OSA type-III and neurocognitive function.

Obstructive sleep apnea (OSA) due to a hypertrophy of the adenoids and/or the tonsils in otherwise healthy children is associated with neurocognitive dysfunction and behavioural disorders with various degrees of hyperactivity, aggressiveness, sometimes evolving to a label of attention-deficit hyperactivity disorder. Children with anatomical and/or functional abnormalities of the upper airways represent a very specific population which is at high risk of OSA (also called complex OSA or OSA type III). Surprisingly, the neurocognitive consequences of OSA have been poorly studied in these children, despite the fact that OSA is more common and more severe than in their healthy counterparts. This may be explained by that fact that screening for OSA and sleep-disordered breathing is not systematically performed, the performance of sleep studies and neurocognitive tests may be challenging, and the respective role of the underlining disease, OSA, but also poor sleep quality, is complex. However, the few studies that have been performed in these children, and mainly children with Down syndrome, tend to show that OSA, but even more disruption of sleep architecture and poor sleep quality, aggravate the neurocognitive impairment and abnormal behaviour in these patients, underlining the need for a systematic and early in life assessment of sleep and neurocognitive function and behaviour in children with OSA type III.

Assessment of obstructive sleep apnoea in children: What are the challenges we face?

There is an increasing demand for the assessment of sleep-disordered breathing in children of all ages to prevent the deleterious neurocognitive and behaviour consequences of the under-diagnosis and under-treatment of obstructive sleep apnoea [OSA]. OSA can be considered in three broad categories based on predominating contributory features: OSA type 1 [enlarged tonsils and adenoids], type II [Obesity] and type III [craniofacial abnormalities, syndromal, storage diseases and neuromuscular conditions]. The reality is that sleep questionnaires or calculations of body mass index in isolation are poorly predictive of OSA in individuals. Globally, the access to testing in tertiary referral centres is comprehensively overwhelmed by the demand and financial cost. This has prompted the need for better awareness and focussed history taking, matched with simpler tools with acceptable accuracy used in the setting of likely OSA. Consequently, we present key indications for polysomnography and present scalable, existing alternatives for assessment of OSA in the hospital or home setting, using polygraphy, oximetry or contactless sleep monitoring.

Sleep in children and young adults with cystic fibrosis.

Large gains have been made in the management of respiratory diseases associated with cystic fibrosis (CF). Initial studies evaluating sleep issues in CF focused on respiratory problems of nocturnal hypoxia, alveolar hypoventilation and risk of airway obstruction from nasal polyps with treatment evaluations including long term oxygen therapy or noninvasive ventilation in case of nocturnal hypercapnia. More recent studies include patients whose lung function is better preserved, and have permitted more focus on sleep patterns and sleep quality. This literature identified that reduced sleep duration and poor sleep quality are common and may be explained by chronic pain and cough, frequent stools, gastro-oesophageal reflux, nasal obstruction or sinusitis, and drugs such as corticosteroids or beta-agonists. In the teenage years, poor sleep hygiene, sleep debt and poor sleep quality are associated with depression, poor academic performance, less physical activity, and a decrease in quality of life. Restless leg syndrome also seems to be common in adult patients with CF. These sleep problems seem more important in patients with a low lung function but may also be observed in patients with preserved lung function. The consequences of poor sleep may potentially exaggerate the multi-organ morbidity of CF, such as pain, inflammation, susceptibility to infection, and glucose intolerance, but these aspects are largely under-evaluated. Sleep should be evaluated on a routine basis in CF and prospective studies assessing the benefits of interventions aiming at improving sleep duration and sleep quality urgently needed.

Behavioral and emotional characteristics of Canadian children with obesity and moderate-severe sleep-disordered breathing treated with positive airway pressure: longitudinal changes and associations with adherence.

STUDY OBJECTIVES: Behavioral characteristics and outcomes of positive airway pressure (PAP) therapy in children with obesity and moderate-severe sleep-disordered breathing (SDB) have not been reported. Our aims were to 1) determine baseline behavioral/emotional symptoms of this population and characterize changes over time with PAP, and 2) examine associations between baseline behavioral/emotional symptoms and PAP adherence. METHODS: This multicenter prospective cohort study of children with obesity prescribed PAP for moderate-severe SDB assessed PAP adherence (≥ 4 h/night, >50% of nights, usage diaries, downloads) and compared behavioral/emotional characteristics with parent- and child-reported Conners Rating Scale (Conners) and the Child Behavior Checklist (CBCL) at baseline and 1 year after PAP prescription between adherent and nonadherent participants; scores at baseline were compared retrospectively between adherence groups. RESULTS: Twenty-four children were included (median 14.1 years [IQR:12.4,16.0]; 87.5% males). Baseline Conners and CBCL scores were elevated (parent- and child-reported Conners inattention and hyperactivity subscales and CBCL subscales [total, internalizing, externalizing]). Baseline parent-reported Conners scores were significantly more elevated in the nonadherent than adherent group (inattention: 73.3 ± 8.5 vs 60.5 ± 14.6, P = .01; hyperactivity: 70.9 ± 11.1 vs 59.1 ± 16.0, P = .05). This difference was present 1 year later for inattention (P = .01) but not for hyperactivity (P = .09). Parent-reported CBCL scores improved over 1 year in adherent but not nonadherent participants. CONCLUSIONS: We found that children with obesity and moderate-severe SDB have elevated symptoms of behavioral/emotional concerns on standardized testing. Parent-reported emotional characteristics improved in the adherent but not in the nonadherent group. Children with greater inattention/hyperactivity at baseline were less adherent to PAP, suggesting this may contribute to PAP nonadherence. CITATION: Constantin E, MacLean JE, Barrowman N, et al. Behavioral and emotional characteristics of Canadian children with obesity and moderate-severe sleep-disordered breathing treated with positive airway pressure: longitudinal changes and associations with adherence. J Clin Sleep Med. 2023;19(3):555-562.

The effects of e-cigarette taxes on e-cigarette prices and tobacco product sales: Evidence from retail panel data.

We estimate the effect of e-cigarette tax rates on e-cigarette prices, e-cigarette sales, and sales of other tobacco products using NielsenIQ Retail Scanner data from 2013 to 2019. We find that 90% of e-cigarette taxes are passed on to consumer retail prices. We then estimate reduced form and instrumental variables regressions to examine the effects of e-cigarette and cigarette taxes and prices on sales. We calculate an e-cigarette own-price elasticity of -2.2 and particularly large elasticity of demand for flavored e-cigarettes. Further, we document a cigarette own-price elasticity of -0.4 and positive cross-price elasticities of demand between e-cigarettes and cigarettes, suggesting economic substitution.

Long-Term Non-invasive Ventilation in Children With Down Syndrome: A Systematic Review.

Context: Children with Down syndrome are at risk for obstructive sleep apnea, which may not be resolved by adenotonsillecotmy, as well as other respiratory disorders that may impact breathing during sleep. Long-term non-invasive ventilation, including continuous and bilevel positive airway pressure delivery, is an alternate treatment strategy. Objective: To assess the use and outcomes of long-term non-invasive ventilation in children with Down syndrome including comparison to other children using long-term non-invasive ventilation. Data Sources: The search strategy for the scoping review used Medical Subject Headings (MeSH) and free-text terms for "child" and "non-invasive ventilation." MEDLINE (Ovid), Embase (Ovid), CINAHL (Ebsco), Cochrane Library (Wiley), and PubMed databases were searched (1990-2021). Study Selection: The scoping review results were searched to identify studies including data on at least three children with Down Syndrome using long-term non-invasive ventilation. Data Extraction: Study characteristics, subject characteristics, technology type, and outcome measurements were extracted. Results: A total of 28 articles included 543 children with Down syndrome using long-term non-invasive ventilation. Children with Down syndrome accounted for 18% of children using long-term non-invasive ventilation. Data on efficacy, feasibility, and adherence in children with Down syndrome are comparable to other children. Children with Down syndrome may have greater difficulty initiating long-term non-invasive ventilation, longer time to establish use, and a higher rate of inability to establish use. Outcome data is limited but suggest favorable impact on cardiac function and attention. Limitations: Articles related to long-term non-invasive ventilation use in adolescents and young adults may have been excluded. Conclusions: Children with Down syndrome make up a significant portion of the population of children using long-term non-invasive ventilation. While there is more limited data available with respect to the use and outcomes for children with Down syndrome compared to the other children, long-term non-invasive ventilation is an effective and well-tolerated therapy with no clear differences in the use or outcomes for children with Down syndrome. Additional work is needed to understand potential challenges around establishing long-term non-invasive ventilation use in children with Down syndrome. Systematic Review Registration: https://www.crd.york.ac.uk/prospero/display_record.php?RecordID=206533, identifier: CRD206533.

Comparison of airway pressures and expired gas washout for nasal high flow versus CPAP in child airway replicas.

BACKGROUND: For children and adults, the standard treatment for obstructive sleep apnea is the delivery of continuous positive airway pressure (CPAP). Though effective, CPAP masks can be uncomfortable to patients, contributing to adherence concerns. Recently, nasal high flow (NHF) therapy has been investigated as an alternative, especially in CPAP-intolerant children. The present study aimed to compare and contrast the positive airway pressures and expired gas washout generated by NHF versus CPAP in child nasal airway replicas. METHODS: NHF therapy was investigated at a flow rate of 20 L/min and compared to CPAP at 5 cmH2O and 10 cmH2O for 10 nasal airway replicas, built from computed tomography scans of children aged 4-8 years. NHF was delivered with three different high flow nasal cannula models provided by the same manufacturer, and CPAP was delivered with a sealed nasal mask. Tidal breathing through each replica was imposed using a lung simulator, and airway pressure at the trachea was recorded over time. For expired gas washout measurements, carbon dioxide was injected at the lung simulator, and end-tidal carbon dioxide (EtCO2) was measured at the trachea. Changes in EtCO2 compared to baseline values (no intervention) were assessed. RESULTS: NHF therapy generated an average positive end-expiratory pressure (PEEP) of 5.17 ± 2.09 cmH2O (mean ± SD, n = 10), similar to PEEP of 4.95 ± 0.03 cmH2O generated by nominally 5 cmH2O CPAP. Variation in tracheal pressure was higher between airway replicas for NHF compared to CPAP. EtCO2 decreased from baseline during administration of NHF, whereas it increased during CPAP. No statistical difference in tracheal pressure nor EtCO2 was found between the three high flow nasal cannulas. CONCLUSION: In child airway replicas, NHF at 20 L/min generated average PEEP similar to CPAP at 5 cm H2O. Variation in tracheal pressure was higher between airway replicas for NHF than for CPAP. The delivery of NHF yielded expired gas washout, whereas CPAP impeded expired gas washout due to the increased dead space of the sealed mask.

Predictors of longitudinal outcomes for children using long-term noninvasive ventilation.

BACKGROUND: Noninvasive ventilation (NIV) is a first-line therapy for sleep-related breathing disorders and chronic respiratory insufficiency. Evidence about predictors that may impact long-term NIV outcomes, however, is scarce. The aim of this study is to determine demographic, clinical, and technology-related predictors of long-term NIV outcomes. METHODS: A 10-year multicentred retrospective review of children started on long-term continuous or bilevel positive airway pressure (CPAP or BPAP) in Alberta. Demographic, technology-related, and longitudinal clinical data were collected. Long-term outcomes examined included ongoing NIV use, discontinuation due to improvement in underlying conditions, switch to invasive mechanical ventilation (IMV) or death, patient/family therapy declination, transfer of services, and hospital admissions. RESULTS: A total of 622 children were included. Both younger age and CPAP use predicted higher likelihood for NIV discontinuation due to improvement in underlying conditions (p < .05 and p < .01). Children with upper airway disorders or bronchopulmonary dysplasia were less likely to continue NIV (p < .05), while presence of central nervous system disorders had a higher likelihood of hospitalizations (p < .01). The presence of obesity/metabolic syndrome and early NIV-associated complications predicted higher risk for NIV declination (p < .05). Children with more comorbidities or use of additional therapies required more hospitalizations (p < .05 and p < .01) and the latter also predicted higher risk for being switched to IMV or death (p < .001). CONCLUSIONS: Demographic, clinical data, and NIV type impact long-term NIV outcomes and need to be considered during initial discussions about therapy expectations with families. Knowledge of factors that may impact long-term NIV outcomes might help to better monitor at-risk patients and minimize adverse outcomes.

Assessment and treatment of pediatric obstructive sleep apnea in Canada: history and current state of affairs.

AIM: To highlight Canada's contributions to the assessment and treatment of pediatric obstructive sleep apnea as well as outline the current state of pediatric obstructive sleep apnea in Canada. METHODS: A search was conducted in MEDLINE (Ovid) using Medical Subject Headings (MeSH) and free-text terms for 'child' and 'obstructive sleep apnea' with subsequent 'human' limit. The results were reviewed to identify publications where any author's listed a Canadian institution. RESULTS: Canadian contributions to the field of pediatric obstructive sleep apnea have grown over the last 30 years with an increase in number of contributors and centres. Much of the early work stemmed from McGill University with important contributions in examining alternatives to polysomnography and post-adenotonsillectomy respiratory compromise. Today, contributors from centres across the country are engaged in the field and come from a greater diversity of disciplines. With continued challenges and opportunities, Canada will continue to help advance the field of pediatric OSA. CONCLUSION: Canada has a strong community of people invested in continuing to work to improve the lives of Canadian children with pediatric OSA.

Insulin Resistance and Hypertension in Obese Youth With Sleep-Disordered Breathing Treated With Positive Airway Pressure: A Prospective Multicenter Study.

STUDY OBJECTIVES: There is evidence that cardiometabolic disease associated with obesity and sleep-disordered breathing (SDB) in adults is present in youth. SDB is often treated with positive airway pressure (PAP) in youth with obesity. Our aims were to determine: (1) the prevalence of cardiometabolic disease and (2) whether PAP improves markers of cardiometabolic disease, in youth with obesity and newly diagnosed moderate-severe SDB. METHODS: A prospective multicenter cohort study was conducted in youth (8 to 16 years old) with obesity, prescribed PAP therapy for newly diagnosed moderate-severe SDB. Assessments occurred at baseline and at 6 and 12 months. Outcomes included markers of insulin resistance (change in homeostasis model assessment of insulin resistance (HOMA-IR) at 6 months = primary outcome), hypertension (24-hour ambulatory/blood pressure) and inflammation (high-sensitivity C-reactive protein: hs-CRP). RESULTS: Twenty-seven participants were enrolled. Of those with baseline testing available, 10/25 (40%) had HOMA-IR above the 97th percentile, 10/23 (44%) were hypertensive, 16/23 (70%) had loss of nocturnal blood pressure dip and hs-CRP was elevated in 16/27 (64%). There were no significant changes over time in markers of metabolic dysfunction or blood pressure, nor between PAP-adherent and non-adherent subgroups. CONCLUSIONS: In youth with obesity and SDB, metabolic dysfunction and hypertension were highly prevalent. There were no statistically significant improvements in cardiometabolic markers 1 year after the prescription of PAP therapy, although clinically relevant improvements were seen in insulin resistance and systolic blood pressure load, important predictors of future risk of cardiovascular disease. Larger, longer-term studies are needed to determine whether PAP improves cardiometabolic outcomes in obese youth. COMMENTARY: A commentary on this article appears in this issue on page 1025.

Developing an index for the orthodontic treatment need in paediatric patients with obstructive sleep apnoea: a protocol for a novel communication tool between physicians and orthodontists.

INTRODUCTION: Sleep disordered breathing in the paediatric population can manifest as an array of different systemic symptoms; among them is a distinct malocclusion and craniofacial phenotype. Emerging research suggests that the treatment of this malocclusion and/or craniofacial phenotype through orthodontic intervention may help with the symptoms of these patients. Selecting the patients who would benefit from orthodontic treatment can be a difficult task for the physician with minimal dental training. Therefore the aim of this study is to develop a simple index to be used by medical professionals to identify those paediatric patients with orthodontic treatment needs who may benefit their obstructive sleep apnoea (OSA) symptoms. METHODS AND ANALYSIS: The methodology in this project has been devised through the WHO's recommendations on developing an index, with modifications based on the specific needs of this study. Based on the available literature, a draft index will be produced and subjected to multiple iterative revisions based on the feedback from: the Index Development Group, a group of multidisciplinary and internationally acclaimed experts in the field; the External Review Group, a group of potential end users and interested parties and the Steering Committee. Once the index has been formalised, it will be subjected to a pair of reliability tests using physicians and orthodontists scored 2 weeks apart. Subsequently, the index will be validated using dichotomous responses from orthodontists on whether they would treat a patient for OSA symptoms, and comparing the responses to the score of the index on the same patient. ETHICS AND DISSEMINATION: The index will be translated into French and will be presented in orthodontic and medical conferences, workshops, seminars, round table discussions, and free copies for download will be made available on the website of the University of Alberta Interdisciplinary Airway Research Clinic (iarc.ualberta.ca). Furthermore, the index will be published in a peer-reviewed medical journal to further increase the exposure of the index.

Craniofacial morphology in pediatric patients with persistent obstructive sleep apnea with or without positive airway pressure therapy: a cross-sectional cephalometric comparison with controls.

INTRODUCTION: Compression on the midface with nasal mask-delivered positive airway pressure (PAP) therapy in growing patients might contribute to midface retrusion. The objective of this study was to investigate the association between long-term PAP use and craniofacial morphologic pattern in children with persistent obstructive sleep apnea. METHODS: Images generated with cone-beam volumetric imaging were used to complete lateral cephalometric analyses of anteroposterior projection of the midface region. The study group included 12 subjects (10 boys, 2 girls; mean age, 9.0 years) who used PAP therapy for at least 6 months and at least 6 hours per night. Measurements from this group were compared with those of a control group of 11 subjects (5 boys, 6 girls; mean age, 9.6 years) with obstructive sleep apnea who did not have PAP. Measurements were taken at 1 time point. RESULTS: No significant differences were identified between the groups for any cephalometric variable. Multivariate linear regression analysis also did not identify a significant association between the number of months of PAP therapy and the cephalometric variables. Cephalometric data for both groups were pooled for comparison with appropriate published normative values for age and sex. Anterior cranial base length, overall anteroposterior length of the maxillary base, and mandibular body length were significantly shorter than normal in the subjects compared with published normative values. CONCLUSIONS: No association was demonstrated between midface projection and PAP use in growing patients. When compared with normative data for anterior cranial base, children with obstructive sleep apnea had shorter maxillary and mandibular lengths.

Full-night versus 4h evening polysomnography in children less than 2years of age.

OBJECTIVE: Pediatric polysomnogaphy (PSG) is associated with significant burden in terms of personnel time, resource use, and patient/family discomfort. We hypothesized that 4-h abbreviated PSG may be a suitable alternative to full-night PSG in children 24months of age and younger. METHODS: PSG results from the first 4-h were compared to the full-length studies from 105 children. Outcomes included total, obstructive, and central apnea indices. Sleep disordered breathing (SDB) was defined as an apnea-hypopnea index (AHI) >1.5events/h and obstructive sleep apnea (OSA) was defined as an obstructive AHI>1.5 events/h. Cutoffs for central apneas were 3events/h for subjects >6months of age and 10events/h for subjects ⩽6months of age. RESULTS: All but one subject had abnormal SDB by the full-night PSG and all individuals had at least one REM period in the first 4h of sleep. Mean oxygen saturations and end-tidal CO(2), did not significantly differ between full-night and 4-h PSG. 4-h PSG showed high sensitivity for total AHI (100% for ⩽6months and 92.9% for >6months respectively), obstructive AHI (97.9%; 91.1% respectively), and central apnea index (100%; 72.2% respectively). Agreement was lower for those with lower AHI. CONCLUSIONS: The high prevalence of SDB observed suggests that the goals of PSG in this age group at our center may be to determine the type and severity of SDB rather than presence or absence. The high sensitivity between full-night and 4-h PSG supports the use of 4-h PSG in children 24months and under, especially those ⩽6months of age.

The spectrum of sleep-disordered breathing symptoms and respiratory events in infants with cleft lip and/or palate.

OBJECTIVE: To determine the prevalence of sleep-disordered breathing (SDB) symptoms and respiratory events during sleep in infants with cleft lip and/or palate (CL/P). DESIGN: Prospective observational study. SETTING: Cleft palate clinic, tertiary care paediatric hospital, before palate surgery. PATIENTS: Consecutive newborn infants with CL/P. MAIN OUTCOME MEASURES: Demographics, clinical history, sleep symptoms, facial measurement and polysomnography (PSG; sleep study) data. RESULTS: Fifty infants completed PSG at 2.7±2.3 months; 56% were male, and 30% had a clinical diagnosis of Pierre Robin sequence (PRS) or a syndrome. The majority of infants (75%) were reported to snore frequently or constantly, while 74% were reported to have heavy or loud breathing during sleep. The frequency of parent-reported difficulty with breathing during sleep was 10% for infants with isolated CL/P, 33% for those with syndrome, and 43% for PRS (χ(2) 16.1, p<0.05). All infants had an Obstructive-Mixed Apnoea-Hypopnoea Index (OMAHI) >1 event/h, and 75% had an OMAHI >3 events/h. Infants with PRS had higher OMAHI (34.3±5.1) than infants with isolated CL/P (7.6±1.2) or infants with syndromes (15.6±5.7, F stat, p<0.001). Multivariate analysis showed that PRS was associated with higher OMAHI (B 0.53±0.22, p=0.022), but the majority of the variance for SDB was unexplained (constant B 1.31±0.55, p=0.024). CONCLUSIONS: The results highlight that infants across the spectrum of CL/P have a high risk of SDB symptoms and obstructive respiratory events before palate surgery. Clinicians should enquire about symptoms of SDB and consider investigation with polysomnography in all infants with CL/P.

Cystic fibrosis newborn screening does not delay the identification of cystic fibrosis in children with negative results.

BACKGROUND: Several studies have demonstrated the benefit of Cystic Fibrosis Newborn Screening (CFNBS) for early diagnosis and, hence, intervention but the impact of CFNBS on those children not detected on CFNBS is not known. CFNBS may provide false reassurance that all CF has been detected and, therefore, lead to a delay in the diagnosis of children with CF which is not detected on CFNBS. The aim of this study was to determine the impact of CFNBS on the presenting features of children with CF where CF was not detected on CFNBS. METHODS: Subjects at the CFNBS center were selected if CF was identified subsequent to a negative CFNBS with subjects at the No CFNBS selected based on the absence of ΔF508 mutations. Children presenting with features that would lead to investigation for CF independent of clinical status were excluded. Presenting features at diagnosis and pulmonary function at 6 years of age were extracted from medical records. RESULTS: Twelve children from the CFNBS site and 19 from the No CFNBS site were included in the analysis. The only significant difference between the two in features at diagnosis was lower mean weight z-scores at the No CFNBS site (-2.9 ± 1.8) compared to the CFNBS center (-1.4 ± 1.3, p<0.05). Age at diagnosis, presenting complaint and nutritional status did not differ by site. Growth parameters and pulmonary function at 6years of age showed no differences between sites. CONCLUSIONS: This study demonstrates that access to CFNBS does not result in delay in diagnosis or poorer outcomes in those children for whom CF was not detected on CFNBS. In addition, children with CF not detected on CFNBS present with typical features of CF and sweat chloride results that are diagnostic of CF.

Family matters: a twelve-year-old male with respiratory symptoms, cachexia and clubbing.

The presentation of HIV in children varies between developed and developing countries. In addition, the presentation in adolescents may differ from that in younger children. This report highlights a rare presentation and complication of HIV in children and emphasizes the importance of cultural competency when approaching children and families from different cultural backgrounds.

High-resolution computed tomography (HRCT) should not be considered as a routine assessment method in cystic fibrosis lung disease.

High-resolution computed tomography (HRCT) scanning of the chest should not be considered a routine clinical investigation in the management of CF. Although it demonstrates the detection of early lung damage in children with cystic fibrosis (CF), before HRCT can be considered for routine clinical use in CF it needs to be shown that the benefit from the information obtained will out-weigh potential risks. There is insufficient evidence for the benefit of HRCT for its inclusion into routine care. Moreover, in the absence of information resulting in change in management, HRCT has the potential to increase anxiety for both clinicians and families. In order to advocate for incorporating this technology into routine CF care, further support for its role in management decisions is needed.