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1.
J Clin Sleep Med ; 16(6): 907-916, 2020 06 15.
Article in English | MEDLINE | ID: mdl-32043963

ABSTRACT

STUDY OBJECTIVES: Hypertension is a complication of obstructive sleep apnea (OSA) syndrome in adults. A correlation between OSA syndrome and elevated blood pressure (BP) is suggested in children, but its pathogenesis remains unclear. Our aim was to study the effects of sleep and sleep apnea on BP and sympathetic nervous system activation as measured by serum cortisol and urinary catecholamines. We hypothesized that children with OSA syndrome would have higher BP, urinary catecholamines, and cortisol compared with controls. METHODS: We measured BP during polysomnography in 78 children with suspected sleep-disordered breathing and 18 nonsnoring controls. BP was measured during wakefulness and every 30-60 minutes throughout the night. All participants had 24-hour urinary catecholamine and free cortisol collections 48 hours before polysomnography. RESULTS: BP varied with sleep stage; it was highest during wakefulness and N1 and lowest during non-rapid eye movement stage 3. Children classified as high apnea-hypopnea index (AHI) snorers (AHI >5 events/h) had a greater prevalence of systolic hypertension (57%) than low-AHI snorers (22%) and nonsnoring controls (22%; P = .04). The high-AHI snorers also had higher diastolic BP (P < .02) as well as blunted nocturnal diastolic BP changes during sleep (P = .02) compared with low-AHI snorers (AHI <5 events/h). Twenty-hour urinary free cortisol and 24-hour urinary catecholamines were not associated with BP. CONCLUSIONS: BP in children varies with sleep stage. OSA is associated with systolic hypertension, higher BP during rapid eye movement sleep, as well as elevation of diastolic BP and blunted BP changes with sleep.


Subject(s)
Hypertension , Sleep Apnea, Obstructive , Adult , Blood Pressure , Catecholamines , Child , Humans , Hydrocortisone , Hypertension/complications , Polysomnography
2.
J Clin Sleep Med ; 15(6): 915-921, 2019 06 15.
Article in English | MEDLINE | ID: mdl-31138379

ABSTRACT

STUDY OBJECTIVES: To determine whether adherence to positive airway pressure (PAP) differs in children with developmental disabilities (DD) compared to typically developing (TD) children. METHODS: PAP adherence of 240 children initiated on PAP for obstructive sleep apnea (OSA) was retrospectively analyzed. Adherence between groups, expressed as percentage of nights used and hours of usage on nights used at 3 and 6 months, was compared. Predictive factors of adherence were studied using a median regression model. RESULTS: A total of 103 children with DD (median [interquartile range] age = 7.9 [3.2-13.1] years) and 137 TD (11.0 [5.5-16.1], P = .005) children were included. Percentage of nights used was significantly higher in children with DD at 3 (DD = 86.7 [33.9-97.9], TD = 62.9 [30.8-87.8] P = .01) and 6 months (DD = 90.0 [53.3-100], TD = 70.7 [29.2-90.8], P = .003). Hours of usage on nights used at 3 and 6 months were similar between groups (DD = 5.0 [1.4-7.9], TD = 4.6 [1.9-7.2], P = .715; DD = 6.4 [1.8-8.3], TD = 5.7 [2.5-7.3], P = .345, respectively). This adherence measure improved over time in both groups (DD, P = .007; TD, P = .005). At 6 months, higher median neighborhood income and titration at or before 6 months were significantly predictive for percentage of nights used; higher PAP pressure was significantly predictive for hours of usage in both groups. CONCLUSIONS: Children with DD had better PAP adherence expressed as percentage of nights used than TD children. Hours of usage on nights used at 3 and 6 months were similar between groups and improved over time. Higher income and titration at or before 6 months were predictive of adherence in all children. These findings indicate that children with DD can successfully wear PAP.


Subject(s)
Continuous Positive Airway Pressure/psychology , Developmental Disabilities/complications , Patient Compliance/statistics & numerical data , Sleep Apnea, Obstructive/therapy , Age Factors , Child , Child Behavior/psychology , Child Development , Continuous Positive Airway Pressure/methods , Developmental Disabilities/psychology , Female , Follow-Up Studies , Humans , Male , Polysomnography , Retrospective Studies , Sleep Apnea, Obstructive/psychology , Treatment Outcome
3.
J Clin Sleep Med ; 15(3): 375-381, 2019 03 15.
Article in English | MEDLINE | ID: mdl-30853040

ABSTRACT

STUDY OBJECTIVES: Beckwith-Wiedemann syndrome (BWS) is a rare pediatric overgrowth disorder that includes a spectrum of clinical findings including macroglossia, especially in those with loss of methylation at the imprinting control region (IC2 LOM) on chromosome 11. Children with BWS can have very severe obstructive sleep apnea (OSA), but the prevalence of OSA in this population is poorly understood, as is the relationship between OSA and the BWS genotype/phenotype. We hypothesized that there would be a high prevalence of OSA in children with BWS, and that OSA would be more severe in children with IC2 LOM. METHODS: Medical records from children evaluated from March 2015 through January 2018 were reviewed for results from polysomnography, genetic testing, and clinical assessment. RESULTS: A total of 26 children with BWS not previously treated for OSA underwent polysomnography, genetic testing, and clinical assessment. Median (range) age was 5 months (3 days to 33 months). Most children, 20 (76.9%), had an obstructive apnea-hypopnea index (OAHI) > 2 events/h. Median (range) OAHI was 4.4 events/h (0 to 56.1 events/h). OAHI was significantly greater in participants younger than 6 months compared with those older than 6 months (P = .008). Those with IC2 LOM did not have a greater OAHI (P = .61) than those with other genetic causes of BWS, but OAHI had a strong positive correlation with BWS clinical score (Spearman rho = .54, P = .004). CONCLUSIONS: There is a high prevalence of OSA in children with BWS with macroglossia. Younger children with BWS and those with more phenotypic features may be at greatest risk of OSA.


Subject(s)
Beckwith-Wiedemann Syndrome/complications , Sleep Apnea, Obstructive/etiology , Beckwith-Wiedemann Syndrome/pathology , Child, Preschool , Female , Humans , Infant , Infant, Newborn , Male , Polysomnography , Prevalence , Retrospective Studies , Sleep Apnea, Obstructive/epidemiology
4.
Sleep ; 41(12)2018 12 01.
Article in English | MEDLINE | ID: mdl-30212861

ABSTRACT

Study Objectives: Depressive symptoms following adenotonsillectomy (AT) relative to controls were examined in children with obstructive sleep apnea syndrome (OSAS). Methods: The Childhood Adenotonsillectomy Trial (CHAT) multisite study examined the impact of AT in 453 children aged 5 to 9.9 years with polysomnographic evidence of OSAS without prolonged desaturation, randomized to early adenotonsillectomy (eAT) or watchful waiting with supportive care (WWSC). One hundred seventy-six children (eAT n = 83; WWSC n = 93) with complete evaluations for depressive symptomatology between baseline and after a 7-month intervention period were included in this secondary analysis. Results: Exact binomial test assessed proportion of depressive symptomatology relative to norms, while effects of AT and OSAS resolution were assessed through linear quantile mixed-models. Treatment group assignment did not significantly impact depression symptoms, although self-reported depression symptoms improved over time (p < 0.001). Resolution of OSAS symptoms demonstrated a small interaction effect in an unexpected direction, with more improvement in parent ratings of anxious/depressed symptoms for children without resolution (p = 0.030). Black children reported more severe depressive symptoms (p = 0.026) and parents of overweight/obese children reported more withdrawn/depressed symptoms (p = 0.004). Desaturation nadir during sleep was associated with self-report depressed (r = -0.17, p = 0.028), parent-reported anxious/depressed (r = -0.15, p = 0.049), and withdrawn/depressed (r = -0.24, p = 0.002) symptoms. Conclusions: Increased risk for depressed and withdrawn/depressed symptoms was detected among children with OSAS, and different demographic variables contributed to risk in self-reported and parent-reported depression symptoms. Arterial oxygen desaturation nadir during sleep was strongly associated with depressed symptoms. However, despite improvements in child-reported depressed symptoms over time, changes were unrelated to either treatment group or OSAS resolution status. Trials Registration: Childhood Adenotonsillectomy Study for Children with OSAS (CHAT), https://clinicaltrials.gov/show/NCT00560859, NCT00560859.


Subject(s)
Adenoidectomy/psychology , Depression/epidemiology , Depression/psychology , Sleep Apnea, Obstructive/psychology , Tonsillectomy/psychology , Black or African American , Child , Child, Preschool , Demography , Female , Humans , Incidence , Male , Obesity/physiopathology , Oxygen/blood , Parents , Self Report , Sleep/physiology , Sleep Apnea, Obstructive/physiopathology
5.
Sleep ; 41(6)2018 06 01.
Article in English | MEDLINE | ID: mdl-29546286

ABSTRACT

Study Objectives: To develop and evaluate the measurement properties of child-report and parent-proxy versions of the Patient-Reported Outcomes Measurement Information System (PROMIS) Pediatric Sleep Disturbance and Sleep-Related Impairment item banks. Methods: A national sample of 1104 children (8-17 years old) and 1477 parents of children 5-17 years old was recruited from an internet panel to evaluate the psychometric properties of 43 sleep health items. A convenience sample of children and parents recruited from a pediatric sleep clinic was obtained to provide evidence of the measures' validity; polysomnography data were collected from a subgroup of these children. Results: Factor analyses suggested two dimensions: sleep disturbance and daytime sleep-related impairment. The final item banks included 15 items for Sleep Disturbance and 13 for Sleep-Related Impairment. Items were calibrated using the graded response model from item-response theory. Of the 28 items, 16 are included in the parallel PROMIS adult sleep health measures. Reliability of the measures exceeded 0.90. Validity was supported by correlations with existing measures of pediatric sleep health and higher sleep disturbance and sleep-related impairment scores for children with sleep problems and those with chronic and neurodevelopmental disorders. The sleep health measures were not correlated with results from polysomnography. Conclusions: The PROMIS Pediatric Sleep Disturbance and Sleep-Related Impairment item banks provide subjective assessments of child's difficulty falling and staying asleep as well as daytime sleepiness and its impact on functioning. They may prove useful in the future for clinical research and practice. Future research should evaluate their responsiveness to clinical change in diverse patient populations.


Subject(s)
Patient Reported Outcome Measures , Polysomnography/standards , Sleep Wake Disorders/diagnosis , Surveys and Questionnaires/standards , Adolescent , Child , Child, Preschool , Female , Humans , Male , Parents/psychology , Polysomnography/methods , Psychometrics/methods , Quality of Life/psychology , Reproducibility of Results , Sleep Wake Disorders/physiopathology , Sleep Wake Disorders/psychology
9.
Sleep ; 40(7)2017 07 01.
Article in English | MEDLINE | ID: mdl-28541508

ABSTRACT

Study Objectives: Infants, children, and adolescents are increasingly being prescribed continuous positive airway pressure (CPAP) for treatment of obstructive sleep apnea syndrome (OSAS), yet adherence is often poor. The purpose of this study was to examine the relationship between caregiver and patient-reported health cognitions about CPAP prior to starting CPAP and CPAP adherence at 1 month. We hypothesized that greater caregiver-reported self-efficacy would be positively associated with CPAP adherence in children. We also evaluated patient-reported self-efficacy and caregiver- and patient-reported risk perception and outcome expectations as they related to adherence, as well as how demographic factors influenced these relationships. Methods: A pediatric modification of the Self-Efficacy Measure for Sleep Apnea Questionnaire was administered to children and adolescents with OSAS-prescribed CPAP and their caregivers during the clinical CPAP-initiation visit. The primary outcome variable for adherence was the average total minutes of CPAP usage across all days from the date that CPAP was initiated to 31 days later. Results: Unadjusted ordinary least-square regression showed a significant association between caregiver-reported self-efficacy and adherence (p = .007), indicating that mean daily CPAP usage increased by 48.4 minutes when caregiver-reported self-efficacy increased by one point (95% confidence interval 13.4-83.4 minutes). No other caregiver- or patient-reported cognitive health variables were related to CPAP use. Conclusions: This study indicates that caregiver CPAP-specific self-efficacy is an important factor to consider when starting youth on CPAP therapy for OSAS. Employing strategies to improve caregiver self-efficacy, beginning at CPAP initiation, may promote CPAP adherence.


Subject(s)
Continuous Positive Airway Pressure/psychology , Continuous Positive Airway Pressure/statistics & numerical data , Patient Compliance , Self Efficacy , Sleep Apnea, Obstructive/therapy , Adolescent , Caregivers , Child , Female , Humans , Male , Self Report , Surveys and Questionnaires
11.
Sleep ; 40(2)2017 Feb 01.
Article in English | MEDLINE | ID: mdl-28364508

ABSTRACT

Study Objectives: Continuous positive airway pressure (CPAP) is effective in treating obstructive sleep apnea in children, but adherence to therapy is low. Our center created an intensive program that aimed to improve adherence. Our objective was to estimate the program's efficacy, cost, revenue and break-even point in a generalizable manner relative to a standard approach. Methods: The intensive program included device consignment, behavioral psychology counseling, and follow-up telephone calls. Economic modeling considered the costs, revenue and break-even point. Costs were derived from national salary reports and the Pediatric Health Information System. The 2015 Medicare reimbursement schedule provided revenue estimates. Results: Prior to the intensive CPAP program, only 67.6% of 244 patients initially prescribed CPAP appeared for follow-up visits and only 38.1% had titration polysomnograms. In contrast, 81.4% of 275 patients in the intensive program appeared for follow-up visits (p < .001) and 83.6% had titration polysomnograms (p < .001). Medicare reimbursement levels would be insufficient to cover the estimated costs of the intensive program; break-even points would need to be 1.29-2.08 times higher to cover the costs. Conclusions: An intensive CPAP program leads to substantially higher follow-up and CPAP titration rates, but costs are higher. While affordable at our institution due to the local payer mix and revenue, Medicare reimbursement levels would not cover estimated costs. This study highlights the need for enhanced funding for pediatric CPAP programs, due to the special needs of this population and the long-term health risks of suboptimally treated obstructive sleep apnea.


Subject(s)
Continuous Positive Airway Pressure/economics , Cost-Benefit Analysis/methods , Patient Compliance , Pediatrics/economics , Sleep Apnea, Obstructive/economics , Sleep Apnea, Obstructive/therapy , Adolescent , Child , Child, Preschool , Continuous Positive Airway Pressure/methods , Female , Follow-Up Studies , Humans , Male , Pediatrics/methods , Polysomnography/economics , Polysomnography/methods
12.
Sleep Med ; 30: 77-81, 2017 02.
Article in English | MEDLINE | ID: mdl-28215268

ABSTRACT

INTRODUCTION: Little is known about the pediatric population at an increased risk of restless legs syndrome (RLS) and periodic limb movement disorder (PLMD). Polysomnographic data from the Caffeine for Apnea of Prematurity-Sleep (CAPS) study showed a high prevalence of elevated periodic limb movement index (PLMI) in a cohort of ex-preterm children, but the clinical importance of this finding, such as association with RLS, is unknown. We hypothesized that ex-preterm children would have a high prevalence of RLS and PLMD. METHODS: Ex-preterm children enrolled in CAPS, now aged 5-12 years, completed home polysomnography (PSG) and standardized questionnaires. A diagnosis of RLS or PLMD was established by participants meeting the International Classification of Sleep Disorders, 3rd edition, criteria based on questionnaires and polysomnograms. The clinically available serum ferritin levels were assessed. RESULTS: In total, 167 participants underwent polysomnography and completed all questionnaires. The overall prevalence of RLS was 14/167 (8.4%). An additional 13 subjects (7.8%) were found to have PLMD. Of the 26 participants who had PLMI > 5/h, seven (26.9%) had RLS and 13 (50%) had PLMD. The serum ferritin levels were <50 mcg/L (range -38.4) for all eight participants referred for testing. CONCLUSIONS: Children with a history of prematurity have a high prevalence of RLS, particularly those with elevated periodic limb movements. Iron deficiency likely contributes to RLS and PLMD symptoms in this population. Clinicians evaluating ex-preterm children with sleep disturbances should evaluate for RLS and PLMD. Further studies including serum ferritin evaluation are required to confirm these findings.


Subject(s)
Nocturnal Myoclonus Syndrome/epidemiology , Premature Birth , Restless Legs Syndrome/epidemiology , Child , Child, Preschool , Cohort Studies , Female , Ferritins/blood , Humans , Male , Pregnancy , Prevalence
13.
Sleep ; 40(4)2017 04 01.
Article in English | MEDLINE | ID: mdl-28199697

ABSTRACT

Objectives: The childhood obstructive sleep apnea syndrome (OSAS) is associated with behavioral abnormalities. Studies on the effects of OSAS treatment on behavior are conflicting, with few studies using a randomized design. Further, studies may be confounded by the inclusion of behavioral outcome measures directly related to sleep. The objective of this study was to determine the effect of adenotonsillectomy on behavior in children with OSAS. We hypothesized that surgery would improve behavioral ratings, even when sleep symptom items were excluded from the analysis. Methods: This was a secondary analysis of Child Behavior Checklist (CBCL) data, with and without exclusion of sleep-specific items, from the Childhood Adenotonsillectomy Trial (CHAT). CBCL was completed by caregivers of 380 children (7.0+1.4 [range 5-9] years) with OSAS randomized to early adenotonsillectomy (eAT) versus 7 months of watchful waiting with supportive care (WWSC). Results: There was a high prevalence of behavioral problems at baseline; 16.6% of children had a Total Problems score in the clinically abnormal range. At follow-up, there were significant improvements in Total Problems (p < .001), Internalizing Behaviors (p = .04), Somatic Complaints (p = .01), and Thought Problems (p = .01) in eAT vs. WWSC participants. When specific sleep-related question items were removed from the analysis, eAT showed an overall improvement in Total (p = .02) and Other (p = .01) problems. Black children had less improvement in behavior following eAT than white children, but this difference attenuated when sleep-related items were excluded. Conclusions: This large, randomized trial showed that adenotonsillectomy for OSAS improved parent-rated behavioral problems, even when sleep-specific behavioral issues were excluded from the analysis.


Subject(s)
Adenoidectomy , Child Behavior , Parents/psychology , Sleep Apnea, Obstructive/psychology , Sleep Apnea, Obstructive/surgery , Tonsillectomy , Black or African American , Child , Child, Preschool , Female , Humans , Male , Prevalence , Sleep , Sleep Apnea, Obstructive/physiopathology , White People
14.
Paediatr Respir Rev ; 22: 72-75, 2017 Mar.
Article in English | MEDLINE | ID: mdl-27818068

ABSTRACT

Over the last few decades, asthma and sleep disordered breathing (SDB) in children have experienced similar increases in prevalence, and have both been shown to have airway inflammation, leading investigators to postulate an association between asthma and SDB. However, whether this relationship is causal or not needs to be proven. In this manuscript, we use the most widely accepted epidemiologic criteria for causality, the Bradford Hill criteria, to test step-by-step whether the relation between asthma and SBD in children is causal or not. We found studies supporting 8 of the 9 criteria (strength, consistency, specificity, biological gradient, coherence and biological plausibility, experiment, and analogy) for association between asthma and SDB. However, we did not find any study showing temporality or directionality between asthma and SDB. Therefore, establishing a causal association between asthma and SDB is not yet possible.


Subject(s)
Asthma/epidemiology , Sleep Apnea Syndromes/epidemiology , Causality , Child , Child, Preschool , Humans , Infant
15.
Ann Am Thorac Soc ; 14(1): 76-84, 2017 Jan.
Article in English | MEDLINE | ID: mdl-27768852

ABSTRACT

RATIONALE: Prior researchers found that individual-level environmental and social indicators did not explain the racial disparity in obstructive sleep apnea syndrome. Neighborhood socioeconomic variables, as well as risk factors for a range of adverse behavioral and health outcomes, may better explain this racial disparity and help identify modifiable intervention targets. OBJECTIVES: To evaluate the associations of neighborhood socioeconomic variables with obstructive sleep apnea severity and to assess whether the neighborhood variables explain the association between race and obstructive sleep apnea severity. METHODS: We performed a cross-sectional analysis of data of 774 children in six cities who participated in the Childhood Adenotonsillectomy Trial. The outcome variable was the apnea-hypopnea index (AHI). Neighborhood socioeconomic variables were obtained on the basis of the children's residential addresses and information in the American Community Survey. Regression models were used to assess the associations among neighborhood conditions, race, and AHI. MEASUREMENTS AND MAIN RESULTS: Higher poverty rate and percentage of single-female-headed households were associated with higher AHI (P = 0.008 and 0.002, respectively). African American race was associated with a 1.33 (1.08-1.64 95% confidence interval)-fold increase in AHI, adjusting for age and sex. After controlling for poverty rate or percentage of single-female-headed households with children, the association between race and AHI levels was no longer significant (P = 0.15 and 0.26, respectively), and the magnitude of race association decreased 34 or 55%, suggesting that the association between race and AHI levels was largely explained by poverty rate or percentage of single-female-headed households with children. CONCLUSIONS: Neighborhood socioeconomic variables in comparison with individual-level socioeconomic indicators provides better explanations for the racial disparity in pediatric obstructive sleep apnea syndrome. Further research aimed at identifying factors that aggregate in disadvantaged neighborhoods and increase sleep apnea risk may suggest modifiable intervention targets. Clinical trial registered with clinicaltrials.gov (NCT00560859).


Subject(s)
Ethnicity , Health Status Disparities , Poverty , Residence Characteristics , Single-Parent Family , Sleep Apnea, Obstructive/epidemiology , Social Class , Black or African American , Child , Child, Preschool , Female , Hispanic or Latino , Humans , Male , Severity of Illness Index , Sleep Apnea, Obstructive/physiopathology , White People
16.
Thorax ; 72(8): 720-728, 2017 08.
Article in English | MEDLINE | ID: mdl-27503232

ABSTRACT

BACKGROUND: The obstructive sleep apnoea syndrome (OSAS) results from a combination of structural and neuromotor factors; however, the relative contributions of these factors have not been studied during the important developmental phase of adolescence. We hypothesised that adenotonsillar volume (ATV), nasopharyngeal airway volume (NPAV), upper airway critical closing pressure (Pcrit) in the hypotonic and activated neuromotor states, upper airway electromyographic response to subatmospheric pressure and the ventilatory response to CO2 during sleep would be major predictors of OSAS risk. METHODS: 42 obese adolescents with OSAS and 37 weight-matched controls underwent upper airway MRI, measurements of Pcrit, genioglossal electromyography and ventilatory response to CO2 during wakefulness and sleep. RESULTS: ATV, NPAV, activated and hypotonic Pcrit, genioglossal electromyography and ventilatory response to CO2 during sleep were all associated with OSAS risk. Multivariate models adjusted for age, gender, body mass index and race indicated that ATV, NPAV and activated Pcrit each independently affected apnoea risk in adolescents; genioglossal electromyography was independently associated in a reduced sample. There was significant interaction between NPAV and activated Pcrit (p=0.021), with activated Pcrit more strongly associated with OSAS in adolescents with larger NPAVs and NPAV more strongly associated with OSAS in adolescents with more negative activated closing pressure. CONCLUSIONS: OSAS in adolescents is mediated by a combination of anatomic (ATV, NPAV) and neuromotor factors (activated Pcrit). This may have important implications for the management of OSAS in adolescents.


Subject(s)
Obesity/complications , Sleep Apnea, Obstructive/physiopathology , Sleep/physiology , Adolescent , Child , Electromyography , Female , Humans , Magnetic Resonance Imaging , Male , Palate, Soft/diagnostic imaging , Polysomnography , Sleep Apnea, Obstructive/diagnosis , Sleep Apnea, Obstructive/etiology
17.
Neurology ; 87(7 Suppl 1): S13-20, 2016 Aug 16.
Article in English | MEDLINE | ID: mdl-27527645

ABSTRACT

OBJECTIVE: Plexiform neurofibromas (PNs) are complex, benign nerve sheath tumors that occur in approximately 25%-50% of individuals with neurofibromatosis type 1 (NF1). PNs that cause airway compromise or pulmonary dysfunction are uncommon but clinically important. Because improvement in sleep quality or airway function represents direct clinical benefit, measures of sleep and pulmonary function may be more meaningful than tumor size as endpoints in therapeutic clinical trials targeting airway PN. METHODS: The Response Evaluation in Neurofibromatosis and Schwannomatosis functional outcomes group reviewed currently available endpoints for sleep and pulmonary outcomes and developed consensus recommendations for response evaluation in NF clinical trials. RESULTS: For patients with airway PNs, polysomnography, impulse oscillometry, and spirometry should be performed to identify abnormal function that will be targeted by the agent under clinical investigation. The functional group endorsed the use of the apnea hypopnea index (AHI) as the primary sleep endpoint, and pulmonary resistance at 10 Hz (R10) or forced expiratory volume in 1 or 0.75 seconds (FEV1 or FEV0.75) as primary pulmonary endpoints. The group defined minimum changes in AHI, R10, and FEV1 or FEV0.75 for response criteria. Secondary sleep outcomes include desaturation and hypercapnia during sleep and arousal index. Secondary pulmonary outcomes include pulmonary resistance and reactance measurements at 5, 10, and 20 Hz; forced vital capacity; peak expiratory flow; and forced expiratory flows. CONCLUSIONS: These recommended sleep and pulmonary evaluations are intended to provide researchers with a standardized set of clinically meaningful endpoints for response evaluation in trials of NF1-related airway PNs.


Subject(s)
Clinical Trials as Topic/methods , Neurofibroma, Plexiform/therapy , Neurofibromatosis 1/therapy , Oscillometry/methods , Polysomnography/methods , Spirometry/methods , Humans , Lung/physiopathology , Neurofibroma, Plexiform/physiopathology , Neurofibromatosis 1/physiopathology , Sleep/physiology , Treatment Outcome
18.
Sleep ; 39(11): 2005-2012, 2016 Nov 01.
Article in English | MEDLINE | ID: mdl-27568804

ABSTRACT

STUDY OBJECTIVES: To describe parental reports of sleepiness and sleep duration in children with polysomnography (PSG)-confirmed obstructive sleep apnea (OSA) randomized to early adenotonsillectomy (eAT) or watchful waiting with supportive care (WWSC) in the ChildHood Adenotonsillectomy Trial (CHAT). We hypothesized children with OSA would have a larger improvement in sleepiness 6 mo following eAT compared to WWSC. METHODS: Parents of children aged 5.0-9.9 y completed the Epworth Sleepiness Scale modified for children (mESS) and the Pediatric Sleep Questionnaire-Sleepiness Subscale (PSQ-SS). PSG was performed at baseline and at 7-mo endpoint. Children underwent early adenotonsillectomy or WWSC. RESULTS: The mESS and PSQ-SS classified 24% and 53% of the sample as excessively sleepy, respectively. At baseline, mean mESS score was 7.4 ± 5.0 (SD) and mean PSQ-SS score was 0.44 ± 0.30. Sleepiness scores were higher in African American children; children with shorter sleep duration; older children; and overweight children. At endpoint, mean mESS score decreased by 2.0 ± 4.2 in the eAT group versus 0.3 ± 4.0 in the WWSC group (P < 0.0001); mean PSQ-SS score decreased 0.29 ± 0.40 in eAT versus 0.08 ± 0.40 in the WWSC group (P < 0.0001). Despite higher baseline sleepiness, African American children experienced similar improvement with adenotonsillectomy than other children. Improvement in sleepiness was weakly associated with improved apnea-hypopnea index or oxygen desaturation indices, but not with change in other polysomnographic measures. CONCLUSIONS: Sleepiness assessed by parent report was prevalent; improved more after eAT than after WWSC; and was not strongly predicted by sleep disturbances identified by PSG. CLINICAL TRIAL REGISTRATION: Childhood Adenotonsillectomy Study for Children with OSA (CHAT). ClinicalTrials.gov Identifier #NCT00560859.


Subject(s)
Adenoidectomy , Disorders of Excessive Somnolence/etiology , Sleep Apnea, Obstructive/surgery , Sleep Stages , Tonsillectomy , Child , Child, Preschool , Disorders of Excessive Somnolence/diagnosis , Female , Follow-Up Studies , Humans , Male , Parents , Sleep Apnea, Obstructive/complications , Treatment Outcome , Watchful Waiting
19.
Pediatrics ; 138(2)2016 08.
Article in English | MEDLINE | ID: mdl-27464674

ABSTRACT

OBJECTIVE: Research reveals mixed evidence for the effects of adenotonsillectomy (AT) on cognitive tests in children with obstructive sleep apnea syndrome (OSAS). The primary aim of the study was to investigate effects of AT on cognitive test scores in the randomized Childhood Adenotonsillectomy Trial. METHODS: Children ages 5 to 9 years with OSAS without prolonged oxyhemoglobin desaturation were randomly assigned to watchful waiting with supportive care (n = 227) or early AT (eAT, n = 226). Neuropsychological tests were administered before the intervention and 7 months after the intervention. Mixed model analysis compared the groups on changes in test scores across follow-up, and regression analysis examined associations of these changes in the eAT group with changes in sleep measures. RESULTS: Mean test scores were within the average range for both groups. Scores improved significantly (P < .05) more across follow-up for the eAT group than for the watchful waiting group. These differences were found only on measures of nonverbal reasoning, fine motor skills, and selective attention and had small effects sizes (Cohen's d, 0.20-0.24). As additional evidence for AT-related effects on scores, gains in test scores for the eAT group were associated with improvements in sleep measures. CONCLUSIONS: Small and selective effects of AT were observed on cognitive tests in children with OSAS without prolonged desaturation. Relative to evidence from Childhood Adenotonsillectomy Trial for larger effects of surgery on sleep, behavior, and quality of life, AT may have limited benefits in reversing any cognitive effects of OSAS, or these benefits may require more extended follow-up to become manifest.


Subject(s)
Adenoidectomy , Cognition , Sleep Apnea, Obstructive/surgery , Tonsillectomy , Child , Child, Preschool , Cognition Disorders/etiology , Female , Humans , Male , Neuropsychological Tests , Sleep Apnea, Obstructive/complications
20.
Sleep ; 39(9): 1647-52, 2016 Sep 01.
Article in English | MEDLINE | ID: mdl-27253764

ABSTRACT

STUDY OBJECTIVES: Children with the obstructive sleep apnea (OSA) have impaired upper airway two-point discrimination compared to controls. In addition, blunted vibration threshold detection (VT) in the palate has been recognized in adults with OSA, but has not been studied in children. Both findings are indicative of a defect in the afferent limb of the upper airway dilator reflex that could prevent upper airway dilation secondary to airway loading, resulting in airway collapse. We hypothesized that children with OSA have impaired palate VT compared to controls, and that this improves after OSA treatment. METHODS: Case-control study. Children with OSA and healthy non-snoring controls underwent polysomnography and palate VT measurements. Children with OSA were retested after adenotonsillectomy. RESULTS: 29 children with OSA (median [interquartile range] age = 9.5 [7.5-12.6] years, obstructive apnea-hypopnea index [OAHI] = 11.3 [5.7-19.5] events/h, BMI z = 1.8 [1.3-2.1]) and 32 controls (age = 11.2 [9.3-13.5] years, P = 0.1; OAHI = 0.5 [0.1-0.7] events/h, P < 0.001; BMI z = 1 [0.3-1.7], P = 0.004) were tested. OSA palate VT (1.0 [0.8-1.5] vibration units) was similar to that of controls (1 [0.8-1.3], P = 0.37). 20 children with OSA were retested 4.4 (3.2-7.1) months after treatment. OAHI decreased from 13.1 (5.8-19) to 0.6 (0.2-2.5) events per hour (P < 0.001) postoperatively, but palate VT did not change (before = 1 [0.7-1.5], after = 1.2 [0.8-1.4], P = 0.37). CONCLUSIONS: Children with OSA and controls have similar palate VT. Unlike in adults, palate VT does not seem to be affected by childhood OSA.


Subject(s)
Palate/physiopathology , Sensory Thresholds/physiology , Sleep Apnea, Obstructive/physiopathology , Vibration , Adenoidectomy , Adolescent , Case-Control Studies , Child , Female , Humans , Male , Polysomnography , Single-Blind Method , Sleep Apnea, Obstructive/surgery , Tonsillectomy , Treatment Outcome
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