The Type, Severity, and Impact of Sleep Problems in Children With Angelman Syndrome and Parental Help-seeking Patterns.

Angelman syndrome (AS) is a rare genetic developmental disability that presents with high rates of co-occurring sleep difficulties. Most existing research has focused on the pathophysiology of sleep problems in people with AS, and suggests that sleep problems are the result of genetic and neurobiological factors. However, little is known about the role of the social environment and learning in sleep problems in children with AS. This descriptive study used survey data from 139 parents of children with AS to investigate: 1) the type, topography and severity of children's sleep problems; 2) the collateral child, parent and family impacts of the sleep problems; 3) treatment selection practices and the perceived effectiveness of these treatments; and 4) sources of support and treatment advice received. Parents reported that the majority of children experienced sleep problems, resulting in numerous deleterious effects on child and family functioning. They also reported high levels of concern about these sleep problems, but low levels of perceived support. Study findings highlight the need to establish a disability-specific profile of the type and impact of sleep problems experienced by children with AS, and have further implications for the delivery of clinical services and support provided to parents of children with AS.

Sequential Implementation of Functional Behavior Assessment-Informed Treatment Components for Sleep Disturbance in Autism: A Case Study.

Background: Sleep disturbances are a significant problem for people with autism spectrum disorder (ASD). Existing research supports the use of parent-implemented, functional behavior assessment (FBA)-informed interventions for sleep problems in children with ASD. There is also emerging evidence for combined parent- and young person-implemented behavioral sleep interventions for older children and adolescents with ASD. However, the active treatment components of such interventions have not been identified in previous studies, as components have not been evaluated independently of one another.Methods: The current study sequentially implemented FBA-informed treatment components (in the order of least to most restrictive and time intensive) within a single-case AB design, to evaluate at which point treatment resulted in a statistically and clinically substantive reduction in target sleep variables. Combined parent- and young person-implemented intervention components consisted of: (a) white noise; (b) white noise and relaxation instruction; and (c) white noise, relaxation instruction, and stimulus control.Participant: The participant was a 9-year-old girl with autism and selective mutism.Results: The combined use of white noise, relaxation instruction, and stimulus control resolved the participant's sleep problems. Other more restrictive and/or time intensive interventions were unnecessary. Treatment effects were maintained at 10-week follow-up.Conclusions: The current study illustrates the feasibility of administering FBA-informed treatment components sequentially, to ensure application of minimally sufficient interventions.

Novel Mineral-Vitamin Treatment for Reduction in Cigarette Smoking: A Fully Blinded Randomized Placebo-Controlled Trial.

INTRODUCTION: Many smokers do not achieve abstinence using current smoking cessation options. This randomized controlled trial (RCT) investigated a novel nutritional supplement to assist with quitting smoking. METHODS: Following a baseline phase where cigarettes per day and nicotine dependence were measured, participants (n = 107) were randomized to placebo (n = 50) or micronutrient conditions (n = 57). A 4-week pre-quit phase permitted titration up to 12 capsules/day. During the quit phase (12 weeks), participants were registered with a public Quitline while consuming micronutrients or placebo. Carbon monoxide levels were measured to confirm smoking cessation. RESULTS: Forty-five (42%) participants completed the trial. Treatment and placebo groups did not differ on the primary outcome of continuous abstinence at 12 weeks using intention-to-treat analysis; however, 28% of the micronutrient-treated group had quit versus 18% for placebo (odds ratio [OR] = 1.78, 95% confidence interval [CI] = 0.71 to 4.48), with number needed to treat = 10. Comparison of cigarette consumption (cigarettes per day) between micronutrient and placebo groups showed that those taking micronutrients reported reduced consumption throughout the trial, notably at pre-quit weeks 1 and 4, and at quit phase week 4. There were no serious adverse events, blinding was successful, and there were no substantive group differences in side effects or dropout rate. CONCLUSION: This is the first RCT investigating the impact of micronutrients on smoking reduction, finding that micronutrients reduced harm through reduction in number of cigarettes smoked relative to placebo. The small sample and high dropout rate limit confidence in the conclusions and generalizability of the study; however, assessed by number needed to treat, micronutrients are comparable to other smoking cessation treatments but with fewer side effects. Future research using larger and longer trials including cost-effectiveness and biomarker measures is encouraged. IMPLICATIONS: Micronutrients are being increasingly studied for the treatment of psychiatric conditions, but direct application of micronutrients as a treatment for addictions is novel. There is extensive evidence that micronutrients alleviate stress. Given that tobacco smoking is often used to cope with stress, taking micronutrients may moderate the stress of withdrawal and increase the chance of a successful quit attempt. This study is the first known RCT to investigate the use of micronutrients to support smoking cessation. Treatments that are safe, effective, relatively inexpensive, and readily available are needed and micronutrient supplements offer one such possible alternative.

The Lighter Touch: Less-Restriction in Sequentially Implemented Behavioral Sleep Interventions for Children with Rare Genetic Neurodevelopmental Conditions.

PURPOSE: The prevalence of sleep difficulties among children with rare genetic neurodevelopmental conditions (RGNC) is high. Behavioral interventions are commonly used in the treatment of sleep difficulties in children with neurodevelopmental conditions such as autism, however, research is scarce in children with RGNC. The range of co-occurring complexities within this population, means there is a need for research to not only determine the effectiveness of behavioral sleep interventions, but also which components might be the least restrictive (i.e., intensive/aversive) and minimally sufficient. METHODS: This study used a single-case multiple baseline design to investigate the effectiveness and acceptability of behavioral sleep interventions, indicated within a Functional Behavior formulation in eight children with RGNC (M = 7.3 years). Intervention components were sequentially administered across up to three phases, based on the principle of less restriction (from least to relatively more intensive) to determine what might be minimally sufficient. RESULTS: Results showed an improvement in sleep onset latency, night wakings, early morning waking and unwanted bed-sharing for 7/7, 6/7, 3/3 and 3/3 children respectively. Improvement was observed for most participants following the less restrictive phases of intervention (circadian modifications, antecedent modifications and positive reinforcement), however, more restrictive, albeit modified, extinction procedures were still implemented for five participants. Improvements were maintained at follow-up and interventions were deemed acceptable to parents. CONCLUSIONS: Less restrictive function-based behavioral strategies are an effective, and in some cases sufficient, contribution to a sequence of interventions for a range of sleep difficulties. They should be implemented first, before more restrictive strategies.

Collateral Child and Parent Effects of Function­Based Behavioral Interventions for Sleep Problems in Children and Adolescents with Autism.

This study follows McLay et al., Journal of Autism and Developmental Disorders, (2020) to investigate whether the function-based behavioral sleep interventions received by 41 children and adolescents with autism spectrum disorder (ASD) produced collateral improvements in ASD severity, internalizing and externalizing symptoms and parent relationship quality, ratings of depression, anxiety and stress, and personal sleep quality. Concomitant with reduced sleep problem severity, improvements were found in children's internalizing and externalizing behavior and ASD symptom severity. Small improvements were also found in maternal sleep quality and parental stress. There was little change in parental relationship quality post-treatment, possibly reflecting high baseline scores. Overall, collateral benefits were generally small but positive, consistent with the limited extant research, and underscore the importance of investigating collateral effects across a range of variables.

Sleep and stereotypy in children with autism: effectiveness of function-based behavioral treatment.

OBJECTIVES: Children with autism spectrum disorder (ASD) experience high rates of sleep problems, which exacerbate the core symptoms of ASD, including stereotypy (restricted and repetitive behaviors). Conversely, stereotypy can interfere with sleep by actively competing with sleep-facilitative behaviors (eg, lying down quietly). Behavioral interventions informed by functional behavioral assessment (FBA) significantly reduce sleep problems in children with ASD, however, their impact on sleep-interfering stereotypy is not clear. This study investigated the effectiveness of function-based behavioral treatments for sleep problems, including sleep-interfering stereotypy, in children with ASD, the maintenance of these effects, and parents' satisfaction with the treatment process. METHODS: A non-concurrent multiple baselines across participants design was used to evaluate the effectiveness of function-based, individualized treatments for sleep problems and sleep-interfering stereotypy in three children with ASD. For each participant, stereotypy was automatically maintained and interfered with the initiation and/or re-initiation of sleep. Parents implemented multi-component treatments that included a faded bedtime procedure. RESULTS: Treatment reduced sleep problems in 2/3 participants, and the duration of stereotypy was reduced in all participants. Treatment effects were largely maintained at follow-up, and parent-reported satisfaction was high. CONCLUSION: These results support prior research demonstrating the effectiveness of FBA-informed behavioral treatments for sleep problems in children with ASD. Further, this study shows that these treatments may be effective in reducing sleep-interfering stereotypy. Future research should more thoroughly investigate the bidirectional relationships between sleep and core symptoms of ASD, and address how these relationships are assessed and treated in the sleep context.

Endogenous melatonin and sleep in individuals with Rare Genetic Neurodevelopmental Disorders (RGND): A systematic review.

Individuals with Rare Genetic Neurodevelopmental Disorders (RGND) present with significant sleep problems and circadian rhythm abnormalities of uncertain aetiology. Abnormal melatonin secretion may play a role in sleep disturbance in individuals with higher incidence developmental disabilities, however, RGND research is limited. This review compared the melatonin profiles in a range of RGND with that of the general population and considered the impact of any differences on sleep. A systematic search identified 19 studies that met inclusion criteria. Each study was examined to extract data relating to the study design, participant characteristics, objectives, sleep measures and results, and melatonin measures and findings. Studies were evaluated using the BIOCROSS quality appraisal tool. Nine studies focussed on Smith-Magenis syndrome (SMS), the rest included individuals with Angelman (AS), Fragile-X (FXS), Prader-Willi (PWS), septo-optic dysplasia, PAX6/WAGR and Williams (WS) syndromes (N = 349). Individuals with RGND present with a range of sleep problems, particularly dyssomnias. The melatonin profile varied within and between RGND, with low nocturnal melatonin levels commonly reported. Understanding the relationship between specific sleep and melatonin parameters within RGND may help inform sleep intervention.

Motivational interviewing training: a pilot study of the effects on practitioner and patient behaviour.

BACKGROUND: While Motivational Interviewing (MI) is effective in reducing client problem behaviours, including health-related behaviours, there is little evidence about how MI training enhances practitioner skills. AIMS: The current pilot study addressed this lack by training two health practitioners (Diabetes Nurse Educators) in MI, and evaluated the effect of MI training on both practitioner and patient behaviour when MI was delivered in a clinical settting, with patients experiencing difficulties with diabetes self-management. METHODS: Comparisons were made between the practitioners' skills in a baseline condition (Patient Education; PE) and after training in Motivational Enhancement Therapy (MET), a four-session form of MI. At the same time, the effects of the two interventions on patient in-session behaviour were compared. Practitioner and patient data were obtained from transcripts of all PE and MET sessions, which were independently coded using Motivational Interviewing Skills Code therapist and client behaviour counts. RESULTS: Compared with their baseline performance, practitioners, when trained to practice MET, behaved in ways consistent with MI, and this appears to have evoked beneficial in-session behaviour from the patients. CONCLUSIONS: These results suggest that the MI training was effective.

Treatment of emotional dysregulation in full syndrome and subthreshold binge eating disorder.

The link between negative affect and binge eating in those with binge eating disorder (BED) has been well established. The present study examined the efficacy of a treatment for BED designed to increase recognition and regulation of negative emotion, replicating and extending a previous investigation (Clyne, C., & Blampied, N.M. [2004]. Training in emotion regulation as a treatment for binge eating: A preliminary study. Behaviour Change, 21, 269-281) by including a control group, a larger number of participants, and formal diagnoses rather than classifying binge eating symptomatology from self-report questionnaires. Twenty-three women diagnosed with subthreshold or full syndrome BED (using the Eating Disorders Examination) participated in a treatment program that focused on increasing emotional regulation skills. Each participant completed the Eating Disorders Examination Questionnaire, the Binge Eating Scale, the Emotional Eating Scale, and completed self-monitoring records of binge episodes. Binge abstinence rates following treatment (post-treatment and 1 year follow-up were 78% and 87% respectively) were comparable to other empirically supported treatments for BED. Other positive changes in eating and general pathology were observed. These effects were well-maintained up to 1 year later.

Longitudinal Study of Infant Sleep Development: Early Predictors of Sleep Regulation Across the First Year.

BACKGROUND: An important developmental task for infants over their first few years of life is to learn to settle to sleep with a reasonably short latency, maintain sleep through the night and coordinate with family sleeping and waking schedules. A child who can reliably do this is exhibiting self-regulated sleep. Otherwise, children's sleep may have to be other (non-self) regulated to some degree and they may exhibit pediatric sleep disturbances (e.g., extended sleep latency, and/or frequent nightwaking); these are reported by 36-45% of parents of infants between ages four to 12 months. PURPOSE: To answer the question: Can infant and parent factors observed at 1 month of infant age predict which infants will have regulated sleep at 6- and 12-months of age? Prediction from 1 month has not previously been investigated. METHODS: In a prospective longitudinal study, the mothers of 52 typically developing infants completed 6-day sleep diaries at 1, 3, 6, 9 and 12 months from which a composite sleep score (CSS) was derived for each child at each month. Diary reliability was assessed once (for 54% of families) using all-night videosomnography. RESULTS: At 6 months, CSS scores were distributed bi-modally and thus differentiated into two groups by an empirically observed CSS cutoff score, with a majority (56%) of infants classified as self-sleep regulated (S-R) and the rest as non-self sleep-regulated (NS-R). At 12 months, 72% could similarly be classified as S-R, while 28% exhibited some continuing sleep disturbance. Discriminant function analysis investigated the predictors of S-R vs NS-R group membership at 6 and 12 months from parent and child variables recorded at 1 month. Parent presence at sleep onset and less total infant sleep time predicted group membership at 6 months with 94% classification accuracy, and parental presence at sleep onset and frequency of infant night wakings predicted group membership at 12 months with 85% accuracy. At 1 month, parents of infants later classified as NS-R at 6 and 12 months had higher frequencies of all settling activities than parents of those later classified as S-R. CONCLUSION: Variables measured at 1 month that predicted sleep status at 6 and 12 months were parental presence at sleep onset, frequency of infant night waking and total infant sleep time. The overall frequency of parent settling activities at 1 month also clearly differentiated the two sleep groups at the older ages. Parenting behaviours are modifiable factors and thus may have the potential for preventing pediatric sleep disturbances in children.

Attributions, causal beliefs, and help-seeking behavior of parents of children with autism spectrum disorder and sleep problems.

LAY ABSTRACT: Sleep problems are commonly reported among parents of children with autism spectrum disorder (ASD). Without effective treatment, such problems are unlikely to resolve. To date, we know very little about how and why parents of children with ASD seek help for sleep disturbance. Via an online survey, we gathered information about how parents make sense of their children's sleep problems, beliefs about their causes, sources of information, and help-seeking behavior. The analysis of responses from 244 parents revealed that parents commonly view sleep problems (a) as a consequence of their child's ASD, and unlikely to change over time (stable), and (b) as located within the child (intrinsic), stable over time, and difficult to treat. Despite this, parents also rated sleep problems as being important to treat. Eighty-two percent of parents surveyed reported seeking some kind of help for their child's sleep disturbance, and the average parent had tried six different treatment strategies, most commonly medical approaches (e.g. melatonin). The alignment between parents' treatment choices and those strategies that are supported by research was poor, but belief in the effectiveness of treatments was closely related to how often the treatment was used. These findings have important implications for parental education and clinical practice in the treatment of sleep problems in children with ASD.

Evaluating the effectiveness of therapeutic horse riding for children and young people experiencing disability: a single-case experimental design study.

Purpose: Therapeutic horse riding aims to improve the health of children and young people experiencing disability; however, its benefits across a range of health domains, particularly the impact on participation outcomes, are not well known. This research evaluated to what extent there was a change in riders balance, functional performance, social responsiveness, quality of life and participation outcomes as a result of therapeutic horse riding.Methods: A multiple-baseline across participants (n = 12) single-case experimental design, with randomly allocated baseline phase lengths, quantitatively evaluated how riders responded to a 20-week intervention.Results: Social participation outcomes measured using the Canadian Occupational Performance Measure demonstrated the most consistent positive between-phase differences (performance ES = 1.20, 95% CI [0.82, 1.63]; satisfaction ES = 1.11, 95% CI [0.73, 1.55]). A causal relationship was seen in three riders, but improvements only reached clinical significance for two riders when accounting for phase data trends. No significant outcome patterns were found comparing riders with principally physical impairments to those with principally psychosocial impairments.Conclusions: Being involved in therapeutic horse riding may improve rider's social participation in home, school and community settings. We postulate that rider self-concept development may be a mechanism of treatment effect leading to participation-level changes.Implications for rehabilitationSocial participation was the health outcome demonstrating the most consistent change following therapeutic horse riding, regardless of rider impairment.Therapeutic horse riding can improve social participation in settings beyond the riding arena.Greater intervention tailoring based on rider responses may enhance therapeutic horse riding intervention effects.

Systematic review of the effectiveness of behaviorally-based interventions for sleep problems in people with rare genetic neurodevelopmental disorders.

Sleep problems are prevalent in people with rare genetic neurodevelopmental disorders (RGND) and, in some cases, RGNDs are associated with specific forms of sleep disturbance that appear relatively unique. Although a notable amount of research has focused on behavioral intervention for sleep problems in people with higher incidence developmental disorders, research focused on potentially modifiable learning and environmental factors for people with RGND has received less attention. This review summarizes empirical evidence from studies providing behavioral interventions for sleep problems in RGND. A systematic search identified nine studies for inclusion. Studies were coded to extract data on participant characteristics, intervention components, dependent variables, research rigor and intervention effects. Study rigor was then evaluated using an established criteria and effects were classified as positive, neutral or mixed. Seven of the nine studies demonstrated positive treatment effects and two mixed results. In most studies, treatment consisted of multiple intervention components and were implemented by parents in the home. However, only three studies met criteria for an adequate level of rigor, thus greatly limiting certainty of conclusions. This review identifies current intervention practices and potential foci for future research.

Analyzing Therapeutic Change Using Modified Brinley Plots: History, Construction, and Interpretation.

The paper reviews the history, construction, and interpretation of modified Brinley plots, a scatter plot used in therapy outcome research to compare each individual participant's scores on the same dependent variable at Time 1 (normally pretreatment baseline; x-axis), with scores at selected times during or after treatment (y-axis). Since 1965 eponymously named Brinley plots have occasionally been used in experimental psychology to display group mean data. Between 1979 and 1995 a number of clinical researchers modified Brinley plots to show individuals' data but these plots have received little subsequent use. When constructed with orthogonal axes having the same origin and scale values, little or no change over time is shown by individuals' data points lying on or closely about the diagonal (450o) while the magnitude and direction of any improvement (or deterioration), outliers, and the extent of replication across cases shows via dispersion of points away from 450o. Interpretation is aided by displaying reliable change boundaries, clinical cutoffs, means, variances, confidence intervals, and effect sizes directly on the graph. Modified Brinley plots are directly informative about individual change during therapy in the context of concurrent change in others in the same (or a different) condition, clearly show if outcomes are replicated and if they are clinically significant, and make nomothetic group information, notably effect sizes, directly available. They usefully complement other forms of analysis in therapy outcome research.

Clinically Significant Symptom Reduction in Children with Attention-Deficit/Hyperactivity Disorder Treated with Micronutrients: An Open-Label Reversal Design Study.

OBJECTIVE: The purpose of this study was to investigate the clinical effect and safety of a broad spectrum, 36 ingredient micronutrient (vitamins and minerals) in treating children with attention-deficit/hyperactivity disorder (ADHD). METHODS: This open-label, on-off-on-off (reversal design) study followed 14 participants (8-12 years of age) with ADHD, diagnosed using standardized instruments, for 6 months with no dropouts. Following baseline assessment, including hematology and biochemistry screening, participants began an 8 week treatment phase with micronutrients titrated up to maximum dose (15 capsules/day). Treatment was withdrawn for 4 weeks, reinstated for a further 8 weeks, and then withdrawn for 4 weeks. Primary outcomes included the Conners' Parent Rating Scale, the Clinical Global Impressions Scale (CGI), and the Strengths and Difficulties Questionnaire - Parent version (SDQ). Secondary outcomes were mood and global functioning. RESULTS: Modified Brinley plots revealed a reduction in ADHD symptoms, improved mood, and improved overall functioning during intervention phases, and deterioration in ADHD symptoms, mood, and overall functioning during the withdrawal phases. Reliable change analyses, Cohen's d and percent superiority effect sizes, 95% confidence intervals and t tests confirmed clinically and statistically significant change between the intervention and withdrawal phases, with large effect sizes observed pre- to post-exposure of micronutrients (d = 1.2-2.2) on ADHD symptoms during intervention phases. Seventy-one percent of participants showed at least a 30% decrease in ADHD symptoms by the end of the second treatment phase, and 79% were identified as "much improved" or "very much improved" at the end of the second phase (5 months) based on the clinician-rated CGI when considering functioning generally. The SDQ showed that these benefits occurred across other areas of functioning including emotional symptoms, conduct problems, and prosocial behaviours. The children's self-reports confirmed the improvements. Excellent adherence to treatment occurred throughout, side effects were mild and transitory, and no safety issues were identified through blood analyses. CONCLUSIONS: This study demonstrates the clinical benefit, feasibility, and safety of broad-spectrum micronutrients in the treatment of childhood ADHD. Replications utilizing double-blind placebo-controlled studies are warranted. Trial is registered with the Australia and New Zealand Clinical Trial Registry: ACTRN12612000645853.

Motivational interviewing in health settings: a review.

There is evidence that patient-centred approaches to health care consultations may have better outcomes than traditional advice giving, especially when lifestyle change is involved. Motivational interviewing (MI) is a patient-centred approach that is gathering increased interest in health settings. It provides a way of working with patients who may not seem ready to make the behaviour changes that are considered necessary by the health practitioner. The current paper provides an overview of MI, with particular reference to its application to health problems.

The consolidation of infants' nocturnal sleep across the first year of life.

This review investigates development in the durations of infants' capabilities for sustained sleep across their first year, a matter of interest to clinicians, parents and researchers. It describes three aspects of sleep development: longest sustained sleep period (sleep sustained without awakening), longest self-regulated sleep period (behavioural quietude including sleep and quiet awakenings), and sleeping through the night (a predetermined nocturnal period). Clear trends were evident despite methodological differences making comparison between studies difficult. The most marked changes were across the first 4 months, particularly ages 1 and 2 months. Minimal changes followed through to 9 months and a small increase in all but the longest sustained sleep period, until age 12 months. Moore and Ucko's early, yet influential definition for sleeping through the night (24:00-05:00 h) may have underestimated infants' capacities for uninterrupted sleep. Infants do meet more stringent criteria and most can sleep 8 h by age 6 months and 9 or more hours thereafter. These findings have implications for clinicians addressing parental concerns around developmentally appropriate expectations of infant sleep. Researchers now have sufficient evidence to identify developmentally sensitive timing for preventive interventions for infant sleep disturbance.

Sleeping through the night: the consolidation of self-regulated sleep across the first year of life.

OBJECTIVE: To investigate the consolidation of infants' self-regulated nocturnal sleep over the first year, to determine when infants first sleep through the night from 24:00 to 05:00 hours (criterion 1), for 8 hours (criterion 2), or between 22:00 and 06:00 hours (the family-congruent criterion 3). METHODS: This was a prospective longitudinal study with repeated measures. Parents of 75 typically developing infants completed sleep diaries for 6 days each month for 12 months. Accuracy of parent reports were assessed by using videosomnography. RESULTS: The largest mean increase (504 minutes) in self-regulated sleep length occurred from 1 to 4 months. The survival function decreased most rapidly (indicating greatest probability of meeting criteria) for criterion 1 at 2 months, criterion 2 at 3 months, and criterion 3 at 4 months. A 50% probability of meeting criteria 1 and 2 occurred at 3 months and at 5 months for criterion 3. The hazard function identified 2 months (criteria 1 and 2) and 3 months (criterion 3) as the most likely ages for sleeping through the night. At 12 months, 11 infants did not meet criteria 1 or 2, whereas 21 failed to meet criterion 3. CONCLUSIONS: The most rapid consolidation in infant sleep regulation occurs in the first 4 months. Most infants are sleeping through the night at 2 and 3 months, regardless of the criterion used. The most developmentally and socially valid criterion for sleeping through is from 22:00 to 0:600 hours. At 5 months, more than half of infants are sleeping concurrently with their parents.