ABSTRACT: Down syndrome (DS) is the most common genetic cause of intellectual disability and results from an extra chromosome 21 (Trisomy 21). Sleep issues and/or obstructive sleep apnea (OSA) are assumed to be part of the DS phenotype with a high prevalence but are often under recognized. This cross-sectional study of children with DS examines the caregiver-reported sleep behaviors of 108 children with DS, ranging in age from 1.50 to 13.40 years (mean?=?5.18 years) utilizing a standardized assessment tool, the Children's Sleep Habit Questionnaire (CSHQ). The CSHQ revealed 76% of children with DS had sleep problems, which began at a young age, and continue to persist and may recur with increasing age. Furthermore, children with DS who undergone adenoidectomy and tonsillectomy for OSA continued to have sleep problems suggesting that ongoing monitoring of sleep issues is needed in this population. Implications of sleep problems and recommended anticipatory guidance and intervention are discussed.
Project description:The study aims to examine drug-induced sleep endoscopy (DISE) in the decision-making process of pediatric obstructive sleep apnea (OSA) patients with small tonsils. This was a retrospective study of children who underwent awake flexible endoscopy, DISE, and adenoidectomy with/without tonsillectomy at the Shanghai Children's Medical Center between 03/2015 and 12/2016. Tonsillectomy was performed for tonsillar obstruction found by DISE. Adenoidectomy was performed for all children. Cardio-pulmonary coupling (CPC) and oximetry were observed before/after surgery. The study included 126 children: 56 (44.4%) with grade 2 tonsils and 70 (55.6%) with grade 1. Mean age was 5.7±3.2 (range, 2.8-10.4) years and mean BMI of 15.7±5.5 kg/m2. Unexpectedly, DISE showed tonsillar obstruction in 57 (45.2%) children, including 44 (78.6%) with grade 2 tonsils and 13 (18.6%) with grade 1. Therefore, DISE-directed tonsillectomy was performed for 57 patients. There was an improvement in respiratory disturbance index (RDI) and oxygen saturation nadir in the DISE (P = 0.0007, P = 0.037) and control (P = 0.001, P = 0.023) groups 6 months after surgery, but RDI improvement was better in the DISE group compared with controls 1 year after surgery (P = 0.042). DISE is a good way to determine the necessity of tonsillectomy in pediatric OSA patients with small tonsils.
Project description:<h4>Study objective</h4>Adenotonsillectomy is the recommended treatment for children with obstructive sleep apnea (OSA). Since adenoidectomy alone may be associated with significantly lower morbidity, mortality, and cost, we aimed to investigate whether adenoidectomy alone is a reasonable and appropriate treatment for children with OSA.<h4>Methods</h4>Five-hundred fifteen consecutive children diagnosed with moderate-to-severe OSA (apnea-hypopnea index > 5) based on polysomnography and who underwent adenoidectomy or adenotonsillectomy were reevaluated after 17-73 months (mean 41) for residual or recurrent OSA using a validated questionnaire (Pediatric Sleep Questionnaire, PSQ). Failure of OSA resolution was defined as a positive mean PSQ score ≥ 0.33. Contribution of age, obesity, tonsil size, and OSA severity at baseline to adenoidectomy or adenotonsillectomy failure was examined.<h4>Results</h4>Positive PSQ score occurred in 15% of the entire sample and was not influenced by age or gender. No difference in failure rate was observed between adenoidectomy and adenotonsillectomy for children who were not obese with apnea-hypopnea index < 10 and had small tonsils (< 3). Children with apnea-hypopnea index ≥ 10 and/or tonsil size ≥ 3 showed a higher failure rate after adenoidectomy compared to adenotonsillectomy (20% versus 9.8%, p = 0.028).<h4>Conclusions</h4>We suggest that subjective, long term outcomes of adenoidectomy are comparable to those of adenotonsillectomy in non-obese children under 7 years old with moderately OSA and small tonsils. Hence, adenoidectomy alone is a reasonable option in some children. Future prospective randomized studies are warranted to define children who may benefit from adenoidectomy alone and those children in whom adenoidectomy alone is unlikely to succeed.
Project description:<h4>Introduction</h4>Obstructive sleep apnoea (OSA) is a sleep-breathing disorder that seems likely to have long-term negative social and health consequences in children and adolescents. There are no established standard management approaches when the first line of therapy, the tonsillectomy and adenoidectomy (T&A), is not indicated or fails to address paediatric OSA (residual paediatric OSA). This protocol describes a prospective cohort study that aims to assess the effectiveness of orthodontic interventions for managing residual paediatric OSA in patients with concomitant craniofacial issues.<h4>Methods and analysis</h4>Children aged 6-16 years who with an OSA diagnosis and did not benefit from previous T&A or qualified for T&A will be recruited. Orthodontic intervention(s), when adequately indicated (maxillary expansion, mandibular advancement or maxillary complex advancement with skeletal anchored headgear), and a control (orthodontic intervention declined) cohorts will be involved. A sample size of 70 participants (n=35 per cohort) is planned. Effectiveness data will be assessed through nocturnal polysomnography, a craniofacial index, sleep questionnaires and medical records. Additionally, the association of residual OSA and two comorbidities, obesity and asthma, will be investigated through assessing blood, urine and saliva metabolites. The changes on body mass index will also be investigated as a secondary outcome. Other additional outcomes, including association between residual paediatric OSA and periodic limbs movement, restless leg syndrome, insomnia, and the use of abiometric shirt to sleep monitoring purposes will also be considered. All participants will be followed up for 12 months after treatment allocation. The effectiveness of the intervention will be analysed by the assessment of sleep parameters, medical history (from medical chart reviews), questionnaire responses, craniofacial characteristics and metabolomic markers using an algorithm to be developed.<h4>Ethics and dissemination</h4>This study was approved by the Health Research Ethics Board-Health Panel, University of Alberta, Edmonton, Canada (Pro00084763). The findings will be shared with scientific and patient content-specific social network communities to maximise their impact on clinical practice and future research in the study topic.<h4>Trial registration number</h4>NCT03821831; Pre-results.
Project description:Importance:Evidence indicates correlations between lingual tonsil hypertrophy and pediatric obstructive sleep apnea (OSA). However, to our knowledge, a meta-analysis of surgical outcomes for lingual tonsillectomy in children with OSA has not been conducted. Objective:To evaluate the therapeutic outcomes of lingual tonsillectomy for treatment of pediatric OSA. Data Sources:The study protocol was registered on PROSPERO (CRD42015027053). PubMed, MEDLINE, EMBASE, and the Cochrane Reviews databases were searched independently by 2 authors for relevant articles published by September 2016. Study Selection:The literature search identified English-language studies that used polysomnography to evaluate children with lingual tonsil hypertrophy and OSA after lingual tonsillectomy alone. The search keywords were lingual tonsil, lingual tonsillectomy, sleep endoscopy, sleep apnea, and child. Data Extraction and Synthesis:Polysomnographic data from each study were extracted. A random-effects model pooled postoperative sleep variable changes and success rates for lingual tonsillectomy in treating pediatric OSA. Main Outcomes and Measures:Four outcomes for lingual tonsillectomy were analyzed. These included net postoperative changes in the apnea-hypopnea index (AHI), net postoperative changes in the minimum oxygen saturation, the overall success rate for a postoperative AHI less than 1, and the overall success rate for a postoperative AHI less than 5. Results:This meta-analysis consisted of 4 studies (mean sample size, 18.25 patients), with a total of 73 unique patients (mean [SD] age, 8.3 [1.1] years). Fifty-nine percent (27 of 46) of the patients were male, and 1 of the 4 studies did not specify number of males. Lingual tonsillectomy was indicated for persistent OSA after adenotonsillectomy in all cases. Lingual tonsil hypertrophy was evaluated using computed tomography or magnetic resonance imaging in 1 study, sleep endoscopy in 2 studies, and cine magnetic resonance imaging in 1 study. The mean change in the AHI after lingual tonsillectomy was a reduction of 8.9 (95% CI, -12.6 to -5.2) events per hour. The mean change in the minimum oxygen saturation after lingual tonsillectomy was an increase of 6.0% (95% CI, 2.7%-9.2%). The overall success rate was 17% (95% CI, 7%-35%) for a postoperative AHI less than 1 and 51% (95% CI, 25%-76%) for a postoperative AHI less than 5. Postoperative complications that developed included airway obstruction, bleeding, and pneumonia. Conclusions and Relevance:Lingual tonsillectomy is an effective surgical management for children with OSA caused by lingual tonsil hypertrophy, and it achieves significant improvement in the AHI and the minimum oxygen saturation. However, children frequently have residual OSA after lingual tonsillectomy, and postoperative complications must be carefully managed.
Project description:Importance:Ibuprofen is an effective analgesic after tonsillectomy alone or tonsillectomy with adenoidectomy, but concerns remain about whether it increases postoperative hemorrhage. Objective:To investigate the effect of ibuprofen compared with acetaminophen on posttonsillectomy bleeding (PTB) requiring surgical intervention in children. Design, Setting, and Participants:A multicenter, randomized, double-blind noninferiority trial was conducted at 4 tertiary medical centers (Massachusetts Eye and Ear Infirmary, Boston; Naval Medical Center, San Diego, California; Naval Medical Center, Portsmouth, Virginia; Madigan Army Medical Center, Tacoma, Washington). A total of 1832 children were assessed for eligibility (presence of sleep-disordered breathing or obstructive sleep apnea, adenotonsillar hypertrophy, or infectious tonsillitis undergoing extracapsular tonsillectomy by electrocautery). Of these, 1091 were excluded because they did not meet eligibility criteria (n?=?681) or refused to participate (n?=?410); thus, 741 children aged 2 to 18 years undergoing tonsillectomy alone or tonsillectomy with adenoidectomy were enrolled between May 3, 2012, and January 20, 2017. Interventions:Participants were randomized to receive ibuprofen, 10 mg/kg (n?=?372), or acetaminophen, 15 mg/kg (n?=?369), every 6 hours for the first 9 postoperative days. Main Outcomes and Measures:Rate and severity of posttonsillectomy bleeding were recorded using a postoperative bleeding severity scale: type 1 (bleeds that were observed at home or evaluated in the emergency department without further intervention), type 2 (bleeds that required readmission for observation), and type 3 (bleeds that required a return to the operating room for control of hemorrhage). Type 3 bleeding was the main outcome measure. The noninferiority margin was set at 3%, and modified intention-to-treat analysis was used. Results:Of the 741 children enrolled, 688 children (92.8%) (median [interquartile range] age, 5  years; 366 boys [53.2%]) received the study medication and were included in a modified intention-to-treat analysis. The rate of bleeding requiring operative intervention was 1.2% in the acetaminophen group and 2.9% in the ibuprofen group (difference, 1.7%; 97.5% CI upper limit, 3.8%; P?=?.12 for noninferiority). There were no significant adverse events or deaths. Conclusions and Relevance:This study could not exclude a higher rate of severe bleeding in children receiving ibuprofen after tonsillectomy alone or tonsillectomy with adenoidectomy. This finding should be considered when selecting a postoperative analgesic regimen. Further studies are needed to understand if bleeding risk is affected when ibuprofen is used for a shorter duration or in combination with acetaminophen for postoperative analgesia. Trial Registration:ClinicalTrials.gov identifier: NCT01605903.
Project description:<h4>Aims</h4>Children on the autism spectrum are more likely to have sleep problems than non-autistic children. Sleep disturbance may exacerbate emotional and behavioral problems of children on the autism spectrum. A better understanding of the biological mechanisms underlying sleep disturbance provide clues to better management for this co-morbid condition in autism. The goal of the current study is to identify genetic variants associated with sleep disturbance and melatonin levels in autistic children.<h4>Methods</h4>A total of 969 children on the autism spectrum were genotyped using the Global Screening Array v1 or Global Screening Array v2. Sleep problems were assessed using the Children's Sleep Habits Questionnaire (CSHQ). Melatonin levels were measured using the urine samples of 219 probands. The relationship between the melatonin level and CSHQ score was examined using the general linear model. The genetic variants associated with the CSHQ score and melatonin level as two separate quantitative traits were determined using genomewide association studies.<h4>Results</h4>The data indicates that urine melatonin levels were positively associated with CSHQ scores, suggesting that autistic children with a poorer sleep qualiy could has higher melatonin level. Furthermore, genetic assocication studies suggest that genetic pathways involved in pro-inflammatory responses might be involved in sleep disturbance, while genetic pathways involved in catecholamine-secreting PC12 cells and Schwann cells could be associated with melatonin levels.<h4>Conclusions</h4>Taken together, our findings indicate that sleep disturbance and melatonin metabolism could be attributable to distinct biological mechanisms in autistic children since they might not share genetic contributors.
Project description:<h4>Background</h4>Children with severe airway obstruction tend to have a vertical direction of growth, class II malocclusion, and narrow arches. Adenoidectomy and tonsillectomy were recommended for the promotion of balanced dentition growth in these children.The aim of this study was to determine the effect of adenoidectomy and tonsillectomy on the growth of dental morphology in children with airway obstruction.<h4>Methods</h4>A comprehensive search of the Medline, Embase, Web of science, and OVID databases for studies published through to January 17, 2016 was conducted. Prospective, comparative, clinical studies assessing the efficacy of adenoidectomy, or tonsillectomy in children with airway obstruction were included. The weighted mean difference (WMD) and 95% confidence interval (CI) were used for continuous variables. Forest plots were drawn to demonstrate effects in the meta-analyses.<h4>Results</h4>Eight papers were included in our study. We found that adenoidectomy and tonsillectomy led to a significant change in nasal-breathing in children with airway obstruction. Children with airway obstruction had a significantly narrower posterior maxillary dental arch than children without airway obstruction (WMD = -0.94, 95% CI [-1.13, -0.76]; P < 0.001). After surgery, these children still had a significantly narrower dental arch than the nasal-breathing children (WMD = -0.60, 95% CI [-0.79, -0.42]; P < 0.001). In terms of dental arch width, malocclusion, palatal height, overjet, overbite, dental arch perimeter, and arch length, a tendency toward normalization was evident following adenoidectomy or tonsillectomy, with no significant differences evident between the surgical group and the normal group. The small number of studies and lack of randomized controlled trials were the main limitations of this meta-analysis.<h4>Conclusions</h4>Following adenoidectomy and tonsillectomy, the malocclusion and narrow arch width of children with airway obstruction could not be completely reversed. Therefore, other treatments such as functional training or orthodontic maxillary widening should be considered after removing the obstruction in the airway.
Project description:BACKGROUND:To delineate sleep habits and problems in children with 22q11.2 deletion syndrome (22q11DS). METHODS:Thirty children, age 1-15 (mean 6.8) years, participated in the study, which was an internet-based anonymous survey of parents of children with 22q11DS administered via the 22q11.2 Foundation. The main outcome was the Childhood Sleep Habits Questionnaire (CSHQ). RESULTS:Scores on the CSHQ demonstrated clinically significant sleep problems in 29 of the 30 children. When compared with previously reported normative values for typically developing children of the same age, children with 22q11DS had significantly greater sleep problems. Only 30% of children had previously undergone sleep study. While about half of children had tried a medication for sleep, it usually was not felt to be helpful. In contrast, parents reported that behavioral interventions, such as consistent bedtime routine and appropriate sleep environment, were helpful. This is one of the first studies to specifically address sleep problems other than obstructive sleep apnea in children with 22q11DS. CONCLUSIONS:The findings suggest children with 22q11DS may have a higher risk of experiencing clinical sleep problems, compared to typically developing children. Consideration of additional screening and treatment of sleep disorders in children with 22q11DS is warranted.
Project description:Background:Autism spectrum disorder (ASD) is a type of neurodevelopmental disease that is frequently accompanied by sleep disorder. Herein, we investigated changes in the gut microbiota and its metabolites correlated with core symptoms and sleep problems in children with ASD. Methods:One hundred and twenty children diagnosed with ASD based on Diagnostic and Statistical Manual of Mental Disorders (DSM-5) criteria were enrolled in our study. The Autism Behavior Checklist (ABC), Social Responsiveness Scale (SRS), and Childhood Autism Rating Scale (CARS) were used to assess autism symptoms, and the Children Sleep Habits Questionnaire (CSHQ) was employed to evaluate sleep problems in children with ASD. The 120 children were divided into a sleep disorder group (n = 60) and a control group without sleep disorder (n = 60) according to the CSHQ answers. Illumina MiSeq analysis of 16S rRNA genes was used to compare differences in gut microbiota, and metabolomics analysis was employed to asses associated metabolites. Results:SRS and CARS scores for the sleep disorder group were significantly higher than for the control group (p < 0.05). The abundances of butyrate-producing bacteria Faecalibacterium and Agathobacter were reduced significantly in the sleep disorder group (p < 0.05), and this was negatively correlated with CSHQ score (p = 0.007 and p = 0.014, respectively). The abundance of Agathobacter was also negatively associated with the ABC language score (p = 0.044). Furthermore, levels of 3-hydroxybutyric acid and melatonin were significantly lower (p < 0.05) while serotonin levels were higher (p < 0.05) in the sleep disorder group. The 3-hydroxybutyric acid level was positively associated with Faecalibacterium abundance (p = 0.000), and melatonin was positively associated with the abundance of Faecalibacterium (p = 0.036) and Agathobacter (p = 0.041). We also observed negative correlations between 3-hydroxybutyric acid and CSHQ (p = 0.000) and CARS (p = 0.009), between melatonin and CSHQ (p = 0.002) and ABC sensory score (p = 0.021), and a positive correlation between serotonin and CSHQ (p = 0.002) and ABC sensory score (p = 0.025). Conclusions:ASD children with sleep disorder exhibited declines in the abundance of Faecalibacterium and Agathobacter, decreased levels of 3-hydroxybutyric acid and melatonin, and an increase in serotonin. These changes may aggravate sleep problems and core symptoms in children with ASD.
Project description:<h4>Introduction</h4>Diagnosis and treatment of obstructive sleep apnea (OSA) in children is often delayed due to the high prevalence and limited physician and sleep testing resources. As a result, children may be referred to multiple specialties, such as pediatric sleep medicine and pediatric otolaryngology, resulting in long waitlists.<h4>Method</h4>We used data from our pediatric OSA clinic to identify predictors of tonsillectomy and/or adenoidectomy (AT). Before being seen in the clinic, parents completed the Pediatric Sleep Questionnaire (PSQ) and screening questionnaires for restless leg syndrome (RLS), nasal rhinitis, and gastroesophageal reflux disease (GERD). Tonsil size data were obtained from patient charts and graded using the Brodsky-five grade scale. Children completed an overnight oximetry study before being seen in the clinic, and a McGill oximetry score (MOS) was assigned based on the number and depth of oxygen desaturations. Logistic regression, controlling for otolaryngology physician, was used to identify significant predictors of AT. Three triage algorithms were subsequently generated based on the univariate and multivariate results to predict AT.<h4>Results</h4>From the OSA cohort, there were 469 eligible children (47% female, mean age = 8.19 years, SD = 3.59), with 89% of children reported snoring. Significant predictors of AT in univariate analysis included tonsil size and four PSQ questions, (1) struggles to breathe at night, (2) apneas, (3) daytime mouth breathing, and (4) AM dry mouth. The first triage algorithm, only using the four PSQ questions, had an odds ratio (OR) of 4.02 for predicting AT (sensitivity = 0.28, specificity = 0.91). Using only tonsil size, the second algorithm had an OR to predict AT of 9.11 (sensitivity = 0.72, specificity = 0.78). The third algorithm, where MOS was used to stratify risk for AT among those children with 2+ tonsils, had the same OR, sensitivity, and specificity as the tonsil-only algorithm.<h4>Conclusion</h4>Tonsil size was the strongest predictor of AT, while oximetry helped stratify individual risk for AT. We recommend that referral letters for snoring children include graded tonsil size to aid in the triage based on our findings. Children with 2+ tonsil sizes should be triaged to otolaryngology, while the remainder should be referred to a pediatric sleep specialist.