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This preliminary study investigated the tolerability and efficacy of a novel mattress technology-the Sound-To-Sleep (STS) system-in the treatment of sleep problems in children with autism.After screening, 45 children, ages 2.5 to 12.9 years, were randomized to order of mattress technology use (On-Off vs. Off-On). Treatment conditions (On vs. Off) lasted two weeks with immediate crossover. Tolerability, including study discontinuation and parent-report of mattress tolerance and ease of use, was tracked throughout the study. Efficacy assessments were obtained at baseline, prior to crossover, and end of study and included measures of autism traits, other psychopathology symptoms, sensory abnormalities, communication difficulties, quality of life, sleep diary parameters, and single-blinded actigraphy-derived sleep parameters. Statistical analyses evaluated differences in tolerability and efficacy when the STS system was on versus off.STS system use was well tolerated (n = 2, 4.4% dropout) and resulted in parent-reported sleep quality improvements (STS off mean = 4.3, 95% CI = 4.05-4.54 vs. on mean = 4.9, 95%CI = 4.67-5.14). The technology was described by parents as very easy to use and child tolerance was rated as good. Parent-diary outcomes indicated improvements in falling asleep and reduced daytime challenging behavior. Actigraphy-derived sleep parameters indicated improved sleep duration and sleep efficiency. Improvements in child and family quality of life were identified on parent questionnaires.A future large sample phase 2 trial of the STS system is warranted and would benefit from extended study duration, an objective primary efficacy outcome, and careful attention to methodological issues that promote compliance with the intervention and study procedures.

Objective There is a lack of consensus and a paucity of data regarding how to best treat pediatric patients with mild obstructive sleep apnea. The objective of our study was to compare outcomes following adenotonsillectomy vs. observation in children with mild obstructive sleep apnea based on polysomnography results. Methods A retrospective chart review was performed on children ages 9 months to 9 years with 2 or more polysomnograms completed at a tertiary care academic center. Children diagnosed with mild obstructive sleep apnea (obstructive apnea–hypopnea index 1–5) on polysomnography performed from 1999 to 2013 were included. Patients underwent adenotonsillectomy or watchful waiting for obstructive sleep apnea. The primary outcome was the change in apnea–hypopnea index. Results There were 62 patients who met inclusion criteria for the study; 19 of the 62 patients were obese, while 15 had a craniofacial syndrome or hypotonia. Eighteen patients underwent adenotonsillectomy for mild obstructive sleep apnea while 44 were observed. The mean apnea–hypopnea index of patients after adenotonsillectomy improved from 3.50 (95% Confidence Interval [CI] 2.97–4.03) to 2.69 (95% CI 1.48–3.90), while the mean apnea–hypopnea index of the observation group worsened from 3.09 (95% CI 2.76–3.42) to 5.18 (95% CI 2.46–7.90). Between-group analysis showed significant improvement in the surgery group ( p = 0.03), with a persistent improvement on multivariate analysis adjusting for baseline apnea–hypopnea index ( p = 0.05). This difference was seen mostly in non-obese, non-syndromic children ( p = 0.04). There was no significant difference between groups amongst obese ( p = 0.25) and syndromic ( p = 0.36) patients. Conclusions Adenotonsillectomy leads to a significant improvement in apnea–hypopnea index on follow-up polysomnography over an observational approach, especially in non-obese, non-syndromic children. A prospective, randomized trial is necessary to help determine appropriate treatment strategies for pediatric mild obstructive sleep apnea.

5p- is a well-defined syndrome, but phenotypic correlations of 5q are poorly described in the literature. We present a case of a female child with interstitial deletion in the 5q13.1q15 region. Comparison of the clinical features of this patient with others reported in the literature suggests an emerging clinical syndrome defined by short stature, failure to thrive, mental retardation, slanting palpebral fissures, malformed ears, short neck and depressed nasal bridge. Based on our endocrine testing, we hypothesize that the short stature could be, in part, due to growth hormone deficiency. The recent assignment of growth hormone receptor gene to the short arm of chromosome 5 and the presence of several genes for growth factors and growth factor receptors on 5q raise interesting possibilities for the explanation of short stature in such cases.

Caffeine, a commonly consumed psychoactive substance, can have significant effects on sleep. Caffeine intake among children is increasing, mainly in the form of sodas. However, adolescent caffeine consumers may lack knowledge about the caffeine content in common beverages. If true, this very fact may hamper the assessment of the effects of caffeine consumption on sleep in children if such assessments are a priori dependent on responders being able to reliably distinguish between caffeinated and noncaffeinated beverages. This preliminary study investigated adolescents' caffeine knowledge and intake at a Cleveland-area public middle school.Seventh- and eighth-grade students were surveyed using: (1) the Caffeine Literacy and Sleep Study (CLASS), a 15-question pilot instrument designed to assess caffeine knowledge and intake by type, quantity and timing, as well as sleep habits; and (2) the Cleveland Adolescent Sleepiness Questionnaire (CASQ), a validated survey measuring excessive daytime sleepiness in adolescents. These questionnaires were distributed and collected during a specified class period.Of the 635 seventh- and eighth-grade students who attended school on the day of the study, 555 (87%) participated. Lack of knowledge about caffeine content of particular drinks was noted in seventh and eighth graders of both sexes with nearly 29% unaware that their favorite drinks contain caffeine and more than 50% unable to correctly identify the drinks with the most caffeine. A low percentage of students correctly identified light-colored sodas lacking caffeine: 7-Up (24.1%), Sierra Mist (38.9%), ginger ale (39.8%), Sprite (39.8%), and Fresca (53.7%). The percentages of students correctly identifying caffeinated light-colored beverages were: Arizona Green Tea (43.5%), Mello Yellow (50.9%), and AW cream soda (67.6%). However, Mountain Dew was correctly identified by most (93.5%) as caffeinated.Students were not consistently able to identify caffeine content or lack thereof in some common beverages. The results of this pilot study show that caffeine literacy in adolescents warrants further investigation and educational intervention.

While neck circumference (NC) is a useful predictor of obstructive sleep apnea (OSA) in adults, childhood OSA is difficult to predict clinically. We utilized the neck circumference-height ratio (NHR) to normalize NC in growing children. Our study aimed to determine if (1) NC is a reproducible clinical measurement; (2) NHR predicts OSA in children; (3) this metric translates to adults.For this retrospective study, paired NC measurements (from clinic and sleep laboratory) in 100 consecutive adult subjects were used to confirm inter-observer reproducibility. Polysomnographic (PSG) and anthropometric data from children aged 5-18 years presenting consecutively between July 2007 and February 2012 was obtained. Children with genetic syndromes, severe neurological disorders, craniofacial abnormalities, tracheostomy, past adenotonsillectomy, in-hospital PSG or sleep efficiency80% were excluded. Data were analyzed using χ(2) test and logistic and linear regression models. These analyses were also applied to 99 adult patients with similar exclusion criteria.Adult NC measurement had inter-observer correlation of 0.85 (N = 100). Among children, after correcting for BMI-Z scores, NHR conferred additional predictive value, in both logistic regression and linear models, for both apnea-hypopnea index (AHI)2 and5 (N = 507). In children, for NHR0.25, the odds ratio of AHI2 was 3.47. In adults, for NHR0.25, the odds ratio of AHI5 was 18.NHR can be included as a simple screening tool for OSA in children and adults, which along with other predictors, may improve the ability of clinicians to triage children and adults at risk for OSA for further evaluation with PSG.

Advanced radiological imaging has largely replaced exploratory operations and has become an essential diagnostic tool clinicians routinely rely on. However, physicians are faced with a lot of radiological findings without histological proof, and assuming a more serious diagnosis may lead to unnecessary investigations and emotional stress for patients. We report an unusual presentation of chronic appendicitis with a synchronous peritoneal nodule on CT in a 76-year-old woman who presented with poor appetite, weight loss and a mass in the right iliac fossa. The coincidental finding of the nodule in addition to the suspicious appearance of the appendix raised concerns for primary appendiceal cancer with peritoneal metastasis. The case illustrates the patient's management and reflects on the learnt lessons with regard to careful use of invasive radiology-guided biopsies and interval imaging, as these could sometimes delay the diagnosis and management of a readily treatable disease.

Children with Down syndrome (DS) commonly have obstructive sleep apnea syndrome (OSAS) and may assume a unique sleeping position not systematically described previously. We describe this sleep position in DS and explore its relationship with OSAS in comparison to control children (CC).Overnight video-polysomnograms (PSG) of consecutive children with DS (age 2-18 y), referred to our center between April 2008 and October 2009, were retrospectively analyzed by a single scorer (ES). CC group comprised age and gender matched, non-syndromic, neurologically intact children referred to us for suspected OSAS over the same period.Each group had 17 subjects matched for age (median [IQR]; 6 [4-8]) and gender (65% female). DS group had higher BMI (median [IQR]; 18.8 [17.4-21.0]) than CC (17 [14.7 -18.8]; p = 0.03). There were however no significant differences (median [IQR]) between DS and CC with respect to sleep time in minutes (460 [425-499] vs 424[410-483]), sleep efficiency (%) (90.9 [87.4-92.4] vs 88.6 [79.9-93.1]), REM time (%) (17.1 [14.2-22.1] vs 19.2 [14.9-22.1]), supine time (%) (40.7 [24.8-56.0] vs 15.8 [0.40-44.5], p 0.06), mean oxygen saturation (%) (95 [94-96] vs 96 [95-97]), oxygen saturation nadir (89 [86-91] vs 89[94-92]), or total apnea-hypopnea index (4.3 [3-7.8] vs 5.1[1.9-9.6]). Despite these similarities between the groups, 9 (53%) DS children slept seated bent forward with head resting on bed for at least part of the total sleep time (%) (7.8 ± 10.9, range 0.8-35.7).This was absent in the CC group (p = 0.06).Some DS children assume a peculiar body position, sitting cross-legged flopped-forward with head resting on bed while asleep. This is absent in age- and gender-matched controls showing otherwise similar PSG characteristics. The reason for this posture is unclear from this study. However, this novel report of a unique sleeping position would provide us with a basis to conduct a prospective study involving a larger population to ascertain the contribution of this position for OSAS protection or to determine if it may be forme fruste parasomnia.

This study was designed to determine the need for water supplementation to maintain water homoeostasis in exclusively breastfed infants during summer in a tropical country. A prestudy questionnaire revealed that 97% of 34 nurses and 63% of 70 doctors advocated such supplementation. 45 healthy, male, exclusively breastfed babies, aged 1-4 months, were recruited from a well-baby clinic. 9 who had never received supplemental water plus a random selection of 14 others were allocated to group I (breastmilk only); the remaining 22 infants were allocated to group II (breastmilk plus supplemental fluid according to the mother's usual practice). The babies were studied at the hospital for 8 h; breastmilk intake was measured by weighing the infant before and after each feed, water intake by calibrated bottles, and urine output by accurate collection and measurement. The maximum room temperatures were 34-41 degrees C and relative humidities 9-60% (below 50% in all but 3 infants). In group II the mean water intake was 11% (95% confidence interval 7-16%) of the total fluid intake. Both breastmilk intake (274 vs 210 ml) and total fluid intake (274 vs 233 ml) were higher in group I than in group II (p = 0.003, p = 0.073, respectively), after adjustment for age, weight, length, room temperature, and humidity. However, there were no significant differences between the groups in urine output, urine or serum osmolality, weight change, or rectal temperature whether or not the factors adjusted for included total fluid intake. Thus, exclusively breastfed infants do not need supplemental water to maintain water homoeostasis; a reduced breastmilk intake is a potential disadvantage of this practice.

Oxidant stress-related mechanisms have been proposed as a major contributor to the increased prevalence of cardiovascular morbidity in adult patients with sleep-disordered breathing. Isoprostanes provide a reliable biomarker of oxidant injury in vivo. The purpose of the present study was to examine the hypothesis that oxidant stress, as evidenced by increased levels of F2-isoprostane metabolites (IsoP-m) in urine, is present in children with a spectrum of sleep-disordered breathing. Assays were performed on urinary samples obtained from each of 47 pediatric patients immediately upon awakening after standard overnight polysomnography. Of the subjects, 15% had mild, 9% had moderate, and 6% had severe sleep-disordered breathing. After controlling for correlations between BMI and IsoP-m and SpO2 values, IsoP-m values were unrelated to any polysomnographic measures. The absence of increased levels of urinary F2-isoprostane metabolites in children with sleep-disordered breathing suggests that oxidative stress is not a significant feature of pediatric sleep-disordered breathing.

Upper airway inflammation is now recognized in adults with obstructive sleep apnea (OSA) syndrome. However, the role played by eicosanoids such as leukotrienes and prostaglandins is unclear.To investigate whether eicosanoids are measurable in exhaled breath condensate (EBC), and to determine whether differences in these inflammatory mediators emerge among children with and without sleep-disordered breathing (SDB).EBC was collected from 50 consecutive snoring children undergoing overnight polysomnography for suspected SDB, and from 12 nonsnoring control subjects. Prostaglandin E2 (PGE2), leukotriene B4 (LTB4), and cysteinyl leukotrienes (cys-LTs: leukotriene C4 [LTC4]/leukotriene D4 [LTD4]/leukotriene E4 [LTE4]) EBC levels were analyzed using enzyme-linked immunosorbent assay.LTB4 levels were elevated in children with an apnea-hypopnea index (AHI)5/h (SDB; 97.6 +/- 6.3 pg/mL) compared to children with an AHI5/h (mild SDB; 66.4 +/- 19.1 pg/mL; p0.01) and control subjects (27.8 +/- 3.7 pg/mL; p0.01). Similarly, cys-LT (LTC4/LTD4/LTE4) concentrations were also increased in SDB (45.1 +/- 10.6 pg/mL in SDB vs 27.6 +/- 8.3 pg/mL in mild SDB, and 15.7 +/- 7.6 pg/mL in control subjects; p0.01). In contrast, PGE2 concentrations were similar among the three groups.Inflammatory mediators such as leukotrienes and prostaglandins can be readily quantified in EBC collected from the upper airway of children. Disease severity-dependent increases in leukotriene concentrations (LTB4 and LTC4/LTD4/LTE4) emerge among children and may serve as a noninvasive tool in the clinical assessment of these children.

To investigate the relative contribution of various risk factors to the surgical outcome of adenotonsillectomy for obstructive sleep apnea syndrome in children.Children (n = 110; mean age, 6.4 +/- 3.9 years) underwent two polysomnographic evaluations before and after adenotonsillectomy. In addition, 22 control children were studied. History for allergy and family history of sleep-disordered breathing was taken before each polysomnographic evaluation.Significant changes in sleep stage percentages and sleep fragmentation were found in the postsurgery study compared with the presurgery study; 25% of the children had apnea/hypopnea index (AHI)/=1, 46% had AHI1 and5, and 29% had AHI/=5 in the postsurgery study. The frequency of subjects with AHI/=1 after surgery was significantly lower among obese subjects (P.05). Comparison between the children who had AHI/=1 after surgery and 22 control children showed complete normalization of sleep architecture after surgery.Adenotonsillectomy yields improvements in respiratory abnormalities in children with obstructive sleep apnea syndrome. Complete normalization occurs in only 25% of the patients. Obesity and AHI at diagnosis are the major determinant for surgical outcome. When normalization of respiratory measures occurs after surgery, normalization of sleep architecture will also ensue.

We present a case of a Caucasian multigravida with advanced maternal age who showed evidence of an unusual heterochromatin banding pattern on chromosome 9 and a similar fetal karyotype on amniocentesis. Although unusual banding patterns of this region have been described earlier, we report a new, clinically insignificant pattern. This case illustrates the care needed in analysing the heterochromatin region for accurate clinical interpretation of chromosome 9 polymorphisms.

Background Obstructive sleep apnea (OSA) is the most common form of sleep-disordered breathing, with almost 15 million Americans affected and many more at risk. Current diagnostic approach to OSA requires polysomnography, which is laborious, onerous, and time-consuming. There is ample evidence that inflammatory responses to the perturbations associated with OSA trigger a variety of genes and signaling cascades that ultimately lead to end-organ injury and changes in kidney function and protein expression. The aim of this study was therefore to analyze proteins in human urine and assess whether differential expression of particular proteins is associated with the presence of OSA. Methods Eleven OSA and 11 control children between the ages of three and 14 (males=17; females=5) underwent overnight sleep studies followed by a first-morning urine sample. Proteomic analysis of urine samples yielded a unique map of proteins, of which, five spots were selected for further analysis due to their significant intensity differences between OSA and control groups. Results Mass spectrometry followed by peptide mass fingerprinting conclusively identified four of the five spots as gelsolin, perlecan (a heparan sulfate proteoglycan), albumin, and immunoglobulin ( P 67). Conclusions Overall, increased expression of gelsolin and perlecan in the urinary proteome of children with OSA suggests that the episodic hypoxia associated with OSA may lead to changes in protein permeability or alternatively to increased catabolism of these proteins and their excretion in urine.

Denovo deletions of 18p without other associated rearrangement are uncommon. For such a deletion to profoundly affect the fetus of a near normal phenotypic carrier would be rarer. We present such a case in which the chance of a cryptic rearrangement was ruled out by fluorescence in situ hybridization (FISH) analysis. Possible explanations for wide variations in clinical expression are discussed.

To identify the prevalence of clinically relevant findings during cardiac evaluations of pediatric patients with obstructive sleep apnea (OSA) undergoing adenotonsillectomy (TA), and to determine the association between cardiac findings and postoperative respiratory complications.Retrospective medical chart review.Pediatric patients aged 10 months to 15 years who underwent both echocardiography and polysomnography (PSG) within 6 months prior to TA for OSA from April 2007 through April 2011.Two pediatric cardiologists independently reviewed echocardiographic studies for evidence of cardiovascular disease. Patients were stratified based on apnea-hypopnea index (AHI) severity (1-5,5-10, and10). These groups were compared according to demographic, electrocardiographic (ECG), and echocardiographic values, and postoperative respiratory complications.The medical charts of 57 of 900 patients identified were reviewed following exclusion of those with congenital cardiac abnormalities. The AHI groupings did not differ demographically. No clinically relevant abnormalities were identified on the echocardiogram of any patient. There was a statistically significant association between increased AHI and the appearance of postoperative respiratory complications (P .05). Indicators of myocardial hypertrophy, such as left ventricular mass index, were not significantly related to AHI in contrast to previously published studies. No echocardiographic or ECG findings were identified that were associated with increased number of postoperative respiratory complications or OSA severity based on AHI.The lack of clinically relevant findings during preoperative cardiac evaluations suggests that aggressive cardiac workup in pediatric patients with OSA may not be indicated unless dictated by comorbidities. Consistent with results in prior studies, preoperative AHI can identify patients at risk for respiratory complications following TA.