[Show abstract][Hide abstract]ABSTRACT: Objective:
To pilot a clinician-based outcome measure that provides complementary information to objective measures and parent-based questionnaires for insomnia in children with autism spectrum disorders (ASD).
The authors developed a Pediatric Sleep Clinical Global Impressions Scale (CGI). Questions included (1) the child's ability to fall asleep and remain sleeping independently (i.e., apart from parents); (2) bedtime resistance; (3) sleep onset delay; (4) night awakening; (5) parental satisfaction with their child's current sleep patterns; (6) family functioning as affected by their child's current sleep patterns; and (7) clinician's overall concern with the child's sleep. After refining the instrument through the evaluation of vignettes by ASD and sleep experts, the authors piloted the Pediatric Sleep CGI in a 12-week randomized trial of iron supplementation in children with ASD. Clinicians completed Pediatric Sleep CGIs and structured sleep histories, parents completed the Children's Sleep Habits Questionnaire (CSHQ), and children wore actigraphy watches.
In repeated measures models, the Pediatric Sleep CGI and CSHQ were correlated for sleep onset delay (r = .66, p < .001), night wakings (r = .40, p < .001), and total score (r = .29, p < .001). The CGI-S sleep onset delay and actigraphy sleep onset delay scores (r = .75, p = .0095) were also correlated. The overall CGI-S showed improvement with therapy (p = .047).
The Pediatric Sleep CGI shows promise in measuring clinician-rated outcomes in pediatric insomnia in children with ASD. Larger samples will be necessary to examine reliability, validity, and measure to change, as well as applicability to other populations with pediatric insomnia.
Article · Jun 2016 · Journal of Developmental & Behavioral Pediatrics
[Show abstract][Hide abstract]ABSTRACT: Key Clinical Message
Patients presenting with infantile spasms, dysmorphic features, and periventricular nodular heterotopia may benefit from genetic copy number variation microarray, or whole‐exome sequencing to identify candidate genes. This will allow personalized diagnosis and prognostication and the eventual understanding of single and combined gene functions in brain health and disease.
[Show abstract][Hide abstract]ABSTRACT: The objective of this systematic review was to examine the relationships between objectively and subjectively measured sleep duration and various health indicators in children and youth aged 5–17 years. Online databases were searched in January 2015 with no date or study design limits. Included studies were peer-reviewed and met the a priori-determined population (apparently healthy children and youth aged 5–17 years), intervention/exposure/comparator (various sleep durations), and outcome (adiposity, emotional regulation, cognition/academic achievement, quality of life/well-being, harms/injuries, and cardiometabolic biomarkers) criteria. Because of high levels of heterogeneity across studies, narrative syntheses were employed. A total of 141 articles (110 unique samples), including 592 215 unique participants from 40 different countries, met inclusion criteria. Overall, longer sleep duration was associated with lower adiposity indicators, better emotional regulation, better academic achievement, and better quality of life/well-being. The evidence was mixed and/or limited for the association between sleep duration and cognition, harms/injuries, and cardiometabolic biomarkers. The quality of evidence ranged from very low to high across study designs and health indicators. In conclusion, we confirmed previous investigations showing that shorter sleep duration is associated with adverse physical and mental health outcomes. However, the available evidence relies heavily on cross-sectional studies using self-reported sleep. To better inform contemporary sleep recommendations, there is a need for sleep restriction/extension interventions that examine the changes in different outcome measures against various amounts of objectively measured sleep to have a better sense of dose–response relationships.
Article · Jun 2016 · Applied Physiology Nutrition and Metabolism
[Show abstract][Hide abstract]ABSTRACT: Leaders from the Canadian Society for Exercise Physiology convened representatives of national organizations, content experts, methodologists, stakeholders, and end-users who followed rigorous and transparent guideline development procedures to create the Canadian 24-Hour Movement Guidelines for Children and Youth: An Integration of Physical Activity, Sedentary Behaviour, and Sleep. These novel guidelines for children and youth aged 5-17 years respect the natural and intuitive integration of movement behaviours across the whole day (24-h period). The development process was guided by the Appraisal of Guidelines for Research Evaluation (AGREE) II instrument and systematic reviews of evidence informing the guidelines were assessed using the Grading of Recommendations Assessment, Development, and Evaluation (GRADE) approach. Four systematic reviews (physical activity, sedentary behaviour, sleep, integrated behaviours) examining the relationships between and among movement behaviours and several health indicators were completed and interpreted by expert consensus. Complementary compositional analyses were performed using Canadian Health Measures Survey data to examine the relationships between movement behaviours and health indicators. A stakeholder survey was employed (n = 590) and 28 focus groups/stakeholder interviews (n = 104) were completed to gather feedback on draft guidelines. Following an introductory preamble, the guidelines provide evidence-informed recommendations for a healthy day (24 h), comprising a combination of sleep, sedentary behaviours, light-, moderate-, and vigorous-intensity physical activity. Proactive dissemination, promotion, implementation, and evaluation plans have been prepared in an effort to optimize uptake and activation of the new guidelines. Future research should consider the integrated relationships among movement behaviours, and similar integrated guidelines for other age groups should be developed.
Full-text available · Article · Jun 2016 · Applied Physiology Nutrition and Metabolism
[Show abstract][Hide abstract]ABSTRACT: Objective Better Nights/Better Days, a distance intervention for insomnia in school-aged children (with and without attention-deficit/hyperactivity disorder
[ADHD]), was evaluated to determine its effectiveness on children’s sleep and psychosocial functioning. Methods A single center, parallel group design randomized controlled trial (stratified on ADHD diagnosis) was conducted. Parents
were randomized to intervention (n = 31) or waitlist control (n = 30), and completed questionnaires administered over the phone at baseline, postintervention (2 months), and follow-up (6
months). Actigraphy was also collected. The intervention consisted of a five-session manual and weekly telephone coach support. Results The intervention group demonstrated a significant reduction in sleep problems and improved psychosocial functioning at postintervention
and follow-up. Actigraphy results indicated improved sleep onset, but not sleep duration. Children with and without ADHD responded
in a similar manner to this intervention. Parents provided high satisfaction ratings. Conclusions This intervention holds promise as an accessible, sustainable, and effective program to address insomnia in school-aged children.
Article · May 2016 · Journal of Pediatric Psychology
[Show abstract][Hide abstract]ABSTRACT: Health Canada, in collaboration with Statistics Canada, and other external experts, conducted the Community Noise and Health Study to better understand the impacts of wind turbinenoise (WTN) on health and well-being. A cross-sectional epidemiological study was carried out between May and September 2013 in southwestern Ontario and Prince Edward Island on 1238 randomly selected participants (606 males, 632 females) aged 18–79 years, living between 0.25 and 11.22 km from operational wind turbines. Calculated outdoor WTN levels at the dwelling reached 46 dBA. Response rate was 78.9% and did not significantly differ across sample strata. Self-reported health effects (e.g., migraines, tinnitus, dizziness, etc.), sleep disturbance, sleep disorders, quality of life, and perceived stress were not related to WTN levels. Visual and auditory perception of wind turbines as reported by respondents increased significantly with increasing WTN levels as did high annoyance toward several wind turbine features, including the following: noise, blinking lights, shadow flicker, visual impacts, and vibrations. Concern for physical safety and closing bedroom windows to reduce WTN during sleep also increased with increasing WTN levels. Other sample characteristics are discussed in relation to WTN levels. Beyond annoyance, results do not support an association between exposure to WTN up to 46 dBA and the evaluated health-related endpoints.
Full-text available · Article · Mar 2016 · The Journal of the Acoustical Society of America
[Show abstract][Hide abstract]ABSTRACT: Background:
Little knowledge exists on the availability of academic and community paediatric neurology positions. This knowledge is crucial for making workforce decisions. Our study aimed to: 1) obtain information regarding the availability of positions for paediatric neurologists in academic centres; 2) survey paediatric neurology trainees regarding their perceptions of employment issues and career plans; 3) survey practicing community paediatric neurologists 4) convene a group of paediatric neurologists to develop consensus regarding how to address these workforce issues.
Surveys addressing workforce issues regarding paediatric neurology in Canada were sent to: 1) all paediatric neurology program directors in Canada (n=9) who then solicited information from division heads and from paediatric neurologists in surrounding areas; 2) paediatric neurology trainees in Canada (n=57) and; 3) community paediatric neurologists (n=27). A meeting was held with relevant stakeholders to develop a consensus on how to approach employment issues.
The response rate was 100% from program directors, 57.9% from residents and 44% from community paediatric neurologists. We found that the number of projected positions in academic paediatric neurology is fewer than the number of paediatric neurologists that are being trained over the next five to ten years, despite a clinical need for paediatric neurologists. Paediatric neurology residents are concerned about job availability and desire more career counselling.
There is a current and projected clinical demand for paediatric neurologists despite a lack of academic positions. Training programs should focus on community neurology as a viable career option.
Full-text available · Article · Jan 2016 · The Canadian journal of neurological sciences. Le journal canadien des sciences neurologiques
[Show abstract][Hide abstract]ABSTRACT: OBJECT Hemispherectomy for unilateral, medically refractory epilepsy is associated with excellent long-term seizure control. However, for patients with recurrent seizures following disconnection, workup and investigation can be challenging, and surgical options may be limited. Few studies have examined the role of repeat hemispherotomy in these patients. The authors hypothesized that residual fiber connections between the hemispheres could be the underlying cause of recurrent epilepsy in these patients. Diffusion tensor imaging (DTI) was used to test this hypothesis, and to target residual connections at reoperation using neuronavigation. METHODS The authors identified 8 patients with recurrent seizures following hemispherectomy who underwent surgery between 1995 and 2012. Prolonged video electroencephalography recordings documented persistent seizures arising from the affected hemisphere. In all patients, DTI demonstrated residual white matter association fibers connecting the hemispheres. A repeat craniotomy and neuronavigation-guided targeted disconnection of these residual fibers was performed. Engel class was used to determine outcome after surgery at a minimum of 2 years of follow-up. RESULTS Two patients underwent initial hemidecortication and 6 had periinsular hemispherotomy as their first procedures at a median age of 9.7 months. Initial pathologies included hemimegalencephaly (n = 4), multilobar cortical dysplasia (n = 3), and Rasmussen's encephalitis (n = 1). The mean duration of seizure freedom for the group after the initial procedure was 32.5 months (range 6-77 months). In all patients, DTI showed limited but definite residual connections between the 2 hemispheres, primarily across the rostrum/genu of the corpus callosum. The median age at reoperation was 6.8 years (range 1.3-14 years). The average time taken for reoperation was 3 hours (range 1.8-4.3 hours), with a mean blood loss of 150 ml (range 50-250 ml). One patient required a blood transfusion. Five patients are seizure free, and the remaining 3 patients are Engel Class II, with a minimum follow-up of 24 months for the group. CONCLUSIONS Repeat hemispherotomy is an option for consideration in patients with recurrent intractable epilepsy following failed surgery for catastrophic epilepsy. In conjunction with other modalities to establish seizure onset zones, advanced MRI and DTI sequences may be of value in identifying patients with residual connectivity between the affected and unaffected hemispheres. Targeted disconnection of these residual areas of connectivity using neuronavigation may result in improved seizure outcomes, with minimal and acceptable morbidity.
Article · Dec 2015 · Journal of Neurosurgery Pediatrics
[Show abstract][Hide abstract]ABSTRACT: Study objectives:
To investigate the association between self-reported and objective measures of sleep and wind turbine noise (WTN) exposure.
The Community Noise and Health Study, a cross-sectional epidemiological study, included an in-house computer-assisted survey and sleep pattern monitoring over a 7 d period. Outdoor WTN levels were calculated following international standards for conditions that typically approximate the highest long-term average levels at each dwelling. Survey data were collected between May and September 2013 from adults, aged 18-79 y (606 males, 632 females) randomly selected from each household and living between 0.25 and 11.22 kilometers from operational wind turbines in two Canadian provinces. Self-reported sleep quality over the past 30 d was assessed using the Pittsburgh Sleep Quality Index. Additional questions assessed the prevalence of diagnosed sleep disorders and the magnitude of sleep disturbance over the previous year. Objective measures for sleep latency, sleep efficiency, total sleep time, rate of awakening bouts, and wake duration after sleep onset were recorded using the wrist worn Actiwatch2® from a subsample of 654 participants for a total of 3,772 sleep nights.
Participant response rate for the survey was 78.9%. Outdoor WTN levels reached 46 dB(A) with an arithmetic mean of 35.6 and a standard deviation of 7.4. Self-reported and objectively measured sleep outcomes consistently revealed no apparent pattern or statistically significant relationship to WTN levels. However, sleep was significantly influenced by other factors, including, but not limited to, the use of sleep medication, other health conditions (including sleep disorders), caffeine consumption, and annoyance with blinking lights on wind turbines.
Study results do not support an association between exposure to outdoor WTN up to 46 dB(A) and an increase in the prevalence of disturbed sleep. Conclusions are based on WTN levels averaged over 1 y and, in some cases, may be strengthened with an analysis that examines sleep quality in relation to WTN levels calculated during the precise sleep period time.
[Show abstract][Hide abstract]ABSTRACT: Objective
Infantile spasms (IS) are a severe form of childhood epilepsy associated with autism spectrum disorders (ASD) in up to 35% of cases. The objective of this post hoc analysis of our randomized control trial was to determine whether rapid diagnosis and treatment of IS could limit the incidence of ASD while identifying risk factors related to ASD outcome.Methods
Patients with IS were randomized in a standardized diagnostic and treatment protocol. Clinical and electroencephalogram (EEG) evaluations were completed at all eight visits over 5 years, while cognitive evaluations were administered at 0, 6, 24 and 60 months, respectively. Autism was initially screened by means of the Checklist for Autism in Toddlers (CHAT) at 24 months, and formally assessed at the 30-and 60-month follow-ups using the Autism Diagnostic Observation Schedule—Generic (ADOS-G).ResultsOf the 69 patients included in the study, 25 could not be assessed due to severe delay or death. Eleven of the 42 patients screened with CHAT, were found to be at risk of an ASD outcome. ADOS was performed in 44 and 10 were diagnosed with ASD. The CHAT proved to correlate highly with the ADOS (80% ppv). Only patients with symptomatic IS developed ASD (p = 0.003). Earlier diagnosis or successful treatment did not correlate with a reduced rate of ASD. Other risk factors were identified such as having chronic epileptic discharges in the frontotemporal areas after disappearance of hypsarrhythmia (p = 0.005 and p = 0.007) and being of nonwhite origin (p = 0.009).SignificanceASD was only observed in children with sympyomatic IS. Other clinical risk factors include chronic frontotemporal epileptic activity and being of non-white origin. Early diagnosis and treatment did not prevent ASD as an outcome of IS. However, patients at risk for ASD could be identified early on and should in the future benefit from early intervention to potentially improve their long-term outcome.
Full-text available · Article · May 2015 · Epilepsia
[Show abstract][Hide abstract]ABSTRACT: Objective
Epilepsy is a common neurologic disorder of childhood. To determine the genetic diagnostic yield in epileptic encephalopathy, we performed a retrospective cohort study in a single epilepsy genetics clinic.Methods
We included all patients with intractable epilepsy, global developmental delay, and cognitive dysfunction seen between January 2012 and June 2014 in the Epilepsy Genetics Clinic. Electronic patient charts were reviewed for clinical features, neuroimaging, biochemical investigations, and molecular genetic investigations including targeted next-generation sequencing of epileptic encephalopathy genes.ResultsGenetic causes were identified in 28% of the 110 patients: 7% had inherited metabolic disorders including pyridoxine dependent epilepsy caused by ALDH7A1 mutation, Menkes disease, pyridox(am)ine-5-phosphate oxidase deficiency, cobalamin G deficiency, methylenetetrahydrofolate reductase deficiency, glucose transporter 1 deficiency, glycine encephalopathy, and pyruvate dehydrogenase complex deficiency; 21% had other genetic causes including genetic syndromes, pathogenic copy number variants on array comparative genomic hybridization, and epileptic encephalopathy related to mutations in the SCN1A, SCN2A, SCN8A, KCNQ2, STXBP1, PCDH19, and SLC9A6 genes. Forty-five percent of patients obtained a genetic diagnosis by targeted next-generation sequencing epileptic encephalopathy panels. It is notable that 4.5% of patients had a treatable inherited metabolic disease.SignificanceTo the best of our knowledge, this is the first study to combine inherited metabolic disorders and other genetic causes of epileptic encephalopathy. Targeted next-generation sequencing panels increased the genetic diagnostic yield from <10% to >25% in patients with epileptic encephalopathy.
[Show abstract][Hide abstract]ABSTRACT: Access to treatment for pediatric insomnia is limited by a number of factors. Recently, the Internet has started to be used as a means of increasing access to evidence-based behavioral treatment. The current article describes the results of 2 studies focused on evaluating potential factors that could influence parents’ use of an Internet intervention for pediatric insomnia. In Study 1, health professionals were asked to report on their perceptions of potential barriers and facilitators to their use of an Internet intervention for pediatric insomnia with their patients. Study 2 was a usability study in which participants (parents and health professionals) were asked to evaluate the usability of a beta version of an Internet intervention for pediatric insomnia (Better Nights, Better Days) that is currently under development. Results from both studies suggest that factors related to the website, as well as factors external to the website, may impact parents’ use of an Internet intervention for pediatric insomnia, as well as perceptions of usability and satisfaction. The results of the current research have implications for the development of both the intervention under investigation and of future e-health interventions.
[Show abstract][Hide abstract]ABSTRACT: Background
The relationship between epilepsy and sleep is bidirectional as seizures disrupt sleep and coexisting sleep disorders have detrimental effects on seizure control and quality of life for both the children and their families. Previous research has reported on sleep disturbance in children with epilepsy primarily by subjective parental reports. Actigraphy may provide a more accurate objective evaluation of sleep, but the validity of this sleep measure for children with epilepsy has not yet been assessed. The primary objective of this study was to validate the use of actigraphy as a tool in studying sleep patterns in children with epilepsy.
This was a prospective study comparing sleep and wake epochs recorded for 24 h simultaneously by actigraphy and by continuous video-electroencephalography (VEEG) monitoring in 27 patients aged 2–18 years with medically refractory epilepsy.
Strong correlations were found between actigraphy and VEEG sleep variables including night sleep period (r = 0.99), night sleep time (r = 0.96), duration of night wake time (r = 0.93) and number of significant wakings during the night (r = 0.81).
The study results validate that actigraphy is a reliable and objective clinical and research tool for evaluating sleep and wakefulness in children with epilepsy.
[Show abstract][Hide abstract]ABSTRACT: Background
Sleep problems are common among children with chronic illnesses such as Juvenile Idiopathic Arthritis (or JIA). However, little is known about the frequency and severity of sleep disturbance(s) and the factors that are associated with sleep problems in children with JIA. The mechanism(s) of the relationships characterizing the development or exacerbation of sleep problems in children with JIA are still unknown, however studies have reported an association. The purpose of this study was to synthesize existing research related to sleep problems in children with JIA.
The Preferred Reporting Items for Systematic Reviews and Meta-analysis (PRISMA) statement guided the conduct and reporting of this review. An experienced librarian conducted searches in MEDLINE, EMBASE, PsychINFO, CINAHL, and the Cochrane Central Register of Controlled Trials from inception to January 2012, to identify potentially relevant citations. Two members independently selected, rated methodological quality using the QUIPS tool, and extracted data from included studies.
Ten studies were included and findings varied across studies; studies were mostly cross-sectional, or case-controlled designs, with only one cohort study available. Four studies found that children and adolescents diagnosed with JIA had significantly more sleep disturbances when compared to healthy controls. Pain was most often associated with sleep disturbances. The heterogeneous findings highlight the complex relationships between JIA and sleep, and low methodological quality of studies in the field.
This review supports an association between poor sleep and increased symptoms related to JIA, specifically the experience of pain. However, results need to be interpreted cautiously given the inconsistent findings regarding factors associated with sleep problems in JIA, the limited evidence available, and its low quality. Furthermore it is not yet determined if the poor sleep patterns predate the symptoms reported with JIA. More research is vital to understanding the factors that predict or perpetuate poor sleep in children and adolescents diagnosed with JIA.
Full-text available · Article · Jun 2014 · Pediatric Rheumatology
[Show abstract][Hide abstract]ABSTRACT: Sleep problems represent a real and troubling aspect of the lives of many children with NDD. As with TD children, the most common sleep problem among children with NDD is behavioral insomnia. Prevalence rates of sleep problemsin general range between 50% and 95% for children with NDD. Given that poor sleep in children with NDD has been associated with impairments in daytime functioning, decreased quality of life for the children, increased NDD symptoms and morbidity, and negative effects on caregivers health and parenting abilities, these high rates are concerning and underscore the need for appropriate screening, diagnostic evaluation, and management. Behavioral interventions have considerable empiric support and should be recommended as first-line treatment of sleep problems in this population. Additional research is needed to establish the effectiveness of specific behavioral strategies, including psychoeducation, healthy sleep practices, and techniques, such as faded bedtime with response cost (FBRC) or extinction, for treating behavioral insomnia in children with NDD.
[Show abstract][Hide abstract]ABSTRACT: The literature has been highly informative for when to use actigraphy and its validity in pediatric research. However, minimal literature exists on how to perform actigraphy, especially in special populations. We determined whether providing actigraphy training to parents and coordinators increased the nights of actigraphy data that could be scored. We compared two studies in children with autism spectrum disorders, one of which provided a basic level of training in a single-site trial and the other of which provided more detailed training to parents and coordinators in a multisite trial. There was an increase in scorable nights in the multisite trial containing a one-hour structured parent training session. Our results support the use of educational tools in clinical trials that use actigraphy.
Full-text available · Article · Mar 2014 · Behavioral Sleep Medicine
[Show abstract][Hide abstract]ABSTRACT: Children with epilepsy have frequent sleep disturbance and challenges in learning and memory. There is little research on the consolidation of memory during sleep in this population. The goal of this pilot study was to determine whether children with epilepsy are able to consolidate memories better after a sleep versus wake period as has been demonstrated in typically developing children.
This study was a prospective evaluation of children with epilepsy to determine if sleep improved episodic memory (using word lists) as compared with memory following a wake period of similar duration. The study was conducted in patients in the Epilepsy Monitoring Unit at a single academic health science center. In the sleep recall condition, the learning trials were presented in the evening, and delayed recall of the words was tested in the morning. In the wake condition, the learning took place in the morning, and the delayed recall took place later in the day. Subjects wore an actigraph to evaluate sleep/wake patterns. Data regarding the children's epilepsy, antiepileptic medications, and frequency of interictal epileptiform discharges were also documented.
Ten children (agd 8-17years) participated in the study. For the entire sample, recall after sleep was better than recall after awake (p=0.03), and 7 of the 10 children showed this effect. However, reanalyses removing an outlier showed no difference between the two recall conditions. The mean number of interictal epileptiform discharges was 8.8 during the recall after sleep and 7.8 during the recall after awake. Three children had seizures during the evaluation.
In this pilot study, we demonstrated that a small cohort of children with epilepsy, with similar interictal epileptiform discharges during sleep and wake, showed no advantage in memory for a word list after a period of sleep than after a period of being awake. This finding requires further study in a larger cohort. Poor memory consolidation during sleep may contribute to the cognitive deficits in children with epilepsy.