because data were either lacking or inconclusive, a task force was established by the International Pediatric Sleep Association (IPSA) to examine the literature based on clinical trials from 2012 onwards. A summary of evidence pertaining to melatonin's utility and potential side effects, practice-related caveats, and insights for use are published herewith.

Polysomnographic parameters and clinical risk factors predicting postoperative respiratory complications in children undergoing supraglottoplasty.

STUDY OBJECTIVES: Postoperative respiratory complications (PORCs) can occur following supraglottoplasty (SGP) for obstructive sleep apnea. However, there are very limited data on risk factors associated with these complications. This study aims to evaluate the occurrence of PORC in children undergoing SGP and to assess clinical factors and polysomnographic parameters predicting these complications. METHODS: A retrospective study was performed in children with laryngomalacia who underwent SGP with preoperative polysomnography. RESULTS: 400 children who underwent SGP met the criteria for entry into the analysis with a total of 416 surgeries with corresponding preoperative polysomnography. The median age (interquartile range) at the time of polysomnography was 0.4 (0.2, 1.5) years. A total of 96 (23.1%) PORCs were noted. Compared with those without complications, children with PORCs had a higher proportion of congenital heart disease (P < .05), higher median obstructive apnea-hypopnea index (obstructive AHI; median 16.0 vs 11.4 events/h; P < .01), and lower median oxygen saturation (SpO2) (P < .001). The unadjusted odd ratio indicated an increased risk of PORCs in children with congenital heart disease (odds ratio 1.66; P < .05) and those with an obstructive AHI > 10 events/h (odds ratio 2.06; P < .01). Multiple regression analysis demonstrated that an obstructive AHI > 10 events/h was the only independent risk factor for PORCs (P < .05). CONCLUSIONS: In our cohort of children with laryngomalacia undergoing SGP, those with underlying congenital heart disease, higher obstructive AHI, and lower SpO2 were more likely to develop PORCs. Only children with an obstructive AHI > 10 events/h were at significantly increased risk for PORCs following SGP. Preoperative polysomnography is useful in preoperative planning in children undergoing SGP. CITATION: Kanavitoon S, Ngamprasertwong P, Gurbani N, et al. Polysomnographic parameters and clinical risk factors predicting postoperative respiratory complications in children undergoing supraglottoplasty. J Clin Sleep Med. 2024;20(1):9-16.

Comparing the Driving Skills of Adolescents with Obstructive Sleep Apnea to Healthy Controls: The Results of a Case-Controlled Observational Study.

Auto crashes are a leading cause of death and injury among adolescents. Untreated obstructive sleep apnea (OSA) can cause sleepiness and inattention, which could negatively impact novice drivers, but OSA-related studies have focused on older drivers. This study used a driving simulator to examine whether licensed 16-19-year-old adolescents with OSA have diminished driving skills. Twenty-one adolescents with OSA and twenty-eight without OSA (both confirmed using polysomnography) completed two randomly ordered driving trials in a simulator (with induced distractions versus without). A mixed ANOVA examined the between-subjects effect of the OSA group, the within-subjects effect of the distraction condition, and the group-by-condition interaction effect on the ability to maintain lane position and the frequency of extended eye glances away from the roadway. T-tests were also used to examine group differences in reported sleepiness and inattention during daily life. The distraction task increased extended off-road glances and difficulties maintaining lane position (p < 0.001). However, adolescents with OSA did not display worse eye glance or lane position than controls and there were no significant group-by-condition interactions. Although the groups differed on polysomonographic features, there were also no significant differences in reported sleepiness or inattention. The distraction task negatively impacted both groups of adolescent drivers, but those with OSA did not fare differentially worse. Most adolescents in our study had mild OSA (median obstructive apnea-hypopnea index = 4.4), the most common form in the community. It remains possible that youth with more severe OSA would show increased driving impairment.

Occurrence of sleep-disordered breathing in children with pulmonary hypertension.

BACKGROUND: Obstructive sleep apnea (OSA) has been associated with pulmonary hypertension (PH) through a complex and bidirectional relationship. The prevalence of OSA and other forms of sleep-disordered breathing (SDB) among children with PH is not well understood. A better understanding could help elucidate contributing factors for PH, guide diagnostic testing, and inform therapies. We therefore determined the proportion of SDB codiagnosis and OSA risk factors among a cohort of children with PH. METHODS: Retrospective chart review was performed for children 0-21 years old with PH who underwent polysomnography (PSG) between 1 January 2010 and 31 August 2020. The primary outcome was OSA occurrence and risk based on demographics, diagnoses, and PH classification. The secondary outcome was occurrence and risk of other SDB diagnoses based on PH classification. RESULTS: Among 89 children identified, diagnoses included OSA (N = 79, 89%), central sleep apnea (N = 11, 12%), sleep-related hypoventilation (N = 6, 7%), and non-apneic hypoxemia (N = 28, 31%). Trisomy 21 diagnosis (N = 38, 43%) was associated with increased OSA risk (RR: 1.24, 95% CI: [1.09-1.42]) and age >12 months at PSG was associated with decreased OSA risk (N = 52, 58%, RR: 0.60, 95% CI: [0.45-0.81]). There was no difference in SDB risk based on Group 1 (N = 56, 63%) or Group 3 (N = 33, 37%) PH and no difference in OSA severity based on demographics and diagnoses. CONCLUSION: OSA was diagnosed among a majority of children with PH, and other SDB diagnoses were identified in a significant proportion of children. These findings support routine screening for SDB with PSG in children with PH.

Increased incidence of pediatric narcolepsy following the 2009 H1N1 pandemic: a report from the pediatric working group of the sleep research network.

This study was aimed to evaluate the yearly incidence of pediatric narcolepsy prior to and following the 2009 H1N1 pandemic and to evaluate seasonal patterns of narcolepsy onset and associations with H1N1 influenza infection in the United States. This was a multicenter retrospective study with prospective follow-up. Participants were recruited from members of the Pediatric Working Group of the Sleep Research Network including 22 sites across the United States. The main outcomes were monthly and yearly incident cases of childhood narcolepsy in the United States, and its relationship to historical H1N1 influenza data. A total of 950 participants were included in the analysis; 487 participants were male (51.3%). The mean age at onset of excessive daytime sleepiness (EDS) was 9.6  ±â€… 3.9 years. Significant trend changes in pediatric narcolepsy incidence based on EDS onset (p  <  .0001) occurred over the 1998-2016 period, peaking in 2010, reflecting a 1.6-fold increase in narcolepsy incidence. In addition, there was significant seasonal variation in narcolepsy incident cases, with increased cases in spring (p  <  .05). Cross-correlation analysis demonstrated a significant correlation between monthly H1N1 infection and monthly narcolepsy incident cases (p  =  .397, p  <  .0001) with a lag time of 8 months. We conclude that there is a significant increase in pediatric narcolepsy incidence after the 2009 H1N1 pandemic in the United States. However, the magnitude of increase is lower than reported in European countries and in China. The temporal correlation between monthly H1N1 infection and monthly narcolepsy incidence, suggests that H1N1 infection may be a contributing factor to the increased pediatric narcolepsy incidence after the 2009 H1N1 pandemics.

Large body movements on video polysomnography are associated with daytime dysfunction in children with restless sleep disorder.

Restless sleep disorder (RSD) is a newly defined sleep-related movement disorder characterized by large muscle movements (LMM) in sleep. We examined the sleep study, clinical characteristics, and daytime functioning in children with RSD and compared them to children with periodic limb movement disorder (PLMD) or restless legs syndrome (RLS). Video polysomnography from 47 children with restless sleep was retrospectively reviewed for LMM and age- and sex-matched to 34 children with PLMD and 12 children with RLS. Data examined included PSG characteristics, ferritin, Pediatric Quality of Life (PedsQL), and Epworth Sleepiness Scale (ESS). Fourteen children met the clinical criteria for RSD with an LMM index of 5 or more per hour of sleep. Mean ESS was elevated in patients with RSD compared to either the PLMD or RLS groups though the result did not reach statistical significance (RSD = 10.20 ± 6.81, PLMD = 6.19 ± 4.14, RLS = 6.25 ± 4.90). The PedsQL score was significantly decreased in the RLS group compared to RSD and was reduced overall in all three groups (PedsQL Total RSD = 70.76 ± 18.05, PLMD = 57.05 ± 20.33, RLS = 53.24 ± 16.97). Serum ferritin values were similar in all three groups (RSD = 26.89 ± 10.29, PLMD = 33.91 ± 20.31, RLS = 23.69 ± 12.94 ng/mL, p = ns). Children with RSD demonstrate increased daytime sleepiness compared to PLMD or RLS and all three disease groups showed decreased quality of life. Further studies are needed to examine long-term consequences of RSD.

Narcolepsy in Children: Sleep disorders in children, A rapidly evolving field seeking consensus.

Narcolepsy is a life-long sleep disorder with two distinct subtypes, narcolepsy type I and narcolepsy type II. It is now well recognized that the loss of hypocretin neurons underlies the pathogenesis of narcolepsy type I, however, the pathogenesis of narcolepsy type II is currently unknown. Both genetic and environmental factors play an important role in the pathogenesis of narcolepsy. There is increasing evidence that autoimmune processes may play a critical role in the loss of hypocretin neurons. Infections especially streptococcus and influenza have been proposed as a potential trigger for the autoimmune-mediated mechanism. Several recent studies have shown increased cases of pediatric narcolepsy following the 2009 H1N1 pandemic. The increased cases in Europe seem to be related to a specific type of H1N1 influenza vaccination (Pandemrix), while the increased cases in China are related to influenza infection. Children with narcolepsy can have an unusual presentation at disease onset including complex motor movements which may lead to delayed diagnosis. All classic narcolepsy tetrads are present in only a small proportion of children. The diagnosis of narcolepsy is confirmed by either obtaining cerebrospinal fluid hypocretin or overnight sleep study with the multiple sleep latency test (MSLT). There are limitations of using MSLT in young children such that a negative MSLT test cannot exclude narcolepsy. HLA markers have limited utility in narcolepsy, but it may be useful in young children with clinical suspicion of narcolepsy. For management, both pharmacologic and non-pharmacologic treatments are important in the management of narcolepsy. Pharmacotherapy is primarily aimed to address excessive daytime sleepiness and REM-related symptoms such as cataplexy. In addition to pharmacotherapy, routine screening of behavioral and psychosocial issues is warranted to identify patients who would benefit from bio-behavior intervention.

Utility of polysomnography for management of chronic invasive mechanical ventilation in children.

OBJECTIVE: With advances in technology, pediatric patients are sent home with chronic invasive mechanical ventilation (CIMV). Ventilation optimization and preparation for weaning are essential processes, however, there is no standard approach to weaning and titrating ventilator settings in children. There is little information in the literature on the utility of polysomnography (PSG) in weaning and titration of CIMV. Our objective is to review the role of PSG on titration and weaning of CIMV in children. METHODS: A retrospective review of medical records and polysomnograms was performed on CIMV dependent patients at Cincinnati Children's Hospital Medical Center from January 2011 to October 2017. Patients underwent overnight ventilator weaning or titration PSG. Subjects with less than 3 h of total sleep time were excluded. RESULTS: About 163 PSGs were divided into 97 PSGs for ventilator titration and 66 PSGs for ventilator weaning. Of the 97 ventilator titration PSGs, 59 (60.8%) had inadequate ventilation, 10 (10.3%) had inadequate oxygenation, 22 (22.7%) had significant tracheostomy leak, 13 (13.4%) had autocycling, 3 (3.1%) had failure to trigger, 4 (4.1%) had central sleep apnea, and 24 (25.8%) had adequate ventilation on current ventilator settings. Of the 66 ventilator weaning PSGs, 48 (72.7%) weaned to lower ventilator settings or off ventilator support and 18 (27.3%) did not wean. DISCUSSION: Our results indicate that PSG is useful in titration and weaning of ventilator settings in children with CIMV. Larger studies are needed to evaluate the subgroup of this population who would benefit most from PSG for assessment of adequate ventilator support.

Validation of pediatric sleep questionnaire in children with Chiari malformation and/or spina bifida with or without myelomeningocele.

Sleep-disordered breathing (SDB) is common in Chiari Malformation (CM) and Spina Bifida (SB) and can lead to adverse consequences if untreated. Therefore, screening is very important but the Pediatric Sleep Questionnaire (PSQ) has not been validated in this population. Further, there is limited data on the validity of this tool in children with central nervous system malformations. Retrospective chart review of CM and SB patients evaluated in our sleep center between 2008 and 2018. The sensitivity, specificity, positive predictive value (PPV), and negative predictive value (NPV) of the PSQ and several of its components were calculated to predict obstructive sleep apnea (OSA). A total of 149 patients met criteria for analysis. The majority were referred to a sleep specialist due to concern for SDB. OSA was found in 36% (53/149) of all patients. The sensitivity and specificity of the PSQ to predict OSA was 73.58% and 20.83%, respectively. The PPV was 33.91%, and the NPV was 58.82%. Specificity values were higher for PSQ as negative predictors of moderate or severe OSA. In this population, the sensitivity of PSQ for OSA is reasonable but lower than values described in other populations. The specificity and NPV are low. Even with a high prevalence of OSA, symptoms of SDB may overlap with those of other comorbidities leading to a low specificity. A PSQ could be used to prioritize which patients need a PSG more urgently than others. Further studies are needed to define an optimal cut-off value of the PSQ in this population.

Sleep disordered breathing in children receiving vagus nerve stimulation therapy.

OBJECTIVE: The effects of vagus nerve stimulation (VNS) on sleep disordered breathing (SDB) have been reported in limited case series. Detailed studies, particularly in the pediatric population, have not been performed. The primary purpose of this study is to describe clinical characteristics, polysomnographic findings, and management of children treated with VNS. METHODS: A retrospective review of medical records and polysomnography data was performed in patients ages 0-20 years old receiving VNS therapy for refractory epilepsy at Cincinnati Children's Hospital Medical Center. RESULTS: 22 subjects met the inclusion criteria. 50% were male. The mean age at the time of VNS insertion was 8.4 ± 4.0 years. The mean age at the first PSG was 10.6 ± 4.3 years. Common presentations to sleep clinics included snoring (77.3%), frequent nighttime awakening (68.1%), and parasomnias (63.6%). The median apnea-hypopnea index (AHI) was 4.5/hr (IQR 3.0-13.1) and the median obstructive index (OI) was 4.1/hr (1.5-12.8). Obstructive sleep apnea (OSA) was diagnosed after VNS insertion in 19 patients (86.4%), 8 of which (36.3%) had severe OSA. Six patients (27.3%) had significant hypoventilation. For management, 6 patients (27.2%) were treated with bilevel PAP, 3 patients (13.6%) with CPAP, 2 patients (9.1%) with ventilator, 4 patients (18.2%) with upper airway surgeries, and 9 patients (40.9%) received medications only. CONCLUSIONS: SDB is common in pediatric patients with medically refractory epilepsy managed with VNS who were referred to sleep medicine clinics. Both OSA and nocturnal alveolar hypoventilation are relatively common in this population. Management of SDB often involves the use of positive airway pressure therapy or upper airway surgeries. Further studies are needed to assess the prevalence, risk factors, and the effect of treatments on epilepsy control. This study highlights the need for screening of SDB prior to and following VNS implantation.

Restless sleep in children: A systematic review.

This systematic review assessed the prevalence of restless sleep in children, documented the association of restless sleep with other conditions, and summarized the existing evidence regarding whether restless sleep should be considered a distinct sleep disorder. A comprehensive search of electronic databases was performed using the broad search term "restless sleep" in all fields. Of the 266 articles retrieved, 107 were retained for inclusion in this review. The majority (n = 93) were observational studies. The studies were grouped under several pathologic/condition categories: sleep-disordered breathing (n = 19); adenotonsillectomy (n = 7); respiratory disorders, otitis media, and smoke exposure (n = 12); sleep-related movement disorders and restless sleep disorder (n = 11); neurologic or psychiatric disorders (n = 7); Down syndrome/other neurodevelopmental disorders (n = 10); sleep-related bruxism and other sleep disorders (n = 7); and restless sleep in the general population/mixed clinical samples (n = 18). A high prevalence of restless sleep was found in children with many of these underlying conditions, likely related to associated inherent sleep disruption and frequent awakenings (e.g., apnea and periodic limb movements), pain, sleep instability, and caregiver perception. The majority of studies identified restless sleep as reported by the caregiver, only 34 studies attempted to define restless sleep further. Four studies provided supportive evidence for designating restless sleep as an independent sleep disorder, restless sleep disorder (RSD). This review highlights the fact that the prevalence, etiology and sequelae (including daytime impairments) of restless sleep in children are important topics deserving of further research and that clinical definitions based on empirical evidence need to be developed. The designation of "primary" versus "secondary" restless sleep may be a useful construct, especially with regard to developing clinical trials and treatment algorithms.

The maturation changes of sleep-related respiratory abnormalities in infants with laryngomalacia.

STUDY OBJECTIVES: Obstructive sleep apnea (OSA) and central sleep apnea (CSA) are common in infants with laryngomalacia. The purpose of this study was to evaluate developmental changes in sleep-related breathing disorders over time in infants with laryngomalacia and understand the effect of supraglottoplasty (SGP) and nonsurgical treatment. METHODS: This is a retrospective review of infants with laryngomalacia who had at least 2 diagnostic polysomnography studies performed from January 2000 and May 2015. We included infants who had either OSA or CSA. Comparison of sleep and respiratory parameters by age group (0-6, 6-12, and >12 months old) was performed in both SGP and non-SGP groups using a mixed-effect regression model. A log-normal mixed model was used to explore the changes in sleep and respiratory parameters with age. The time to resolution of CSA and OSA was analyzed using nonparametric survival analysis. RESULTS: A total of 102 infants were included; 57 had only OSA and 45 had both CSA and OSA. There were significant decreases in apnea-hypopnea index, obstructive index, central apnea index, and arousal index with increasing age in both SGP and non-SGP groups. The mean age at resolution of CSA (central apnea index < 5) was 7.60 months old for SGP and 12.57 months old for non-SGP (P < .05). There were no significant differences in the mean age at resolution of OSA (obstructive index < 1; 35.18 [SGP] vs 41.55 months [non-SGP]; P = .60) between SGP and non-SGP groups. Infants with neurologic disease, congenital anomalies, or genetic syndromes required significantly more time to resolve OSA (28.12 [normal] vs 53.13 [neurological] vs 59.53 months [congenital anomalies and genetic]; P < .01). CONCLUSIONS: Both OSA and CSA improve in infants with laryngomalacia with increasing age regardless of SGP. The mechanism underlying these changes may involve airway growth and maturation of respiratory control. Time to resolution of OSA is affected by the presence of neurologic diseases, congenital anomalies, and genetic syndromes. Further studies are needed to confirm these findings and to evaluate long-term outcomes in this population.

Polysomnographic Outcomes After Observation for Mild Obstructive Sleep Apnea in Children Younger Than 3 Years.

OBJECTIVE: Mild obstructive sleep apnea (OSA), particularly in young children, is often treated with observation. However, there is little evidence regarding the outcomes with this approach. Our aim was to assess the impact of observation on sleep for children aged <3 years with mild OSA. STUDY DESIGN: Case-control study. SETTING: Pediatric tertiary care center. METHODS: We reviewed cases of children (<3 years old) diagnosed with mild OSA (obstructive apnea-hypopnea index, 1-5 events/h) who were treated with observation between 2012 and 2017 and had at least 2 polysomnograms performed 3 to 12 months apart. Demographic data and comorbid diagnoses were collected. RESULTS: Twenty-six children met inclusion criteria; their median age was 7.2 months (95% CI, 1.2-22.8). Nine (35%) were female and 24 (92%) were White. Their median body mass index percentile was 39 (95% CI, 1-76). Comorbidities included cardiac disease (42.3%), laryngomalacia (42.3%), allergies (34.6%), reactive airway disease (23.1%), and prematurity (7.7%). The obstructive apnea-hypopnea index significantly decreased from 2.7 events/h (95% CI, 1-4.5) to 1.3 (95% CI, 0-4.5; P = .013). There was no significant improvement in median saturation nadir (baseline, 86%; P = .76) or median time with end-tidal carbon dioxide >50 mm Hg (baseline, 0 minutes; P = .34). OSA resolved in 8 patients (31%) and worsened in 1 (3.8%). Only race was a significant predictor of resolution per regression analysis; however, only 2 non-White children were included. CONCLUSION: In our cohort, resolution of mild OSA occurred in 31% of patients treated with 3 to 12 months of observation. The presence of laryngomalacia, asthma, and allergies did not affect resolution. Larger studies are needed to better identify factors (including race) associated with persistent OSA and optimal timing of intervention for these children. LEVEL OF EVIDENCE: 4.

Consensus diagnostic criteria for a newly defined pediatric sleep disorder: restless sleep disorder (RSD).

BACKGROUND: Restless sleep is a frequent complaint in clinical practice and has been reported in the medical literature since the 1970s. Most often, it has been described in association with specific sleep or medical conditions. However, more recently, publications have emerged that describe a disorder characterized by restless sleep as the core feature. To assess this further, the International Restless Legs Syndrome Study Group (IRLSSG) appointed a task force composed of international sleep experts. METHODS: A committee of 10 sleep clinicians developed a set of 16 consensus questions to review, conducted a comprehensive literature search, and extensively discussed potential diagnostic criteria. The committee recommendations were reviewed and endorsed by the IRLSSG Executive Committee. RESULTS: Based on the medical literature and expert clinical experience, the task force found sufficient evidence to formulate diagnostic criteria for a clinical entity designated "restless sleep disorder" (RSD). Eight essential criteria were agreed upon, which include a complaint of restless sleep, observed large body movements during sleep, video-polysomnographic documentation of 5 or more large body movements/hour, occurrence at least three times a week for at least three months, clinically significant impairment, and differentiation from other conditions that might secondarily cause restless sleep. However, the current evidence limits application to ages 6-18 years. Diagnostic coding, addition to existing diagnostic nosologies, and name selection are discussed. CONCLUSIONS: Consensus diagnostic criteria for RSD have been developed, which are intended to improve clinical practice and promote further research.

Sleep Outcomes in Neonates with Pierre Robin Sequence Undergoing External Mandibular Distraction: A Longitudinal Analysis.

BACKGROUND: Robin sequence is a common cause of upper airway obstruction in newborns. Herein, we report sleep outcomes in neonates undergoing external mandibular distraction osteogenesis. METHODS: In this retrospective, 14-year, single-institution study of neonates with Robin sequence undergoing mandibular distraction osteogenesis, we compare respiratory parameters and sleep architecture before versus after surgery. RESULTS: Thirty-one neonates were included; age was 13 days (interquartile range, 5 to 34 days) at preoperative polysomnography and 80 days (interquartile range, 50 to 98 days) at postoperative polysomnography. All neonates had severe obstructive sleep apnea at baseline (defined as pre-operative obstructive apnea hypopnea index ≥ 10). Postoperatively, there was a significant reduction in obstructive apnea hypopnea index [38.3 (interquartile range, 23.4 to 61.8) preoperatively versus 9.4 (interquartile range, 5.3 to 17.1) postoperatively; p < 0.0001], and a significant improvement in sleep efficiency and oxygen saturation nadir. Although 26 neonates (84 percent) had a 50 percent reduction in obstructive apnea hypopnea index postoperatively, all neonates had obstructive sleep apnea, and 15 neonates (48 percent) had persistent severe obstructive sleep apnea following surgery. CONCLUSIONS: We report the largest cohort of sleep outcomes in neonates with Robin sequence and severe obstructive sleep apnea undergoing external mandibular distraction osteogenesis. Although the severity of obstructive sleep apnea improves postoperatively, the disease persists in all neonates. We propose that neonates undergo polysomnography before and soon after mandibular distraction osteogenesis to objectively assess improvement in obstructive sleep apnea, as they may require additional evaluation for sites of multilevel airway obstruction and treatment. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

Can overnight portable pulse oximetry be used to stratify obstructive sleep apnea risk in infants? A correlation analysis.

INTRODUCTION: There is limited evidence on the accuracy of oximetry in the evaluation of infant obstructive sleep apnea (OSA). We aimed to determine the utility of overnight oximetry to stratify infants at risk for OSA, to determine urgency for definitive screening with an overnight in-laboratory polysomnogram (PSG). METHODS: Retrospective single-institution cohort study of infants undergoing PSG and a separate overnight oximetry over an 8-year period. Correlations, using oximetry in both in-hospital (attended) or at-home (unattended) settings, for ODI410 (decrease in oxygen saturation ≥ 4% from baseline, duration ≥ 10 seconds) and ODI40 (duration > 0 second) with the obstructive apnea-hypopnea index (AHIo) were obtained. The area under the curve was calculated, and sensitivity and specificity values have been presented as receiver operating characteristic curves. RESULTS: Thirty-eight infants were included. The mean (SD) age (months) was 5.7 (3.9) at diagnostic PSG and 5.5 (3.7) at the time of oximetry. The mean AHIo for the entire cohort was 6.7 (6.2). The mean (SD) ODI40 was 8.6 (9.0) and the mean (SD) ODI410 was 5.4 (5.1).The correlation between ODI and AHIo was statistically significant for the cohort (ODI40 vs. AHIo [r = .59, P < .001] and ODI410 vs AHIo [r = .55, P = .0003]). Using an ODI40 cutoff of 3, the sensitivity, specificity, negative predictive value and positive predictive value for diagnosing OSA was: 86%, 40%, 50%, and 80% respectively for an AHIo greater than 2, and 100%, 35%, 100%, and 58% respectively for an AHIo greater than or equal to 5. CONCLUSION: There is a significant positive correlation between the ODI4 obtained from oximetry and the AHIo obtained from PSG in infants at risk for OSA. An ODI40 greater than 3 may be useful to stratify infants at risk for moderate to severe OSA when used in attended (in-hospital) or unattended (in-home) settings.

Children with Down syndrome and mild OSA: treatment with medication versus observation.

STUDY OBJECTIVES: Children with Down syndrome (DS) have a high prevalence of obstructive sleep apnea (OSA). Anti-inflammatory medications have been shown to be an effective treatment for mild OSA in otherwise healthy children. However, the efficacy in children with DS and mild OSA has not been investigated. Our aim was to examine the polysomnographic changes of children with DS and mild OSA treated with medication. METHODS: A retrospective chart review was performed in children with DS (< 18 years) and mild OSA (obstructive apnea-hypopnea index ≤ 5 events/h) diagnosed by polysomnography (PSG) between 2006 and 2018. Patients were included if they were treated with medications (intranasal corticosteroids and/or montelukast) or by observation with a duration of at least 3 months and had baseline and follow-up PSGs. Demographic data, comorbid diagnoses, and PSG data were collected and analyzed. RESULTS: Forty-five children met inclusion criteria. In the medication group, 29 children were identified. The median age was 7.4 years (interquartile range [IQR] 4.9-9.3). In the observation group, 16 children were identified. The median age was 4.0 years (IQR 3.2-5.3). The median time from baseline to follow-up PSG was 14.0 months (IQR 10.0-22.9) for the medication group and 10.5 months (IQR 6.5-33.5) for the observation group. There were no significant changes in the median obstructive apnea-hypopnea index from the baseline to follow-up PSG in either the medication group (2.8 [IQR 2.2-3.6) versus 3.5 [IQR 1.4-4.8) events/h; P = .25) or the observation group (2.3 [IQR 1.3-3.1] versus 2.9 [IQR 1.9-6.8] events/h; P = .12). Similarly, there were no significant differences in apnea-hypopnea index, oxygen nadir or end-tidal carbon dioxide between the groups (P = .07-1). CONCLUSIONS: In our cohort, medication therapy did not significantly improve polysomnographic measures in children with DS and mild OSA. Several factors such as hypotonia and relative macroglossia may explain the ineffectiveness of medical therapy for OSA in this population. Further prospective studies are necessary to confirm these results and to evaluate if a subgroup of DS children may benefit from medical therapy.

Sleep manifestations, sleep architecture in children with Eosinophilic esophagitis presenting to a sleep clinic.

STUDY OBJECTIVES: To describe sleep manifestations, polysomnographic (PSG) findings, and specific sleep disorders in children with Eosinophilic Esophagitis (EoE). METHODS: This retrospective study included children with EoE who were referred to sleep clinics. Clinical manifestations, PSG variables, and diagnosis of sleep disorders were analyzed. Sleep architecture of patients with EoE was compared to control subjects. RESULTS: In sum, 81 children with EoE met the criteria for entry into the analysis with a mean age of 10.1 ± 4.4 years. Of those, 46 children (57%) presented in the sleep clinic with active EoE symptoms, while 35 (43%) children did not have active EoE symptoms at presentation. Several sleep complaints were common in children with EoE, including snoring (62, 76.5%), restless sleep (54, 66.6%), legs jerking or leg discomfort (35, 43.2%) and daytime sleepiness (47, 58.0%). Comparing sleep architecture with controls, children with EoE had significantly higher NREM2 (P= < 0.001), lower NREM3 (P= < 0.001), lower rapid eye movement (REM) (P = 0.017), increased periodic leg movements (PLM) index (P= < 0.001) and increased arousal index (P = 0.007). There were no significant differences in the sleep efficiency between the EoE and control subjects. Common sleep diagnoses included obstructive sleep apnea (OSA, 30, 37.0%) and periodic limb movements disorder (PLMD, 20, 24.6%). Of note, we found a much higher percentage of PLMD in active EoE compared to inactive EoE (P = 0.004). CONCLUSIONS: Children with EoE have frequent sleep complaints and several sleep disorders identified from the sleep study, including sleep-disordered breathing and PLMD. Analysis of sleep architecture demonstrates significant sleep fragmentation as evidenced by decreased slow-wave sleep and REM sleep and increased arousal index.