Obstructive Apnea-hypopnea Index Research Articles

Sleep-Disordered Breathing and Associated Comorbidities among Preschool-Aged Children with Down Syndrome

Children with Down syndrome (DS) are at high risk of sleep-disordered breathing (SDB). The American Academy of Pediatrics recommends a polysomnogram (PSG) in children with DS prior to the age of 4. This retrospective study examined the frequency of SDB, gas exchange abnormalities, co-morbidities, and surgical management in children with DS aged 2–4 years old at Seattle Children’s Hospital from 2015–2021. A total of 153 children underwent PSG, with 75 meeting the inclusion criteria. The mean age was 3.03 years (SD 0.805), 56% were male, and 54.7% were Caucasian. Comorbidities included (n, %): cardiac (43, 57.3%), dysphagia or aspiration (24, 32.0%), prematurity (17, 22.7%), pulmonary (16, 21.3%), immune dysfunction (2, 2.7%), and hypothyroidism (23, 30.7%). PSG parameter data collected included (mean, SD): obstructive AHI (7.9, 9.4) and central AHI (2.4, 2.4). In total, 94.7% met the criteria for pediatric OSA, 9.5% met the criteria for central apnea, and 9.5% met the criteria for hypoventilation. Only one child met the criteria for hypoxemia. Overall, 60% had surgical intervention, with 88.9% of these being adenotonsillectomy. There was no statistically significant difference in the frequency of OSA at different ages. Children aged 2–4 years with DS have a high frequency of OSA. The most commonly encountered co-morbidities were cardiac and swallowing dysfunction. Among those with OSA, more than half underwent surgical intervention, with improvements in their obstructive apnea hypopnea index, total apnea hypopnea index, oxygen saturation nadir, oxygen desaturation index, total arousal index, and total sleep duration. This highlights the importance of early diagnosis and appropriate treatment. Our study also suggests that adenotonsillar hypertrophy is still a large contributor to upper airway obstruction in this age group.

A Novel Macroglossia Severity Index for Beckwith-Wiedemann Syndrome

Objective/ Summary Background Data: We propose the first classification scheme for macroglossia in patients with Beckwith-Wiedemann Syndrome (BWS), the BWS Index of macroGlossia (BIG). Methods: Patients with molecularly confirmed BWS seen from 2004-2023 were included to develop this system. Relationships among BIG scores, tongue reduction surgery, BWS clinical score, percent mosaicism, and polysomnography findings were examined. Results: Patients were classified from BIG0 to BIG3. BIG0 includes those without macroglossia; BIG1 includes those with macroglossia not protruding beyond the teeth/alveolus; BIG2 includes those with tongue protrusion past the teeth/alveolus to the lips but that can be contained within the mouth; and BIG3 includes those with tongues that protrude beyond the teeth/alveolus and lips but that cannot be closed within the mouth. Of the 459 patients with molecularly confirmed BWS, 266 (58.0%) patients were scored. One hundred and eleven (41.7%) were BIG0, 44 (16.5%) were BIG1, 90 (33.8%) were BIG2, and 21 (7.9%) were BIG3. As scores increased, patients had an increased incidence of tongue reduction surgery (BIG0: 0% versus BIG1: 20.5% versus BIG2: 51.1% versus BIG3: 100%; r=0.66, P<0.01). Higher BIG scores were associated with elevated BWS clinical scores (r=0.68, P<0.01) and increased tissue mosaicism (r=0.50, P<0.01) as well as trends towards worse obstructive apnea-hypopnea indices (r=0.29, P=0.02) and lower SpO2 nadirs (r=−0.29, P=0.02). Conclusion: In this large series of patients with Beckwith-Wiedemann Syndrome, increased BIG score correlates with undergoing tongue reduction surgery and increased phenotypic severity. Adoption of the BIG scoring system may facilitate communication and risk stratification across institutions.

Persistent and symptomatic periodic breathing beyond the neonatal period in full-term infants: A case series

IntroductionPeriodic breathing (PB) is considered physiological in the neonatal period and usually disappears in the first months of life. There are few data available on persistent PB after the neonatal period. The objective of this study was to characterize infants born at term with persistent PB after the age of 1 month through polysomnography (PSG) performed during symptoms. MethodsThis retrospective case series included infants born at term between 2012 and 2021, without an underlying disease, who presented with symptoms of persistent PB during a PSG. Persistent PB was defined as more than 1 % of total sleep time (TST) of PB after 1 month of life, and PB was defined as a succession of at least three episodes of central apnea lasting more than 3 s and separated by less than 20 s of normal breathing. ResultsA total of 10 infants born at term were included. They underwent PSG for brief resolved unexplained events, desaturation, pauses in breathing, cyanosis, and/or signs of respiratory distress. The percentage of TST spent with PB was 18.1 % before 3 months of age (n = 7), and 4.7 % between 3 and 6 months of age (n = 10). During the first PSG, ≥3 % of desaturation events were observed in 77–100 % of the PB episodes. At the first PSG, nine of the 10 infants had an obstructive apnea–hypopnea index of >10/h and five of 10 infants had a central apnea index of >5/h. Gastroesophageal reflux (GER) was suspected in eight infants. All infants showed improvement in the initial symptoms during the first year of life. ConclusionThis study presents cases of persistent and symptomatic PB after 1 month of life in infants born at term. The interesting finding was the presence of obstructive sleep apnea syndrome and/or central apnea syndrome in the majority of children, along with GER.

Outcome of tonsillotomy in children with obstructive sleep-disordered breathing

Objective/BackgroundMost children treated for obstructive sleep disordered breathing (oSDB) are not systematically assessed by polysomnography (PSG) nor by structuredsymptom quantification before surgical treatment. The main objective of this study wasto investigate the effect of adeno-tonsillotomy (ATO) on symptom burden and PSGparameters. MethodsChildren aged 2–10 years with oSDB were selected for ATO based uponclinical findings according to current standards of care in Denmark. Each childunderwent standardized assessment before and 3 months after surgery, including aPSG, tonsil size assessment, and the Pediatric Sleep Questionnaire –Sleep RelatedBreathing Disorder (PSQ) to quantify symptom burden. Obstructive sleep apnea (OSA)was defined as an obstructive apnea-hypopnea index (oAHI) ≥2/h. Successfultreatment was defined as post-surgery oAHI ≤5/h, and complete cure as oAHI ≤2/h. ResultsFifty-two children were included. Mean age was 5.0 years (SD ± 1.76). Thirteen children were identified with baseline oAHI <2/h. Significant improvement inOSA severity was observed in children with moderate-to-severe OSA, in whom oAHI decreased from 15.7/h to 2.6/h (p < 0.001). Treatment success was obtained in 85% and cure was obtained in 42% of children. PSQ-score significantly improved from 0.52 (CI 0.47–0.56) to 0.26 (CI 0.21–0.32) (p < 0.001). Baseline OSA severity was notcorrelated to baseline symptom burden nor to symptom relief after ATO. There were noserious adverse events. ConclusionsAdeno-tonsillotomy significantly reduced symptom burden in otherwise healthy children with oSDB symptoms. Significant improvement in oAHI was observedonly in children with moderate-to-severe OSA. We recommend combining clinicalevaluation with PSQ and oAHI.

Superiorly Based Posterior Pharyngeal Flaps: Using A Care Pathway to Optimize Speech and Airway Outcomes.

Pharyngeal flap (PF) surgery is effective at improving velopharyngeal sufficiency, but historical literature shows a concerning prevalence rate of obstructive sleep apnea (OSA), reported as high as 20%. Our institution has developed a protocol to minimize risk of postoperative obstructive complications and increase safety of PF surgery. We hypothesize that (1) preoperative staged removal of significant adenotonsillar tissue along with (2) multiview videofluoroscopy to guide patient-specific surgical approach via appropriately sized PFs can result in excellent speech outcomes while limiting occurrence of OSA. This was a retrospective chart review of all patients with velopharyngeal insufficiency (VPI) (aged 2-20 years) seen at the University of Rochester from 2015 to 2022 undergoing PF surgery to correct VPI. Nasopharyngoscopy was used for surgical planning and airway evaluation. Patients with tonsillar and adenoid hypertrophy underwent staged adenotonsillectomy at least 2 months before PF. Multiview videofluoroscopy was used to identify anatomic causes of VPI and to determine PF width. Patients underwent polysomnography and speech evaluation before and at least 6 months after PF surgery. Forty-one children aged 8.5 ± 4.1 years (range, 4 to 18 years) who underwent posterior PF surgery for VPI were identified. This included 10 patients with 22q11.2 deletion and 4 patients with Pierre Robin sequence. Thirty-nine patients had both pre- and postoperative speech data and underwent both a pre- and postoperative sleep study. Polysomnography showed no significant difference in obstructive apnea-hypopnea index after posterior PF surgery (obstructive apnea-hypopnea index preop, 1.3 ± 1.2 events per hour; postop, 1.7 ± 2.1 events per hour; P = 0.111). Significant improvements in speech outcome were seen in patients who underwent PF (modified Pittsburgh score preop, 11.52 ± 1.37; postop, 1.09 ± 2.35; P < 0.05). Use of preoperative staged adenotonsillectomy as well as patient-specific PF dimensions results in effective resolution of VPI and a low risk of OSA.

Impact of Adaptive Servo-Ventilation in Heart Failure Patients

Objective: Sleep-related breathing disorders (SRBD) as obstructive apnea, central apnea, and Cheyne-Stokes respiration (CSR), can be seen in patients with chronic heart failure. SRBD can influence the prognosis of heart failure. We aim to reveal sleep-related breathing disorders in heart failure patients and display the effects of adaptive servo-ventilation (ASV) as a new therapeutic modality. Materials and Methods: In this prospective study, 32 patients with heart failure were included. One night polysomnography (PSG) was done. Results: According to the results of PSG, the SRBD ratio was 96.7%. Continuous positive airway pressure (CPAP) and ASV titrations were offered to all patients with an apnea-hypopnea index (AHI) &amp;gt; 5. Demographics, clinical properties, symptoms, PSG findings, Cheyne-Stokes respiration (CSR), and echocardiography results were recorded. Before and after ASV titration, pulmonary function tests and walking tests were performed, and concentrations of transferrin and pro-BNP were recorded. In the groups according to the AHI, severe obstructive sleep apnea syndrome (OSAS) in 18 of 30 patients, four moderate OSAS, five mild OSAS, and two central sleep apnea (CSA). PSG and, CPAP, ASV titrations were done in 7 male and one female patient that obstructive apnea, central apnea, AHI, arousal, and SpO2 min values had significant improvements (p=0,001, p=0,016, p=0,001, p=0,015 and p=0,008 respectively). We determined all CSRs were eliminated with ASV. After ASV titration pro-BNP, walking distance, and FVC values changed significantly (p=0,036, p=0,018, and p=0,018 respectively). Conclusion: As a result, we determined CSR and central apneas persisted with CPAP but were eliminated with a one-night ASV application. ASV also decreased pro-BNP and increased FVC and walking distance values significantly.

Trends in Blood Mosaicism and Clinical Phenotype Score in Patients with Beckwith-Wiedemann Syndrome Evaluated for Tongue Reduction Surgery.

Most patients with Beckwith-Wiedemann syndrome (BWS) have macroglossia with some requiring tongue reduction surgery (TRS). This study reports correlations between levels of affected cells (mosaicism) and BWS clinical score in patients evaluated for TRS. We also show correlations of clinical score and mosaicism with obstructive sleep apnea (OSA) severity. Blood mosaicism levels and BWS clinical score were recorded in patients with macroglossia referred to plastic surgery for evaluation. Associations among blood mosaicism, BWS clinical score, TRS, and OSA were assessed with appropriate statistics. Of the 225 patients included, BWS blood testing was available in 128 (56.9%). Mosaicism levels were higher in those who underwent TRS compared to those who did not (85.9 85.9 (56.5-95.9)% vs. 29.7 (2.8-73.1)%, p<0.001). BWS clinical score was also higher in those requiring TRS (9.0 (8.0-11.0) versus 7.0 (6.0-9.0), p<0.001). There was a positive correlation between clinical score and obstructive apnea-hypopnea index (r=0.320, p=0.011). Receiver operating characteristic curve analysis showed a clinical score ≥11 had 100% specificity and 36.4% sensitivity for detecting patients requiring TRS. Blood mosaicism ≥80% had 63.6% sensitivity and 83.6% specificity for predicting surgery. A combined criteria of BWS clinical score ≥11 or mosaicism ≥80% had 72.7% sensitivity and 83.6% specificity for predicting TRS. Blood mosaicism levels and higher BWS clinical scores appear associated with a greater frequency of having surgery in patients with macroglossia referred for surgical evaluation. Elevations in BWS clinical scoring are associated with increased OSA severity while increased blood mosaicism is not.

Changes in the nasopharyngeal and oropharyngeal microbiota in pediatric obstructive sleep apnea before and after surgery: a prospective study

ObjectiveTo explore the changes and potential mechanisms of microbiome in different parts of the upper airway in the development of pediatric OSA and observe the impact of surgical intervention on oral microbiome for pediatric OSA.MethodsBefore adeno-tonsillectomy, we collected throat swab samples from different parts of the oropharynx and nasopharynx of 30 OSA patients and 10 non-OSA patients and collected throat swab samples from the oropharynx of the above patients one month after the adeno-tonsillectomy. The 16 S rRNA V3–V4 region was sequenced to identify the microbial communities. The correlation analysis was conducted based on clinical characteristics.ResultsThere was a significant difference of alpha diversity in different parts of the upper airway of pediatric OSA, but this difference was not found in children with non-OSA. Beta diversity was significantly different between non-OSA and pediatric OSA. At the genus level, the composition of flora in different parts is different between non-OSA and pediatric OSA. The correlation analysis revealed that the relative abundance of Neisseria was significantly correlated with obstructive apnea hypopnea index. Furthermore, the functional prediction revealed that pathways related to cell proliferation and material metabolism were significantly different between non-OSA and pediatric OSA. Besides, the adeno-tonsillectomy has minimal impact on oral microbiota composition in short term.ConclusionThe changes in upper airway microbiome are highly associated with pediatric OSA. The relative abundance of some bacteria was significantly different between OSA and non-OSA. These bacteria have the potential to become new diagnostic and early warning biomarkers.

Severe obstructive sleep apnea in children with syndromic craniosynostosis: analysis of pulse transit time.

We examined the association between pulse transit time (PTT) and obstructive sleep apnea (OSA) in children with syndromic craniosynostosis (SCS), where OSA is a common problem and may cause cardiorespiratory disturbance. Retrospective study of children (age <18 years) with SCS and moderate-to-severe OSA (i.e., obstructive apnea-hypopnea index [oAHI] ≥ 5), or no OSA (oAHI < 1) who underwent overnight polysomnography (PSG). Children without SCS and normal PSG were included as controls. Reference intervals (RIs) for PTT were computed by non-parametric bootstrap analysis. Based on RIs of controls, the sensitivity and specificity of PTT to detect OSA were determined. In a linear mixed-model the explanatory variables assessed were sex, age, sleep stage, and time after obstructive events. In all 68 included children (19 SCS with OSA, 30 SCS without OSA, 19 controls), obstructive events occurred throughout all sleep stages, most prominently during rapid eye movement sleep (REM) and non-REM sleep stages N1 and N2, with evident PTT changes. Greatest reductions were observed 4 - 8 s after an event (p < 0.05). In SCS with OSA, PTT RIs were lower during all sleep stages compared to SCS without OSA. The highest sensitivity was observed during N1 (55.5%), and the highest specificity during REM (76.5%). Lowest PTT values were identified during N1. Obstructive events occur throughout all sleep stages with transient reductions in PTT. However, PTT as a variable for OSA detection is limited by its sensitivity and specificity.

The relationship between birth weight and obstructive sleep apnea in children

Objective:To study the relationship between children's birth weight and obstructive sleep apnea（OSA）. Methods:The sleep data and birth information of children who underwent polysomnography in the Department of Otorhinolaryngology-Head and Neck Surgery of Henan Children's Hospital from October 2020 to July 2022 were retrospectively analyzed. The data of OSA detection rate, OSA severity, sleep structure and respiratory parameters in different birth weight groups were analyzed. Results:A total of 2 778 children met the inclusion criteria, including 1 833 males and 945 females. According to birth weight, the selected children were divided into three groups: 122 small for gestational age（SGA） group, 2 313 appropriate for gestational age（AGA）, and 343 large for gestational age（LGA） group. There was no significant difference in age between different groups（P=0.061）. In each group, boys are significantly more numerous than girls（P=0.001）. The difference in current body mass index（BMI） between groups was statistically significant: the current BMI was higher in the LGA group（17.51±4.01, P<0.001）. The severity of OSA was different in different birth weight groups（P=0.037）. There was a strong positive correlation between the severity of OSA and birth weight（r=0.992）. Children in the SGA group had shorter rapid eye movement（REM） sleep period（19.00[15.18, 23.33], P=0.012）, higher obstructive apnea-hypopnea index（OAHI） values（1.75[0.60, 5.13], P=0.019）, and had lower central apnea hypopnea index（CAHI） values（0.10[0.00, 0.50], P=0.020）. There were no significant differences in sleep structure and respiratory parameters between the LGA group and the AGA group. Multiple regression analysis of the factors affecting the OAHI index showed that the OAHI index of boys was higher than that of girls（95%CI 1.311-2.096, P<0.001）, and age was negatively correlated with the OAHI index（r=-0.105, 95%CI 0.856-0.946, P<0.001）, current BMI and OAHI index were positively correlated（r=0.037, 95%CI 1.010-1.065, P=0.007）. LGA was positively correlated with OAHI index（r=0.346, 95%CI 1.039-1.921, P=0.027）, and the correlation between LGA and OAHI（r=0.346） was higher than that between SGA and OAHI（r=0.340）. Conclusion:There was no significant difference in the incidence of OSA in children with different birth weight groups, but the OSA severity of LGA group was higher. Gender, age, BMI index and large for gestational age were the influencing factors for the occurrence of OSA in children, which should be paid more attention to in clinical practice.

Comparison of Epworth Sleepiness Scale and OSA-18 Scores With Polysomnography in Children.

Our goal is to determine if there is a correlation between Modified Epworth Sleepiness Scale (M-ESS) scores, obstructive sleep apnea (OSA)-18 scores, and polysomnography (PSG) outcomes in children. Retrospective chart review. Pediatric otolaryngology clinic. Charts of consecutive children presenting from July 2021 to July 2023 were reviewed. Demographics, body mass index (BMI), BMI Z score, M-ESS score, OSA-18 score, PSG results, and sleep apnea severity were included. One-way analysis of variance and Pearson/Spearman correlation coefficients were calculated. Three hundred sixty-seven children were included, 162 (44.1%) girls and 205 (55.9%) boys. Mean patient age was 7.8 (95% confidence interval [CI]: 7.3-8.3) years. M-ESS score was 6.3 (n = 348, 95% CI: 5.8-6.8), mean OSA-18 score was 56.2 (n = 129, 95% CI: 53.0-59.4). Mean apnea-hypopnea index (AHI) was 10.1 (95% CI: 8.7-11.4) events/h, obstructive AHI 9.3 (95% CI: 8.0-12.7) events/h, respiratory distress index 14.6 (95% CI: 8.4-20.8) events/h, and oxygen saturation nadir 89.8% (95% CI: 89.1-90.4). Sixty-two children (17.2%) had mild, 192 (53.5%) moderate, and 105 (29.2%) severe sleep apnea. M-ESS score correlated weakly to AHI (r = .19, P = <.001), and OSA-18 score to oxygen saturation nadir (r = -.16, P = .002). After logistic regression adjusted for age and BMI, neither clinical scores were independently associated with AHI. M-ESS and OSA-18 scores have a weak correlation with OSA severity in children. More reliable, age-appropriate screening tools are needed in pediatric sleep apnea.

Improvement in quality of life with continuous positive airway pressure outweighs the treatment burden in children with obstructive sleep apnea.

Continuous positive airway pressure (CPAP) for treatment of obstructive sleep apnea (OSA) may pose a significant burden on families. We assessed the impact of CPAP for children on quality of life (QOL) and caregiver treatment burden. Prospective cohort study of children commencing outpatient CPAP in a specialist sleep centre 2020-2022. Questionnaires regarding sleep-related symptoms (PROMIS Pediatric Sleep Disturbance and Sleep-Related Impairment), QOL (OSA-18, QI-Disability), caregiver burden (Caregiver Strain Questionnaire) and overall health impact (Glasgow Children's Benefit Inventory) were completed by caregivers at CPAP commencement and 6 weeks later. Twenty-sixpatients completed follow-up (7 female; median age 11.4 year, baseline obstructive apnea hypopnea index 10.3/h; 77% overweight or obese, 73% comorbidity other than obesity). OSA-related QOL (OSA-18) significantly improved at follow-up (p < 0.01), as did child general QOL (p < 0.001), sleep disturbance (p < 0.01) and sleep-related impairment (p < 0.001). Caregivers mostly rated CPAP as beneficial to their child's health but 19% rated CPAP as harmful or having no effect. Caregiver strain reduced at follow-up (p < 0.001) and benefit outweighed inconvenience (p < 0.0001) in 81%. CPAP adherence was correlated with overall health impact (r = 0.67, p < 0.01) but not with caregiver rating of inconvenience. CPAP resulted in improvements in QOL and sleep-related symptoms, and reduced caregiver strain. Perceived benefits outweighed the burden of treatment for most but not all families. CPAP adherence was moderately correlated with family-reported measures of benefit but not related to perceived inconvenience. This study provides reassuring evidence regarding the benefits and impacts of CPAP for children, many of whom already have complex health care needs.

Secondhand Smoke Exposure Measured in Urinary Cotinine Levels and Severity of Pediatric Sleep Apnea

Exposure to secondhand smoke has been associated with numerous health problems in children, including obstructive sleep apnea. Secondhand smoke exposure may be a risk factor for increased pediatric sleep apnea severity. To assess the association of secondhand smoke exposure (SHSe), quantified by urinary cotinine levels, with severity of obstructive sleep apnea (OSA) in children. This was a prospective cohort trial including pediatric patients from 3 to 16 years of age with sleep-disordered breathing who underwent a polysomnogram at a tertiary-level children's hospital in the US in either March 2014 to October 2016 or March 2020 to March 2021. Urine specimens were analyzed for cotinine, an important metabolite of nicotine. Each child's caregiver completed a validated SHSe questionnaire. Data were analyzed from February to June 2023. OSA and secondhand smoke. SHSe and severity of pediatric OSA, quantified by urinary cotinine levels and obstructive apnea hypopnea index (AHI) scores. Secondary outcomes were association of urinary cotinine levels with nadir oxygen saturation, sleep-related quality of life measured by the OSA-18 questionnaire, and caregiver-reported smoking habits (collected through a questionnaire). The study included 116 patients with a median (IQR) age of 6 (5-9) years, among whom 51 (45%) had obesity. The median (IQR) AHI was 3.0 (1.2-8.0), with 28 children (30.0%) having severe disease (AHI >10). Thirty-four children (29.0%) were found to have a positive result for urine cotinine screening, with a mean (SD) level of 11.7 (9.4) ng/mL. The percentage of children with SHSe was less than anticipated. There was no association identified between urinary cotinine levels and either AHI (ρ = -0.04; 95% CI, -0.22 to 0.15) or nadir oxygen saturation (ρ = -0.07; 95% CI, -0.26 to 0.11). Furthermore, SHSe was not associated with the presence of severe OSA (odds ratio, 0.70; 95% CI, 0.26 to 1.90). Children whose caregivers reported indoor SHSe were more likely to have a detectable urinary cotinine level (odds ratio, 20.3; 95% CI, 6.67 to 61.8). This cohort study did not identify any clinically meaningful association between SHSe, quantified by urinary cotinine level, and pediatric OSA severity. Future research with a larger number of children with SHSe is needed to confirm these findings and determine whether SHSe affects OSA treatment outcomes in children.

A case-control study to investigate the prevalence of obstructive sleep apnea and the utility of the PSQ-SRBD questionnaire in children and young people with epilepsy.

Epilepsy and obstructive sleep apnea syndrome (OSAS) are each relatively common in children. OSAS may affect cognition, such that recognition of OSAS is important for children and young people with epilepsy (CYPWE). Published pilot data reported 55% CYPWE had symptoms suggestive of OSAS, compared with 7% typically developing controls. The primary aim of this study was to ascertain OSAS prevalence by polysomnography (PSG) in CYPWE, with secondary aims being to evaluate the utility of sleep questionnaires in CYPWE. Children and young people with epilepsy and age- and sex-matched typically developing controls were studied. A single night of level I attended PSG was undertaken, along with questionnaires [Pediatric Sleep Questionnaire (PSQ-SRBD), Pittsburgh Sleep Quality Index (PSQI) and the childhood and adolescent Epworth Sleepiness Scale (ESS-CHAD)]. OSAS was defined as obstructive apnea-hypopnea index (oAHI) of ≥ 1 event/h. Polysomnography was performed in 72 children including 48 CYPWE (60% male) and 24 controls (54% male). Mean age (11 years) was similar for CYPWE and controls, p= 0.42; with slightly higher BMI z scores (0.7 v 0.1, p=0.03) noted in CYPWE. Mean oAHI was 0.61 in CYPWE versus 0.42 (controls), p=0.62. Despite higher PSQ-SRBD scores in CYPWE (0.38 v 0.12, p<0.001), no difference in OSAS prevalence (10% vs. 4%, p=0.78) was found. Children and young people with epilepsy had higher ESS-CHAD (6 vs. 3.5, p=0.01) and PSQI (5 vs. 3.3, p=0.02) scores indicating greater levels of daytime sleepiness and poorer sleep quality. The study found no evidence for increased OSAS prevalence in CYPWE, whilst the utility of the PSQ-SRBD in predicting OSAS appears limited for CYPWE. Children and young people with epilepsy are, however demonstrably sleepier with poorer sleep quality. The cause for these findings remains unclear. Investigation of Sleep Quality and Prevalence of Sleep-disordered Breathing in Children and Young People With Epilepsy; https://www.clinicaltrials.gov/study/NCT03103841.

Minimally-invasive airway management and early cleft palate repair in infants born with Robin sequence

The objective of this study was to report outcomes of early cleft palate repair in infants born with Robin sequence (RS). A retrospective case series in a tertiary referral paediatric hospital was carried out, examining a consecutive series of 69 infants born with RS and cleft palate. A minimally invasive approach was taken to upper airway obstruction, with liberal nasopharyngeal airway (NPA) and non-invasive ventilation (NIV) use, guided by sleep studies. The palate was repaired between 6 and 9 months with a modified Malek technique. The most frequently used airway adjunct (59.4% of patients) was an NPA and the median duration of use was 5.6 months. All patients underwent a modified Malek cleft palate repair at a median of 7 months of age. Overnight oximetry demonstrated higher mean oxygen saturation (SpO2) across the group from initial neonatal admission to discharge (median 96.5% (interquartile range [IQR] 95–98%) vs 97.45% (IQR 96.5–98%) (P = 0.2, N = 34). Of those with a cardiorespiratory polysomnogram, the obstructive apnoea-hypopnea index (OAHI) was significantly lower postoperatively (5.9 vs 2.8, P = 0.028). This study supports the use of non-surgical airway strategies and early cleft palate repair in infants born with RS and cleft palate.

Does REM AHI Predict Persistent OSA After Pediatric Adenotonsillectomy?

The utility of REM AHI in managing pediatric obstructive sleep apnea (OSA) is not fully understood. This study aimed to evaluate the relationship of preoperative REM AHI to postoperative persistence of OSA in children who underwent adenotonsillectomy. This retrospective chart review identified children under the age of 18 years that received an adenotonsillectomy for OSA and a preoperative and postoperative polysomnogram. Children with craniofacial or neuromuscular disorders or a tracheostomy were excluded. The primary outcome was the postoperative persistence of OSA, defined as a postoperative obstructive apnea-hypopnea index (oAHI) ≥ 1.5 events/hour. REM-predominant OSA was defined as a ratio of REM/NREM AHI ≥ 2. REM AHI minus NREM AHI and REM AHI minus oAHI helped to identify patients with a larger distribution of REM AHI. A total of 353 patients were included. Postoperative persistent OSA was seen in 232 (65.7%) children. The preoperative REM AHI, REM AHI minus NREM AHI, and REM AHI minus oAHI of children with persistent OSA did not differ significantly from children with resolution of OSA. Rates of persistence were not different between those with REM-predominant OSA and REM-independent OSA (63.8% vs 70.7%, P = .218). This study suggests that preoperative REM AHI may be a poor predictor of OSA persistence after adenotonsillectomy. Further study is needed to help characterize how pre-operative REM AHI should impact clinicians' decision making, family counseling and recommendations.

Role of overnight oximetry in assessing the severity of obstructive sleep apnoea in typically developing children: a multicentre study

Background and objectiveCardiorespiratory polygraphy (CRP) is the predominant technology used to diagnose obstructive sleep apnoea (OSA) in tertiary centres in the UK. Nocturnal pulse oximetry (NPO) is, however, cheaper and...

Evaluating the effect of recombinant human growth hormone treatment on sleep-related breathing disorders in toddlers with Prader–Willi syndrome: a one-year retrospective cohort study

BackgroundRecombinant human growth hormone (rhGH) therapy is beneficial for children with Prader–Willi syndrome (PWS) in improving short stature and metabolism, but the effect of early rhGH treatment on respiratory and sleep parameters for PWS children under three years old remains elusive. Thus, this study aimed to investigate the impact of rhGH treatment on sleep-related breathing disorders (SRBDs) for toddlers with PWS.MethodsA total of 17 age-matched PWS patients receiving rhGH treatment (rhGH group) and 17 control individuals not receiving rhGH treatment (non-rhGH group) were recruited for this study between October 2018 and January 2023. Data related to polysomnography-polygraphy (PSG) and serum levels of insulin-like growth factor (IGF-1) and insulin-like growth factor binding protein 3 (IGFBP-3) were collected.ResultsThe mean age in the rhGH group was 20.76 ± 9.22 months, which was comparable to that of the non-rhGH group (25.23 ± 13.81 months). The demographic and anthropometric parameters were similar across the two groups after 52 weeks of treatment. Administration of rhGH to toddlers did not exert adverse effects on the obstructive apnea–hypopnea index (OAHI), central apnea index (CAI), oxygen desaturation index (ODI), mean percutaneous oxygen saturation (SpO2), lowest SpO2, duration when SpO2 is lower than 90%, or proportion of the patients with SpO2 lower than 90%. Furthermore, the increased IGF-1 z-score and IGFBP-3 level did not worsen SRBDs.ConclusionTreatment with rhGH for 52 weeks on young toddlers with PWS showed no deleterious effects on SRBDs. This shed more light on the importance of initiating rhGH therapy early in PWS patients.

Hypoventilation in patients with Prader-Willi syndrome across the pediatric age.

Few data on alveolar hypoventilation in Prader-Willi syndrome (PWS) are available and the respiratory follow-up of these patients is not standardized. The objectives of this study were to evaluate the prevalence of alveolar hypoventilation in children with PWS and identify potential risk factors. This retrospective study included children with PWS recorded by polysomnography (PSG) with transcutaneous carbon dioxide pressure (PtcCO2) or end-tidal CO2 (ETCO2) measurements, between 2007 and 2021, in a tertiary hospital center. The primary outcome was the presence of alveolar hypoventilation defined as partial pressure of carbon dioxide (pCO2) ≥ 50 mmHg during ≥2% of total sleep time (TST) or more than five consecutive minutes. Among the 57 included children (38 boys, median age 4.8 years, range 0.1-15.6, 60% treated with growth hormone [GH], 37% obese), 19 (33%) had moderate-to-severe obstructive sleep apnea syndrome (defined as obstructive apnea-hypopnea index ≥5/h) and 20 (35%) had hypoventilation. The median (range) pCO2 max was 49 mmHg (38-69). Among the children with hypoventilation, 25% were asymptomatic. Median age and GH treatment were significantly higher in children with hypoventilation compared to those without. There was no significant difference in terms of sex, BMI, obstructive or central apnea-hypopnea index between both groups. The frequency of alveolar hypoventilation in children and adolescents with PWS is of concern and may increase with age and GH treatment. A regular screening by oximetry-capnography appears to be indicated whatever the sex, BMI, and rate of obstructive or central apneas.

Changes in behavioral and cognitive abilities after rapid maxillary expansion in children affected by persistent snoring after long-term adenotonsillectomy: A noncontrolled study

The objective of this study was to verify changes in behavioral abilities and cognitive functions after rapid maxillary expansion (RME) in children with refractory sleep-disordered breathing (SDB) in the long term after adenotonsillectomy. A prospective clinical trial study using RME therapy was conducted. Participant inclusion criteria were children who had adenotonsillectomy with maxillary transverse deficiency and persistent SDB (obstructive apnea-hypopnea index ≥1). The study included 24 children aged 5-12 years, and of these 24 children, 13 had primary snoring and 11 had obstructive sleep apnea. The patients underwent laryngeal nasofibroscopy and a complete polysomnography. In addition, patients completed the Obstructive Pediatric Sleep Questionnaire and Obstructive Sleep Apnea 18-Item Quality-of-Life Questionnaire. Behavioral and neurocognitive tests were also completed before and after RME. The Obstructive Pediatric Sleep Questionnaire and Obstructive Sleep Apnea 18-Item Quality-of-Life scores showed a statistically significant decrease in both groups (P<0.001) after RME. The results showed that neurocognitive and behavioral parameters (Child Behavior Checklist scale) were similar in primary snoring and obstructive sleep apnea (OSA) groups before RME. In the OSA group, the mean scores of the "Somatic" and "Aggressiveness" domains decreased significantly (P<0.05). The cognitive functions did not register significant differences pre- and post-RME in any of the cognitive functions, except for visuospatial function in the OSA group. The noncontrolled design was a major limitation of our study. The need for treatment for SDB should consider the association of symptoms and behavioral disturbances with the child's obstructive apnea-hypopnea index. RME might prove to be an alternative treatment for children with SDB refractory to adenotonsillectomy, improving quality of life and behavioral aspects. However, a larger sample size with a control group is needed to substantiate these claims.