Obstructive Apnea-hypopnea Index Research Articles

Management of Obstructive Sleep Apnea in the Infant: A Systematic Review and Meta-analysis.

To evaluate the improvement in respiratory parameters of infants with obstructive sleep apnea (OSA) treated with medical or surgical intervention. A comprehensive review was completed using the PubMed, Web of Science, Embase, and Cochrane libraries including articles published from 1975 to 2024. Interventions studied included adenotonsillectomy, mandibular distraction osteogenesis (MDO), tongue/lip adhesion, partial glossectomy, floor-of-mouth release, supraglottoplasty, oral appliances, tracheostomy, and positioning. Continuous positive airway pressure (CPAP) served as a control. Outcomes studied included pre- and postintervention obstructive apnea-hypopnea index. Studies included randomized controlled trials and case-control trials. A total of 2161 records were identified, and 59 studies were included in the analysis. Data was extracted following Preferred Reporting Items for Systematic Reviews and Meta-analyses guidelines and pooled using a random-effects model. The primary study outcome established prior to data collection was a change in the apnea-hypopnea index. A meta-analysis of MDO performed in infants for OSA demonstrated an overall pooled mean difference (pre-apnea-hypopnea index minus post-apnea hypopnea index) of 30.1 (95% confidence interval: 22.9, 37.4; 10 studies, 373 patients). No other data was pooled for analysis due to study heterogeneity. All CPAP studies showed apnea resolution. Tailored therapies including supraglottoplasty and tongue-lip adhesion reduced but did not resolve apneic events. Current investigations of the evaluation and treatment of infant OSA are limited by heterogeneity in reporting and study. Mandibular distraction and CPAP are promising.

Postoperative Respiratory Complications After Adenotonsillectomy in Children With High-Risk Obstructive Sleep Apnea.

Identify patient characteristics and polysomnogram (PSG) parameters associated with postoperative respiratory complications after adenotonsillectomy (AT) among children with high-risk obstructive sleep apnea (OSA). Case series with chart review. Tertiary care children's hospital. Pediatric patients (<18 years) with high-risk OSA (any 1 of: apnea-hypopnea index [AHI] >30, O2 nadir <80% and peak CO2 >60 mm Hg) on overnight PSG from 2019 to 2021 were included. Primary outcomes were major respiratory intervention during the postoperative admission, prolonged hospitalization, and intensive care unit (ICU) stay. A total of 307 patients met inclusion criteria. Median age was 6.5 years and 63% were male. Twenty-five (8.1%) required major respiratory intervention and 29 (9.7%) required ICU admission after AT. Major interventions and ICU admissions were significantly associated with neuromuscular disease (P < .01), higher obstructive apnea-hypopnea index (oAHI), higher CO2 peak, and lower O2 nadir. Prolonged admission had similar findings except oAHI was not significantly associated. Younger children were significant more likely to require ICU admission or prolonged admission. Increased oAHI and worsening O2 and CO2 parameters on preoperative PSG were associated with postoperative respiratory complications in children with high-risk OSA. Children with neuromuscular disease and age 0 to 2 had higher risk of ICU stay and prolonged hospitalization. Clinicians should recognize the importance of parameters beyond oAHI when anticipating postoperative monitoring.

Weight Gain After Adenotonsillectomy in Children With Mild Obstructive Sleep-Disordered Breathing

It is unknown whether adenotonsillectomy causes undesirable weight gain in children with mild obstructive sleep-disordered breathing (oSDB). To compare changes in anthropometric measures in children with mild oSDB treated with adenotonsillectomy vs watchful waiting. This was an exploratory analysis of the Pediatric Adenotonsillectomy Trial for Snoring (PATS) randomized clinical trial of adenotonsillectomy vs watchful waiting for mild oSDB (snoring with obstructive apnea-hypopnea index of <3 events/hour) that took place at 7 pediatric tertiary care centers across the US and included 458 children aged 3.0 to 12.9 years with mild oSDB. Participants were recruited from June 29, 2016, to February 1, 2021. Anthropomorphic measures taken at baseline and 12 months after randomization were standardized for age and sex, including each participant's percentage of the 95th body mass index percentile (%BMIp95). Data analyses were performed from March 15, 2023, to April 1, 2024. Early adenotonsillectomy (eAT) vs watchful waiting with supportive care (WWSC). Twelve-month change in %BMIp95 from baseline and undesirable weight gain (defined as any weight gain in a child who already had overweight or obesity or an increase from baseline normal weight/underweight to overweight/obesity) at follow-up assessment. The study analysis included 375 children (mean [SD] age, 6.1 [2.3] years; 188 [50.2%] females), of whom 143 (38%) had overweight or obesity at baseline. At 12 months, children in the eAT group experienced a 1.25-point increase in %BMIp95 compared with a 0.59-point increase in the WWSC group (mean difference, 0.93; 95% CI, -0.39 to 2.25). Undesirable weight gain was also similar between the eAT (n = 120; 32%) and WWSC (n = 101; 27%) groups (mean difference, 4%; 95% CI, 5% to 14%). The findings of this exploratory analysis of the PATS trial indicate that adenotonsillectomy was not independently associated with an increased risk of undesirable weight gain in children with mild oSDB. However, one-third of the children gained undesirable weight during the study, which suggests that there is an opportunity to address healthy weight management during the evaluation and treatment of children with mild oSDB. ClinicalTrials.gov Identifier: NCT02562040.

Polysomnogram Findings and Psychosocial and Academic Concerns in Children With Cleft Palate With or Without Cleft Lip.

Children born with a cleft palate with or without a cleft lip (CP±L) are at risk for sleep-disordered breathing, particularly obstructive sleep apnea (OSA). While OSA and CP±L have both been associated with higher risk for psychosocial and academic concerns, their combined risk has been understudied. This paper aimed to describe polysomnography findings and psychosocial and academic concerns among children with isolated (iCP±L) and syndrome-associated (sCP±L) clefts who had undergone primary palatoplasty. Medical records were reviewed from 2004 to 2022 for demographics, medical history, polysomnography results, and family-reported psychosocial and academic variables. Of the 694 patients with CP±L who had a palatoplasty, 147 had a polysomnogram and 82 had at least one follow-up polysomnogram. Across time points, only 19% to 27% of participants had normal polysomnograms and the most frequent finding was mild OSA for 30% to 35% of patients. For children with iCP±L, more frequent oxygen desaturations were significantly associated with receiving special education services and family-reported academic concerns and OSA was associated with a history of receiving mental health services. Children with sCP±L had a significantly higher Obstructive Apnea-Hypopnea Index and a greater proportion of all-day special education classroom placements relative to youth with iCP±L. Study results suggest that cleft providers should monitor for OSA and sleep-disordered breathing symptoms, collaborate with pulmonologists for evaluation and treatment of sleep concerns, and address possible sleep-associated psychosocial and academic issues.

Nocturnal Cardiac Arrhythmias in Heart Failure With Obstructive and Central Sleep Apnea

Both obstructive and central sleep apnea (OSA and CSA) may contribute to nocturnal cardiac arrhythmias (NCA). Data on the prevalence of clinically important nocturnal atrial and ventricular arrythmias in patients with heart failure with reduced ejection fraction (HFrEF) and obstructive (OSA) or central sleep apnea (CSA) are scarce. In a cohort of patients with HFrEF, how does the prevalence of NCA compare among those with OSA, CSA and those with no to mild sleep apnea? Is the severity of OSA or CSA associated with atrial and ventricular NCA? This cross-sectional analysis is an ancillary study of the ADVENT-HF trial. We compared the prevalence of NCA (excessive supraventricular ectopic activity [ESVEA], defined as premature atrial complexes [PAC] ≥30/h or supraventricular tachycardia ≥20 beats); atrial fibrillation/flutter [AF]; and >10 premature ventricular complexes [PVC/h]) on ECGs from polysomnograms of HFrEF patients between those with OSA (apnea-hypopnea index [AHI≥15]), those with CSA (AHI≥15/h) and those with no to mild sleep apnea (AHI<15, control). Compared to controls (n=76), the prevalence of ESVEA was higher in patients with OSA (n=430) and CSA (n=150), 0%, 9% and 12%, respectively. The prevalences of AF in the control, OSA and CSA groups were 9%, 17% and 27%, and of >10 PVC/h 45%, 59% and 63%, respectively. In multivariable regression analyses PAC/h was associated with OSA severity (obstructive AHI) [22.4% increase per 10 events/h (95% confidence interval: 5.2; 42.3); p=0.009], but neither obstructive nor central AHI were associated with AF or >10 PVC/h. In patients with HFrEF, the prevalences of nocturnal ESVEA, AF and PVC >10/h were higher in those with, than in those without OSA or CSA, and OSA severity was related to the burden of nocturnal atrial ectopy. Severity of OSA or CSA were not significantly related to AF or >10 PVC/h.

Longitudinal Success of Tonsillectomy for Obstructive Sleep Apnea in Children with Down Syndrome.

Obstructive sleep apnea is common in children with Down syndrome (DS). Tonsillectomy is recommended as the first-line approach in treating children with obstructive sleep apnea (OSA), however, there is limited data on the long-term outcomes in children with DS who undergo tonsillectomy. In this retrospective study, we examined the long-term polysomnographic and symptomatic outcomes in children with DS who underwent tonsillectomy with or without an adenoidectomy (T&A). We hypothesize that the success of T&A to treat OSA in children with DS will diminish with time. A retrospective chart review of children with DS who underwent T&A between 2009 and 2015 was conducted. Inclusion criteria were children with at least 1 postoperative polysomnogram (PSG) within 6 months of T&A with an obstructive apnea/hypopnea index (OAHI) < 5. Outcomes were determined by subsequent clinic visits and postoperative polysomnograms: OAHI ≥ 5, snoring reported during clinic visit and time to reoccurrence. Childrens Hospital Colorado. Of the 57 children with mild OSA at 1st (initial) PSG, 13/40 (33%) children had OAHI ≥ 5 at the 2nd postoperative PSG. Of the 18 patients who underwent a 3rd PSG, 4 (22%) progressed to moderate/severe OSA. A total of 17 patients out of the original 57 (30%) progressed to moderate/severe OSA with the median time for the additional post-op PSG's being 2.3 years. Children with DS who have at most mild OSA (OAHI < 5) following a T&A are at risk for progressing to at least moderate OSA within 2 years after their T&A. A surveillance PSG 2 years following surgery will identify these children.

Nonsurgical improvement of severe upper airway obstruction in infants with Robin sequence and cleft palate using Stanford Orthodontic Airway Plate treatment.

Severe respiratory distress of neonates with Robin sequence (RS) is traditionally managed by surgery. Stanford Orthodontic Airway Plate treatment (SOAP) is a nonsurgical option. The study aimed to determine if SOAP can improve polysomnography (PSG) parameters of neonates with RS. PSG of neonates with RS treated with SOAP at a single hospital were retrospectively analyzed. Patients without PSG at all 4 time points (pre-, start of-, mid-, and post-treatment) were excluded. Data were analyzed using a linear mixed effects model. Sixteen patients were included. All patients had cleft palate (CP). The median age (min, max) at the start of treatment was 1.1 months (0.5, 2.3) with the treatment duration of 4.5 months (3.5, 6.0). The mean obstructive apnea-hypopnea index (95% confidence interval) decreased from 39.3 events/hour (32.9, 45.7) to 12.2 events/hour (6.7, 17.7) (P < 0.001), obstructive apnea index decreased from 14.1 (11.2, 17.0) events/hour to 1.0 (-1.5, 3.5) events/hour (P < 0.001), and oxygen nadir increased from 79.9% (77.4, 82.5) to 88.2% (85.5, 90.8) (P < 0.001) between pre- and start of treatment. Respiratory improvements were sustained during and after the treatment. All patients avoided mandibular distraction osteogenesis or tracheostomy following SOAP. As being a rare diagnosis, the number of participants was, as expected, low. However, the current study demonstrates that SOAP can improve PSG parameters, demonstrating its potential utility before surgical interventions for neonates with RS and CP experiencing severe respiratory distress.

Quantitative Proteomics Analysis of Serum and Urine With DIA Mass Spectrometry Reveals Biomarkers for Pediatric Obstructive Sleep Apnea

ObjectivesIdentification of suitable biomarkers that facilitate the screening and evaluation of pediatric obstructive sleep apnea (OSA) and its severity was explored. MethodsData-independent acquisition quantitative proteomic analysis was employed to identify serum and urine proteins with differential expression patterns between children with OSA and controls. Differentially expressed proteins that gradually increased or decreased with the severity of OSA were retained as potential biomarkers and underwent ELISA validation. ResultsWe found that with increasing severity of OSA, there was a gradual upregulation of 34 proteins in the serum and 124 proteins in the urine, along with a respective downregulation of 10 serum proteins and 64 urinary proteins in the initial cohort of 40 children. These proteins primarily participate in immune activation, the complement pathway, oxygen transport, and reactive oxygen metabolism. Notably, cathepsin Z exhibited a positive correlation with the obstructive apnea hypopnea index, whereas sex hormone-binding globulin (SHBG) was negatively correlated. These proteins were then validated by ELISA in an independent cohort (n=21). Circulating cathepsin Z and SHBG levels displayed acceptable diagnostic performance of OSA with AUC values of 0.863 and 0.738, respectively. ConclusionsWe identified two promising circulating proteins as novel biomarkers for clinical diagnosis and assessment of pediatric OSA severity. Furthermore, the comprehensive proteomic profile in pediatric OSA should aid in exploring the underlying pathophysiological mechanisms associated with this prevalent condition.

Ethnic/racial and sex disparities in obstructive sleep apnea among adolescents in southern California.

Pediatric obstructive sleep apnea (OSA) is common; however, inclusion of adolescents and especially those of ethnic/racial minorities in research is scarce. We hypothesized that ethnic/racial minority adolescents undergoing polysomnography have higher prevalence and more severe OSA compared to those who are non-Hispanic (NH) White. Retrospective review of 1,745 adolescents undergoing diagnostic polysomnography. Demographic characteristics, age, body mass index percentile, and polysomnography parameters were obtained. Descriptive statistics comparing race/ethnicity were analyzed. Linear regression of log-transformed obstructive apnea-hypopnea index, and logistic regression of moderate-severe OSA (obstructive apnea-hypopnea index ≥ 5 events/h) adjusting for covariates were analyzed. A total of 58.2% adolescents were Hispanic, 24.1% NH-White, 4.3% NH-Asian/Pacific Islander, 4.2% NH-Black/African American, and 6.6% NH-other. Compared to the NH-White group, the Hispanic group had higher obstructive apnea-hypopnea index and any level of OSA severity, the Black/African American group had higher any level of OSA, and the NH-Asian group had higher moderate-severe OSA. Multiple linear regression of log-obstructive apnea-hypopnea index identified a positive association with Hispanic ethnicity (β: 0.25, P value < .05). Compared to the NH-White group, the Hispanic and the Asian/Pacific Islander groups were 1.45 (95% confidence interval: 1.10, 1.93) and 1.81 (95% confidence interval: 1.05, 3.10) times more likely to have moderate-severe OSA, respectively, after adjusting for relevant covariates. Stratified analysis by sex identified an association only among males between Hispanic ethnicity (odds ratio: 1.85, 95% confidence interval: 1.27, 2.70) and Asian/Pacific Islander ethnicity (odds ratio: 2.62, 95% confidence interval: 1.35, 5.11) and moderate-severe OSA, compared to the NH-White group. Among adolescents undergoing polysomnography evaluation, we identified OSA racial/ethnic and sex disparities in Hispanic and NH-Asian adolescents. Community level studies with adequate representation of these minority groups are needed to identify factors associated with the reported increased susceptibility. Landeo-Gutierrez J, Ryu J, Tantisira K, Bhattacharjee R. Ethnic/racial and sex disparities in obstructive sleep apnea among adolescents in southern California. J Clin Sleep Med. 2024;20(10):1637-1645.

Overnight Monitoring Criteria for Children with Obstructive Sleep-Disordered Breathing After Tonsillectomy: Revisited.

Previous studies indicate children who pass an Asleep Room Air Challenge (AsRAC) do not have significant postoperative adverse respiratory events after adenotonsillectomy (T&A). Subsequently, we revised our overnight monitoring (OM) criteria, allowing patients with an obstructive apnea/hypopnea index (OAHI) ≤20 or nonsevere obesity (Class I) to be considered for same-day surgery (SDS) if they passed an AsRAC. Our hypothesis is that our modified OM criteria would not increase the return visits or readmission rates for patients undergoing SDS within 48 h or 15 days of T&A. A retrospective review of all children aged ≥3 and <21 years who underwent T&A at a tertiary children's hospital and its satellite locations was performed from January 2017 to September 2022. Descriptive statistics and outcome measures were compared using a 3% margin noninferiority test before and after the new criteria implementation. Before intervention, 3,266 (58%) T&As were performed as SDS. Afterward, 74% of T&As were performed as SDS (p-value <0.05). There was no difference in the ED revisit rate for SDS within the 3% noninferiority margin. Following intervention, 29% more children with Class I obesity (62% vs. 33%) underwent SDS (p-value <0.001). Afterward, 19% more children with polysomnography underwent SDS (39% vs. 20%), p-value <0.001. After intervention, within 48 h of SDS, six (0.9%) children had revisits for bleeding and seven (1.2%) for vomiting. There were no perioperative respiratory events. Our revised monitoring criteria did not demonstrate an increase in ED visit or readmissions rates within 48 h or 15 days of T&A. Additionally, we found a 29% increase in Class I obese children undergoing SDS T&A. 3 Laryngoscope, 2024.

Presurgical Reduction of the Cleft Palate: Serendipitous Benefit of the Stanford Orthodontic Airway Plate Treatment (SOAP) for Infants with Robin Sequence.

Narrowing of the palatal cleft is often observed in infants with Robin sequence (RS) treated with the Stanford Orthodontic Airway Plate treatment (SOAP) even though SOAP is utilized primarily to establish airway patency. The current study quantified dimensional changes of the cleft palate (CP) in infants with RS treated with SOAP. A retrospective chart review. Infants with RS and CP who completed SOAP and had maxillary arch models at both pre- and post-treatment time points at a single tertiary referral hospital between September 2019 and July 2023. Maxillary arch models were measured and analyzed using Bivariate statistical analysis. Seventeen infants were included in the study. The median age (min, max) was 6.7 weeks (1.1, 21.9) at pre-treatment and 26.6 weeks (18.7, 37.0) at post-treatment. The median Obstructive Apnea Hypopnea Index was 36.2 events/hour (8.1, 103.1) at pre-treatment and 4.1 events/hour (1.9, 8.6) at post-treatment. The pre-treatment width of CP decreased by an average (± standard diviation) of 6.37 mm (± 3.55, p < 0.001) at post-treatment. The ratio of the posterior cleft width to the total maxillary arch width decreased from 40% (± 9.1) at pre-treatment to 22% (± 11) at post-treatment (p < 0.001). The dimensions of CP reduced significantly during SOAP in infants with RS and CP treated for their severe upper airway obstruction. The findings highlight a potential benefit of SOAP that may contribute favorably to the palate repair surgery.

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

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. The purpose of this study was to compare HFNC and low-flow oxygen as treatments for OSA in infants. 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. 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. 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.

Early Adverse Respiratory Outcomes in Obese Pediatric Tonsillectomy Patients.

To determine if body mass index (BMI) is predictive of adverse respiratory events (ARE) in the obese pediatric population undergoing tonsillectomy. Case series with chart review. Single institution academic otolaryngology practice. All patients 3 to 12 years old with BMI ≥95th percentile that underwent tonsillectomy March 1, 2011 to July 15, 2020 were included. The study excluded patients with comorbidities that warranted admission independent of BMI, including Trisomy 21, gross developmental delay, neuromuscular disorders, and congenital heart disease. Perioperative AREs following tonsillectomy were recorded. AREs were defined as postoperative desaturation (SpO2 < 90%), intubation, continuous positive airway pressure (CPAP), or new O2 requirement for >2 hours. Eighteen patients (8%) had at least 1 ARE. There were no children age 5 and older with a BMI 95th percentile to 98.9th percentile who had an early adverse respiratory outcome. Preoperative polysomnogram (PSG) metrics, obstructive apnea-hypopnea index (oAHI), and oxygen saturations (SpO2) nadir was significantly different between patients with and without AREs (mean oAHI 54.3 vs 17.4, P = .02; mean SpO2 nadir 73.1% vs 84.5%, P = .05). There was no significant difference in the BMI z score (2.88 vs 2.45, P = .09) between groups. AREs requiring inpatient management are uncommon in obese children after tonsillectomy. BMI is a poor independent indication for admission except at BMI extremes. We found significantly higher oAHI and lower SpO2 nadir on PSG indicate a higher risk for AREs and can guide admission postoperatively. There may be a subset of obese tonsillectomy patients who can be safely discharged home on the day of surgery.

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.

The Efficacy of Noninvasive Ventilation in Patients Affected by Rett Syndrome With Hypoventilation

BackgroundRett syndrome is a progressive neurological disorder associated to several comorbidities that contribute significantly to impair lung function. Respiratory morbidity represents a major cause of death in this population. Little is known about the benefit of noninvasive ventilation. MethodsWe retrospectively enrolled patients with Rett syndrome who underwent a pneumological evaluation combined with a cardiorespiratory polygraphy and/or a pulse oximetry and capnography from 2012 to 2022. ResultsMedical records of 11 patients with Rett syndrome, mean age 13 ± 6 years, were evaluated. Most patients presented with both epilepsy and scoliosis. Five patients showed a pathologic sleep study and/or impaired night gas exchange: mean obstructive apnea-hypopnea index was 4 ± 3 events/hour; mean and minimal SpO2 were, respectively, 93% ± 2% and 83% ± 6%, while mean and maximal transcutaneous carbon dioxide monitoring (PtcCO2) were, respectively, 51 ± 5 mm Hg and 55 ± 8 mm Hg; and mean oxygen desaturation index was 13 ± 11 events/hour. These patients started noninvasive ventilation with clinical benefit and improved gas exchange mostly in terms of PtcCO2 (mean PtcCO2 51 ± 5 mm Hg before and 46 ± 6 mm Hg after noninvasive ventilation). ConclusionsNoninvasive ventilation is a suitable option for patients with Rett syndrome.

Analysis of Macular Vascularization Using Optical Coherence Tomography Angiography in Patients with Obstructive Sleep Apnea Syndrome: A Prospective Clinical Study.

Background and Objectives: Given the conflicting data available in the literature, this study aimed to investigate the impact of obstructive sleep apnea syndrome (OSAS) on the macular vascular density (VD) and perfusion density (PD). Materials and Methods: Based on the obstructive apnea-hypopnea index (OAHI), 61 prospectively recruited patients were assigned to either a control group (n = 12; OAHI < 5/h) or an OSAS group (n = 49; OAHI ≥ 5/h). The macular VD and PD of the superficial and deep capillary plexuses (SCP and DCP, respectively) were measured in the parafoveolar and perifoveolar areas using Zeiss PLEX Elite 9000 (6 × 6 mm). The values were compared between the control and OSAS groups. Results: Compared with the control group, the OSAS group demonstrated an increased VD of the DCP in the parafoveolar and perifoveolar areas and PD of the DCP in the perifoveolar area. No significant differences in either the macular VD or PD of the SCP were observed. There was no correlation between the OAHI and macular VD or PD. Conclusions: This study indicates that collateral vessel formation and possible retinal vasodilation occur in the DCP of patients with OSAS.

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.

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&lt;0.01). Higher BIG scores were associated with elevated BWS clinical scores (r=0.68, P&lt;0.01) and increased tissue mosaicism (r=0.50, P&lt;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.

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.