Clinically Asymptomatic Sleep-Disordered Breathing in Infants with Single-Ventricle Physiology.

OBJECTIVES: To assess clinically asymptomatic infants with single-ventricle physiology (SVP) for sleep-disordered breathing (SDB) in the supine and car seat positions using polysomnography. Polysomnography results also were compared with results of a standard Car Seat Challenge to measure the dependability of the standard Car Seat Challenge. STUDY DESIGN: This was an observational study of 15 infants with SVP. Polysomnography data included Obstructive Index, Central Index, Arousal Index, Apnea Hypopnea Index, and sleep efficiency. Polysomnography heart rate and oxygen saturation data were used to compare polysomnography with the standard Car Seat Challenge. RESULTS: Polysomnography demonstrated that all 15 infants had SDB and 14 had obstructive sleep apnea (Obstructive Index ≥1/hour) in both the supine and car seat positions. Infants with SVP had a statistically significant greater median Obstructive Index in the car seat compared with supine position (6.3 vs 4.2; P = .03), and median spontaneous Arousal Index was greater in the supine position compared with the car seat (20.4 vs 15.2; P = .01). Comparison of polysomnography to standard Car Seat Challenge results demonstrated 5 of 15 (33%) of infants with SVP with abnormal Obstructive Index by polysomnography would have passed a standard Car Seat Challenge. CONCLUSIONS: Infants with SVP without clinical symptoms of SDB may be at high risk for SDB that appears worse in the car seat position. The standard Car Seat Challenge is not dependable in the identification of infants with SVP and SDB. Further studies are warranted to further delineate its potential impact of SDB on the clinical outcomes of infants with SVP.

Topical Review: A Biopsychosocial Framework for Pediatric Narcolepsy and Idiopathic Hypersomnia.

OBJECTIVE: Provide an overview of current research findings in pediatric central disorders of hypersomnolence (CDH) and propose a biopsychosocial model for clinical management, with a focus on interdisciplinary care and future directions for research and clinical practice. METHODS: Literature review drawing from pediatric and adult narcolepsy, as well as pediatric sleep and chronic pain research to develop an integrative biopsychosocial model for pediatric CDH. RESULTS: Youth with CDH are vulnerable to impairments in academics, emotional, and behavioral functioning, activity engagement and quality of life (QOL). There is a complex interrelationship between neurobiological features of disease, treatment-related factors, and psychological, sleep-related, and contextual factors across development. Research is limited largely to adults and pediatric narcolepsy type 1 and the mechanisms and evolution of morbidity remain poorly understood. CONCLUSIONS: In addition to first-line treatment (pharmacotherapy), routine screening of bio-behavioral and psychosocial functioning and QOL is needed to identify risk for compromised functioning warranting adjunctive interventions with behavioral health specialists.

Comparison of Pulmonary Function Decline in Steroid-Treated and Steroid-Naïve Patients with Duchenne Muscular Dystrophy.

OBJECTIVE: To describe and compare the lung function decline in patients with Duchenne muscular dystrophy on glucocorticoid therapy in contrast with glucocorticoid-naïve patients, and to define the deciles of pulmonary decline in glucocorticoid-treated patients. STUDY DESIGN: This retrospective study examined lung function of patients with Duchenne muscular dystrophy over 6 years of age followed between 2001 and 2015 at 2 centers-glucocorticoid-treated patients in Cincinnati, Ohio, and glucocorticoid-naïve patients in Paris, France. Forced vital capacity (FVC, FVC%), forced expiratory volume in 1 second, maximal inspiratory pressure, maximal expiratory pressure, and peak expiratory flow data were analyzed. Only FVC data were available for the French cohort. RESULTS: There were 170 glucocorticoid-treated patients (92%), 5 patients (2.7%) with past glucocorticoid use, and 50 French glucocorticoid-naïve patients. The peak absolute FVC was higher and was achieved at earlier ages in glucocorticoid-treated compared with glucocorticoid-naïve patients (peak FVC, 2.4 ± 0.6 L vs 1.9 ± 0.7 L; P < .0001; ages 13.5 ± 3.0 years vs 14.3 ± 2.8 years; P = .03). The peak FVC% was also higher and was achieved at earlier ages in glucocorticoid-treated patients (peak FVC%, 105.1 ± 25.1% vs 56 ± 20.9%; P < .0001; ages 11.9 ± 2.9 years vs 13.6 ± 3.2 years; P = .002). Rates of decline for both groups varied with age. Maximal rates of decline were 5.0 ± 0.26% per year (12-20 years) for glucocorticoid-treated and 5.1 ± 0.39% per year for glucocorticoid-naïve patients (11-20 years; P = .2). Deciles of FVC% decline in glucocorticoid-treated patients show that patients experience accelerated decline at variable ages. CONCLUSIONS: These data describe nonlinear rates of decline of pulmonary function in patients with Duchenne muscular dystrophy, with improved function in glucocorticoid-treated patients. FVC% deciles may be a useful tool for clinical and research use.

Epidemiology and Pathophysiology of Childhood Narcolepsy.

It is now recognized that there are two types of narcolepsy. Narcolepsy type I or Narcolepsy with cataplexy is caused by the loss of hypocretin or orexin neurons. Narcolepsy type II or narcolepsy without cataplexy has normal hypocretin and the etiology is unknown. Hypocretin is a neuropeptide produced by neurons in the lateral hypothalamus. Both genetic and environmental factors play a crucial role in the pathogenesis of narcolepsy. Most patients with narcolepsy type I and half of patients with narcolepsy type II carry HLA-DQB1*0602. HLA-DQB1*0602 forms a heterodimer with HLA-DQA1*0102 and may act as an antigen presenter to the T cell receptors, resulting in narcolepsy susceptibility. In addition, narcolepsy has been shown to be linked to polymorphisms in other non-HLA genes that may affect immune regulatory function, leading to speculation that autoimmune processes may play a crucial role in the loss of hypocretin neurons. Infections have been proposed as a potential trigger for the autoimmune-mediated mechanism. Several recent studies have shown increased cases of narcolepsy, especially in children and adolescents in relation with H1N1 influenza. The increased cases in Europe seems to be related to a specific type of H1N1 influenza vaccination (Pandemrix), while the increased cases in China are related to influenza infection. The data from the Pediatric Working Group of the Sleep Research Network have shown similar increases of early onset narcolepsy in the United States.

Nocturnal Oximetry-based Evaluation of Habitually Snoring Children.

RATIONALE: The vast majority of children around the world undergoing adenotonsillectomy for obstructive sleep apnea-hypopnea syndrome (OSA) are not objectively diagnosed by nocturnal polysomnography because of access availability and cost issues. Automated analysis of nocturnal oximetry (nSpO2), which is readily and globally available, could potentially provide a reliable and convenient diagnostic approach for pediatric OSA. METHODS: Deidentified nSpO2 recordings from a total of 4,191 children originating from 13 pediatric sleep laboratories around the world were prospectively evaluated after developing and validating an automated neural network algorithm using an initial set of single-channel nSpO2 recordings from 589 patients referred for suspected OSA. MEASUREMENTS AND MAIN RESULTS: The automatically estimated apnea-hypopnea index (AHI) showed high agreement with AHI from conventional polysomnography (intraclass correlation coefficient, 0.785) when tested in 3,602 additional subjects. Further assessment on the widely used AHI cutoff points of 1, 5, and 10 events/h revealed an incremental diagnostic ability (75.2, 81.7, and 90.2% accuracy; 0.788, 0.854, and 0.913 area under the receiver operating characteristic curve, respectively). CONCLUSIONS: Neural network-based automated analyses of nSpO2 recordings provide accurate identification of OSA severity among habitually snoring children with a high pretest probability of OSA. Thus, nocturnal oximetry may enable a simple and effective diagnostic alternative to nocturnal polysomnography, leading to more timely interventions and potentially improved outcomes.

Sleep disordered breathing in young boys with Duchenne muscular dystrophy.

OBJECTIVES: To describe sleep-disordered breathing (SDB) in young boys with Duchenne muscular dystrophy (DMD) and its relationship with pulmonary function tests (PFTs). STUDY DESIGN: This retrospective study examined diagnostic polysomnogram and PFT data of boys younger than 18 years with DMD and treated with steroids. Spirometry, respiratory muscle strength, body mass index (BMI), sleep architecture variables, and indices of SDB were analyzed. We examined the effect of PFT measures on the risk of each type of respiratory event using logistic regression and have reported results as OR (95% CI). RESULTS: Subjects included 110 boys with DMD, mean age 11.5 (5.6-17.9) years. Mean (±SD) percent forced vital capacity predicted was 79.5% ± 29.1%. Mean BMI for all subjects was 21.9 ± 7.0 kg/m(2), and mean BMI z-score was 0.65 ± 1.93. Seventy (63.6%) subjects had obstructive sleep apnea; 37 (33.6%) subjects had central sleep apnea; 18 (17%) subjects had hypoventilation. Median (IQR) Apnea Hypopnea Index was 2.9 (1.6-6.9) and median Obstructive Index was 1.5 (0.5-3.8). Obstructive Index during rapid eye movement sleep positively correlated with BMI (r = 0.33, P = .002), BMI z-score (r = 0.22, P = .04), and age (r = 0.31, P = .004). Lower forced vital capacity was associated with increased risk of hypoventilation (OR 0.8, P = .001). CONCLUSION: SDB is common in young boys with DMD treated with steroids. It is manifest with rapid eye movement-obstructive sleep apnea, often severe, and strongly influenced by BMI.

Conformational positioning improves sleep in premature infants with feeding difficulties.

OBJECTIVE: To determine whether premature infants' sleep organization, total sleep time, and arousals may be modulated while on a conformational positioner that provides boundaries, customized positioning, and containment compared with standard positioning (standard crib mattress). STUDY DESIGN: A proof of concept trial using a within subject crossover design was conducted among 25 premature infants with feeding difficulties. Infants of 31.5 weeks gestational age served as their own control during overnight polysomnography at postconceptual age 38.4 weeks. Each baby received both interventions (order randomized), 1 for each one-half of the 10.5-hour study. RESULTS: Use of the conformational positioner resulted in higher sleep efficiency of 61% vs 54% for the standard mattress (P < .05). The interventions did not differ for percent active sleep, percent quiet sleep, percent indeterminate sleep, or spontaneous arousals. Sleep efficiency was higher on the conformational positioner than standard positioning for surgical subjects and for subjects with necrotizing enterocolitis or gastroschisis (n = 10). The surgical subjects (n = 9) had lower sleep efficiency, lower percentage of active sleep, and more spontaneous arousals compared with the nonsurgical group. CONCLUSIONS: The use of the conformational positioner improved sleep efficiency vs the standard mattress in premature infants with feeding difficulties. Infants requiring surgery or with gastrointestinal diagnoses may be more susceptible to environmental stress.

Behavioral observation differentiates the effects of an intervention to promote sleep in premature infants: a pilot study.

PURPOSE: Sleep and ongoing cycling of sleep states are required for neurosensory processing, learning, and brain plasticity. Many aspects of neonatal intensive care environments such as handling for routine and invasive procedures, bright lighting, and noise can create stress, disrupt behavior, and interfere with sleep in prematurely born infants. The study empirically investigated whether a 30-minute observation of infant sleep states and behavior could differentiate an intervention to promote sleep in premature infants with feeding difficulties relative to conventional care (standard positioning, standard crib mattress [SP]). We included an intervention to determine the ability of the method to discriminate treatments and generate a benchmark for future improvements. The intervention, a conformational positioner (CP), is contoured around the infant to provide customized containment and boundaries. To more fully verify the 30-minute observational sleep results, standard polysomnography was conducted simultaneously and sleep outcomes for the 2 modalities were compared. SUBJECTS: In a randomized crossover clinical trial, 25 infants, 31.5 ± 0.6 weeks' gestational age and 38.4 ± 0.6 weeks at the study, with gastrointestinal conditions or general feeding difficulties used each intervention during an overnight neonatal intensive care unit sleep study. METHODS: Infant sleep states and behaviors were observed during two 30-minute periods--that is, on the positioner and mattress--using the naturalistic observation of newborn behavior. Two certified developmental care nurses assessed sleep state, self-regulatory, and stress behaviors during 2-minute intervals and summed over 30 minutes. Sleep characteristics from standard polysomnography were measured at the time of behavior observations. RESULTS: Infants on CP spent significantly less time in alert, active awake, or crying states by observation compared with SP. Surgical subjects spent more time awake, active awake, or crying and displayed a higher number of behavior state changes than the nonsurgical infants. The percentage of time in observed deep sleep and quiet sleep was correlated with both percentage sleep efficiency (r = 0.78) and fewer state shifts per hour (r = -0.65) from electroencephalogram (EEG). Sleep efficiency by EEG was greater on CP versus SP. CONCLUSIONS: The CP enabled sleep compared with the standard mattress (SP) over 30-minute observation periods. Sleep status from behavioral observation was verified by standard EEG-based sleep techniques. Behavioral observation of sleep states may be a useful strategy for measuring the effectiveness of strategies to facilitate sleep in premature infants. Surgical subjects may benefit from additional interventions to promote sleep.

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.

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.

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.

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.

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.

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.

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.

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.

Adenotonsillectomy outcomes in treatment of obstructive sleep apnea in children: a multicenter retrospective study.

RATIONALE: The overall efficacy of adenotonsillectomy (AT) in treatment of obstructive sleep apnea syndrome (OSAS) in children is unknown. Although success rates are likely lower than previously estimated, factors that promote incomplete resolution of OSAS after AT remain undefined. OBJECTIVES: To quantify the effect of demographic and clinical confounders known to impact the success of AT in treating OSAS. METHODS: A multicenter collaborative retrospective review of all nocturnal polysomnograms performed both preoperatively and postoperatively on otherwise healthy children undergoing AT for the diagnosis of OSAS was conducted at six pediatric sleep centers in the United States and two in Europe. Multivariate generalized linear modeling was used to assess contributions of specific demographic factors on the post-AT obstructive apnea-hypopnea index (AHI). MEASUREMENTS AND MAIN RESULTS: Data from 578 children (mean age, 6.9 +/- 3.8 yr) were analyzed, of which approximately 50% of included children were obese. AT resulted in a significant AHI reduction from 18.2 +/- 21.4 to 4.1 +/- 6.4/hour total sleep time (P < 0.001). Of the 578 children, only 157 (27.2%) had complete resolution of OSAS (i.e., post-AT AHI <1/h total sleep time). Age and body mass index z-score emerged as the two principal factors contributing to post-AT AHI (P < 0.001), with modest contributions by the presence of asthma and magnitude of pre-AT AHI (P < 0.05) among nonobese children. CONCLUSIONS: AT leads to significant improvements in indices of sleep-disordered breathing in children. However, residual disease is present in a large proportion of children after AT, particularly among older (>7 yr) or obese children. In addition, the presence of severe OSAS in nonobese children or of chronic asthma warrants post-AT nocturnal polysomnography, in view of the higher risk for residual OSAS.

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.

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.