Sleep Treatments For Children With Asd

Download as pdf or txt
Download as pdf or txt
You are on page 1of 15

Developmental Neurorehabilitation

ISSN: 1751-8423 (Print) 1751-8431 (Online) Journal homepage: https://www.tandfonline.com/loi/ipdr20

Quantitative-Analysis of Behavioral Interventions


to Treat Sleep Problems in Children with Autism

Amarie Carnett, Sarah Hansen, Laurie McLay, Leslie Neely & Russell Lang

To cite this article: Amarie Carnett, Sarah Hansen, Laurie McLay, Leslie Neely & Russell Lang
(2019): Quantitative-Analysis of Behavioral Interventions to Treat Sleep Problems in Children with
Autism, Developmental Neurorehabilitation, DOI: 10.1080/17518423.2019.1646340

To link to this article: https://doi.org/10.1080/17518423.2019.1646340

Published online: 29 Jul 2019.

Submit your article to this journal

Article views: 25

View Crossmark data

Full Terms & Conditions of access and use can be found at


https://www.tandfonline.com/action/journalInformation?journalCode=ipdr20
DEVELOPMENTAL NEUROREHABILITATION
https://doi.org/10.1080/17518423.2019.1646340

Quantitative-Analysis of Behavioral Interventions to Treat Sleep Problems in


Children with Autism
Amarie Carnetta, Sarah Hansenb, Laurie McLayc, Leslie Neelyd, and Russell Lange
a
San Antonio Applied Behavior Analysis Research Consortium, University of Texas at San Antonio, San Antonio, TX, USA; bGeorgia State University,
Atlanta, GA, USA; cUniversity of Canterbury, Christchurch, New Zealand; dSan Antonio Applied Behavior Analysis Research Consortium, University of
Texas San Antonio, San Antonio, TX, USA; eClinic for Autism Research Evaluation and Support, Texas State University, San Marcos, TX, USA

ABSTRACT ARTICLE HISTORY


Sleep is an essential activity for human development. Often, children with autism spectrum disorder Received January 23, 2019
(ASD) are affected by a lack of sleep due to various types of sleep problems. We identified and analyzed Revised May 29, 2019
studies that were aimed at utilizing sleep interventions for children with ASD. A systematic search of Accepted July 16, 2019
databases, reference lists, and ancestral searches identified 18 studies for inclusion. Studies were KEYWORDS
summarized in terms of (a) participants, (b) targeted sleep problem and measures, (d) intervention Sleep problems; autism;
components, (e) research design and rigor, and (f) results. The aim of this review was to analyze the sleep intervention;
literature by evaluating the most commonly treated sleep problems, the various treatment components, quantitative-analysis; review
and strength of the results using a between case parametric effect size estimate. The most commonly
treated sleep problems were night wakings and bedtime disturbance. For interventions, all the studies
incorporated multiple treatment components, most often including the use of a consistent bedtime
routine. Effect size calculations indicated a moderate effect size, however, limited due to the small
number of studies. Results suggest the overall effectiveness of behavioral interventions for the treat-
ment of sleep problems for children with ASD. Based on our analysis, suggestions for practitioners
regarding current practices and future directions for research are discussed.

Sleep is essential for healthy human functioning across the life The popularity of pharmacological interventions (e.g., mela-
span and problems that prevent or disturb sleep are associated tonin and trimeprazine) may stem from the ease of delivery
with a myriad of adverse effects on physical and mental (taking a pill) relative to behavioral interventions as well as the
health.1 There are several forms of sleep problems including, immediacy of improvement often experienced when medication
sleep onset delay, nightwakings, bedtime resistance, night is effective.26,27 Further, because sleep is a natural biological
terrors and enuresis. Estimates of the prevalence of sleep process there may be a tendency to assume a biological (medical)
problems in typically developing children range from 15% to solution is necessary.28 However, there are some notable limita-
35%.2–5 However, among children with autism spectrum dis- tions to sole reliance of pharmacological treatments for sleep
order (ASD), sleep problems appear to be much more com- problems. For example, some families may avoid sleep medica-
mon, ranging from 47% to 83%2–5 with behavioral insomnias tions due to side-effects such as difficulty waking, nausea and
(e.g., frequent waking, sleep onset delay, and early morning undesirable interactions with other medications. Although
awakenings) being the most commonly reported.6–9 known sleep medication side-effects tend to be minor in most
In children with ASD, the effects of sleep problems may cases, there is insufficient data on the long-term side-effects of
be particularly harmful; specifically, sleep problems have nightly sleep medication use in children with ASD29. Further,
been linked to increased rates of physical health problems, although sleep medications have been found to be effective for
challenging behavior, cognitive deficits and overall ASD some topographies of sleep problems, they are not always effec-
symptom severity.10–17 In terms of family functioning, tive. For example, medication does not appear to be effective in
sleep problems in children with ASD are associated with the treatment of night-waking.28,29 Finally, financial cost and
elevated levels of parental stress18-20 depression21-23, limited access to medical services are obstacles for some families.
and20,24 fatigue. Given that sleep problems have significant Behavioral intervention approaches are derived from operant
deleterious effects on family functioning and quality of learning theory and have been used to treat sleep problems for
life25, intervention to address sleep problems in children many decades.31–38 Behavioral treatments for sleep problems in
with ASD is warranted. In general, the most common children with ASD have typically focused on antecedent events
approaches to treating sleep problems in children with (e.g., saliency of the discriminative stimulus) and contingent con-
ASD are pharmacological or behavioral intervention. sequences that influence sleep. Specifically, the focus is on

CONTACT Amarie Carnett [email protected] Department of Educational Psychology, The University of Texas at San Antonio, 501 W. Cesar E. Chavez
Blvd, San Antonio, TX 78207, USA
The authors report no conflicts of interests and are solely responsible for the content and writing of this paper.
© 2019 Taylor & Francis Group, LLC
2 A. CARNETT ET AL.

arranging environments and programming contingencies (e.g., Next, reference lists of all articles being considered for inclu-
reinforcement, extinction) such that behaviors likely to facilitate sion were screened for additional studies. Finally, ancestral
sleep (e.g., laying down still and quiet in bed) are supported via searches were completed from 2017, and hand searches cover-
reinforcement and factors that impede sleep (e.g., bright lights and ing the last 2 years (i.e., 2016–2018) were completed in 21
noise) are removed.35,39–41 Jin, Hanley, and Beaulieu42 highlighted journals that appeared during the database search and/or that
the importance of utilizing a comprehensive approach that focused on behavior analytical interventions (e.g., Sleep,
involves a functional assessment of the sleep problem to identify Developmental Journal in Child Neurology, Journal of
its operant function and inform the development of an individua- Applied Behavior Analysis, Behavioral Interventions). This
lized behavioral intervention. For example, behavioral assessment systematic search process was conducted in December 2017
may reveal that a child frequently leaves their bedroom at night in and resulted in a total of 45 articles to consider in detail for
order to escape the dark and therefore treatment might involve inclusion in this quantitative-analysis.
changes to environment (e.g., night light) and reinforcement for
staying in a dimly light room. However, a child with the same
Inclusion and Exclusions
topography of sleep problem might receive an entirely different
intervention if the operant function was to obtain adult attention. After the initial screening, the 45 identified articles were
In this way, behavioral treatment can be aligned with behavioral systematically and independently evaluated by the first
assessment results to tailor intervention components for specific and second authors using predetermined inclusion criteria.
children with respect to their individual needs and context. Specifically, in order to be included, the study had to: (a) be
Due to the prevalence of sleep disturbances among chil- published in an English language peer-reviewed journal (b)
dren with ASD, the impact of sleep problems on child and use a behavioral intervention (as defined by Baer, Wolf &
family well-being, and the need for effective individualized Risely47) to improve a sleep problem (e.g., hasten sleep onset,
treatment options to supplement or supplant sleep medica- decrease night waking, increase hours slept) (c) use an experi-
tion, research toward developing and improving behavioral mental or quasi-experimental research design (i.e., single case,
interventions to treat sleep problems in children with ASD quasi-experimental, randomized control trial) and (d) involve
has accumulated.36 Although a number of quantitative at least one participant with a diagnosis of autism, Asperger’s,
reviews have been conducted that cover behavioral treatments or PDD-NOS between ages 2 to18 years. Grey literature was
for children with ASD in general43–45, none have focused not included. Studies were excluded if the intervention did
exclusively on behavioral treatments of sleep problems in not include behavioral treatment components. Specifically, the
children with ASD. To our knowledge, only systematic study had to include some combination of reinforcement,
reviews and qualitative research syntheses are currently avail- environmental arrangement, antecedent manipulation, extinc-
able to guide evidence-based practice in this area.43-45 tion, and/or consideration of stimulus control. Interventions
This quantitative-analysis consolidates, summarizes, and ana- that relied solely on medication, aromatherapy, meditation,
lyzes the existing corpus of studies involving behavioral treat- nutritional or vitamin supplements or some other alternative
ment of sleep problems in children with ASD. The purpose of medicine or medical treatment were excluded. Studies were
the current quantitative-analysis is to analyze the literature by also excluded if the intervention on sleep was not behavioral
evaluating the most commonly treated sleep problems, the var- in nature (i.e., weighted blankets.48) For the group design
ious treatment components, and strength of the results using studies only, studies were excluded if an intervention compo-
a between case parametric effect size estimate (hedges g).46 nent was not included (i.e., evaluation of sleep problems49).
Overall, our aim is to inform practitioners regarding current These inclusion criteria resulted in a total of 18 studies
practices and offer directions for future research in this area. (13 single-case and five group design studies) being included
in this review. Reliability was conducted on both the abstract
screening level. Agreement on inclusion or exclusion was
Methods obtained for 44 of the 45 studies (98%). The coders discussed
the one disagreement until consensus was reached.
Searches
Systematic searches were completed by the first author in four
Data Extraction
electronic databases: Education Resource Information Center
(ERIC), Psychology and Behavioral Sciences Collection, The 18 studies were summarized with regards to the following
Medline, and PsycInfo. Each database was searched using the variables: (a) participant characteristics, (b) dependent vari-
search terms: “sleep or sleep problem or sleep disorder or able, (c) targeted sleep problem and dependent variable mea-
sleep disturbance or sleeplessness or insomnia or night awa- sures (d) intervention components, dosage, and agent (e)
kening or night waking or bedtime problem or bedtime research design and rigor, and (f) results.
resistance or bedtime refusal” and “auti* or pervasive devel- The following sleep problems were addressed in the 13
opmental disorder or mental retardation or developmental single-case studies and 5 group studies included in this quan-
disability” and “treatment or intervention or training or par- titative-analysis: (a) night wakings – any awakenings that
ent training or approach.” Searches were limited English occur following initial sleep onset and prior to morning
language articles appearing in peer-reviewed journals. Titles awakenings (b) bedtime disturbances – sleep-interfering beha-
and abstracts of the retrieved articles were initially screened viors that occur after the parent/caregiver bids the child good-
by the first author to determine if the study appeared relevant. night, (c) sleep onset delay – the amount of time elapsed from
DEVELOPMENTAL NEUROREHABILITATION 3

when the parent/caregiver bids the child goodnight and the Similar software programs have been shown to reliably digitize
child is asleep, (d) co-sleeping – sleeping in the same bed as graphed data with low error.64 Thirty percent of all digitized data
a parent/caregiver/sibling, (e) sleep duration – the amount of was re-graphed in excel and compared to the original graph to
time a child is asleep, (f) daytime sleeping – sleeping during check for errors. Re-graphed data were examined for visual simi-
daytime hours, not in the context of a scheduled nap time, larity and point-to-point correspondence with initial screenshots
and (g) sleep terrors – parasomnia that involve a sudden in terms of level, trend, and variability. No major differences were
awakening from slow wave sleep and present an intense found.
emotional response (e.g., piercing screams, sweating, rapid
heart rate). For group design studies included in this quanti-
Measurement of Non-overlap Index
tative-analysis, a variety of sleep scales and biopsychological
measures were reported rather than behavioral observation of Tau-U was calculated for all single-case studies meeting mini-
specific sleep problems; however, these measures typically mum criteria for (a) digitizing and (b) calculations of Tau-U
encompass the same range of sleep issues (see Table 1). (i.e., a minimum of three data points per phase, using
In line with typical quality evaluation procedures, studies a multiple baseline, multiple probe, reversal or withdrawal
were evaluated for quality standards using the single case and design) using the digitized data. Tau-U is a non-parametric
group evaluation methods developed by Reichow, Volkmar, non-overlap estimate with limited ability to control for trend
and Cicchetti.60 Specifically, this system of evaluation is based in baseline and autocorrelation.65 Tau-U was calculated using
on rigor, methodological strength, and replicability to system- the calculator available on singlecase.org for all applicable
atically review empirical intervention research related to indi- studies. Tau-U was selected due to its estimation of non-
viduals with autism. For the included group designs, studies overlapping pairs comparable to other non-overlap estimates
were coded in a similar method using the evaluation methods (e.g., NAP), and its increased ability to account for trend in
developed by Reichow, Volkmar, and Cicchetti.60 Specifically, baseline. The baseline correction available at singcase.org was
study report of the effectiveness of the intervention was used only selected if significant risk for autocorrelation (e.g., trend
including effect size measures if reported, significance of in baseline) was detected by the software (available at ktarlow.
effect, and/or pre-post means. Further, each single-case com). While trend correction in baseline can help with the
study results were rated as positive, mixed, or negative based accurate detection of effect, when used unnecessarily it can
on visual analysis of graphed results61 and using the defini- inflate effect estimates.65
tions developed by Lang et al.62 and Davis et al.62 Results were
rated as positive if all the participants showed improvement
Measurement of Effect Size and Forest Plots
across the targeted sleeping behavior(s) above baseline levels.
Results were rated as having mixed result if some, but not all, A between case effect size indicator was calculated using
participants showed improvement in targeted sleep problem a macro created for the Statistical Package for Social
or if all participants showed improvement in some, but not all Sciences (SPSS). The digitized data were coded by participant,
of the targeted sleep problems, and results were rated as phase, study, and session number for all studies meeting
negative if none of the participants showed improvement in minimum requirements for analysis (i.e., at least three
the targeted sleep behavior. The second author checked 100% A-B comparisons, a minimum of two points per phase,
of the table for accuracy, and there were no discrepancies using a multiple baseline or reversal/withdrawal design).
found (see Table 1). A manual for the initial search procedure Studies that primarily used an alternating treatment design
and the data extraction procedures are available from the first or that had fewer than two data points per phase were not
author upon request. analyzed. For studies with an embedded reversal, the reversal
was not analyzed, only the leading phase. Low data studies,
embedded reversals, and alternating treatment designs were
Graph Digitization of Single-Case Graphs
not calculated due to limitations of the SPSS macro used,
The second author took screenshots of graphed data from all which requires a minimum of three data points per phase,
eligible studies for digitization. Data were not digitized if it was three A-B comparisons per study, and studies controlling for
not represented on a line graph with sufficient data to estimate at time (i.e., multiple baseline or reversal/withdrawal). Hedges g,
minimum an A-B comparison (i.e., change in level or trend a d-estimate with controls for small sample size appropriate
between at minimum one baseline to one treatment condition). for single case, was calculated (a) by study, (b) across depen-
For example, Piazza et al.57 were not digitized due to no clear dent variables with a minimum of three studies in order to
definition between sessions. However, visual analysis was still identify scope of evidence per sleep problem area and (c) an
conducted for all studies regardless of digitization. While it is omnibus d-estimate to demonstrate the strength of the evi-
possible to calculate Tau-U values using a rank order procedure, dence in this area.
Hedges-g requires raw data and so we digitized data rather than Hedges g reduces inflation of effect size that often occurs
conduct a rank order procedure. An A-B comparison was defined with small sample sizes through the use of pooled weighted
as a change from baseline to intervention, or from intervention to standard deviations in the place of pooled standard devia-
baseline (i.e., for a reversal or withdrawal design, baseline to tions. Thus, there is lower risk of inflated estimate of the
intervention, intervention to baseline, second baseline to second variance between treatment and control (or in the case of
intervention, etc.) All digitization was completed using Plot single-case data baseline and intervention), due to larger
Digitizer Pro (available from http://plotdigitizer.sourceforge.net). data contributing greater estimation of the variance.46 Using
Table 1. Summary of sleep research.
4

Participant
Characteristics Targeted Sleep Problem & Dependent Variable Research Design
Citation (N, Age & Diagnosis) Measures Intervention Components, Dosage and Agent & Rigor Results
Christodulu & N = 4 (2 male; 2 female) Sleep Problem: Night wakings and bedtime Components: Adjust bedtime routine and sleep Design: MBL2 Effect Sizes: g = 1.29
Durand, Agesa: 2.6 to 5.11 (M = disturbances restriction across Overlap Estimate: Decreases in rate of night time
200431 3:6) Measures: Frequency of bedtime disturbances Dosage: Specific data not provided. Intervention participants disturbances (Tau-U = 0.38) and night time wakings
Diag: Autism; Immune and night wakings per week; Parent sleep phase M= 18 weeks (Joey excluded). Rigor3: Weak (Tau-U = 0.48)
Deficiency; Charge satisfaction questionnaire to assess social Agent: Parent Visual Analysis: Positive and demonstrated
Association; PDD-NOS validity. experimental control.
A. CARNETT ET AL.

Comorbidities: Sensory Generalization: NR


Integration Disorder; Maintenance: NR
Hypotonia Social Validity: Parents reported satisfaction with
treatment and improved sleep.
Didden, Curfs, N = 4 (3 males; 2 Sleep Problem: Night wakings, Sleep onset Components: Functional assessment, extinction Design: Non- Effect Size: Decrease in bedtime disruptions (g = 0.99)
van Driel, & de female) delay, co-sleeping. (parent attention), and Depakine (anticonvulsive concurrent MBL2 Tau-U: Decrease in bedtime disruptions (Tau-U =
Moor39 Agesa: 1:11 to 25 (M= Measure: Number of minutes of night time medication) participant with seizure disorder. across 0.81)
16:4) disruption (e.g., crying, screaming, getting out Dosage: Specific data not provided. Intervention participants and Visual Analysis: Positive and demonstrated
Diag: Mental handicap; of bed). phase approximately M= 67 weeks. a reversal experimental control.
Autism; Down’s Agent: Parent Rigor3: Adequate Generalization: NR
syndrome; Maintenance: Positive
Developmental Delays Social Validity: Parents reported satisfaction with
Comorbidities: Seizure treatment and improved sleep.
Disorder; Visual and
Physical impairments
Durand, N = 2 (females) Sleep Problem: Bedtime disturbances and night Components: Sleep restriction (restricting the Design: MBL2 Effect Sizes: g = 1.29
Christodulu29 Agesa: 4:0 wakings amount of time the child was in bed to 90% of the across Tau-U: Small decrease in bedtime distruptions (Tau-U
Diag: Autism; Measure: Number of bedtime disturbances per total time that the child slept). participants = 0.38), moderate decrease in night wakings (Tau-U
Developmental Delays week. Dosage: Specific data not provided. Intervention Rigor3: Weak = 0.48)
phase M= 19.5 weeks. Visual Analysis: Positive and demonstrated
Agent: Parents experimental control.
Generalization: NR
Maintenance: NR
Social validity: Parent reported satisfaction with
treatment and improved sleep.
Durand, Gernert- N = 5 (2 males; 2 Sleep Problem: Bedtime disturbances, and night Components: Parent implemented consistent Design: MBL2 Effect Sizes: Not calculated
Dott, & females) wakings. bedtime routine, and graduated extinction across Overlap Estimate: Large decrease in bedtime
Mapstone50 Agesa: 2:0 to 12 (M= Measure: Sleep onset, bedtime disturbances, Dosage: Specific data not provided. Intervention participants disruptions (Tau-U = 0.66), small decrease in night
6:4) night wakings, and parent responses (i.e., phase M= 7.5 weeks. Rigor3: Weak wakings (Tau-U = 0.66).
Diag: Down syndrome; staying in bed with child). Agent: Parent Visual Analysis: Positive. All participants showed
Autism; Mental a decrease in the frequency of night wakings and
retardation with sleep disturbances.
a chromosomal Generalization: NR
abnormality; PDD and Maintenance: NR
VI. Social validity: NR
50
Durand N = 3 (2 males; 1 Sleep Problem: Sleep terrors Components: Parent implemented pre-scheduled Design: MBL2 Effect Sizes: Large decrease in sleep terrors (g = 3.45).
female) Measure: Onset and duration of sleep terrors. awakenings based on the child’s typical sleep terror across Overlap Estimate: Decrease in sleep terrors (Tau-U =
Agesa: 3:0 to 7:5 (M= time. participants 0.98)
5:1) Dosage: Specific data not provided. Intervention design Visual Analysis: Positive. All participants decreased in
Diag: Autism phase M= 19 weeks. Rigor3: Adequate the frequency of night terrors to zero occurrences
Agent: Parent per night and maintained during follow-up.
Generalization: NR
Maintenance: Positive
Social validity: Social validity data indicated parent
overall parent satisfaction with the treatment and
outcomes.
(Continued )
Table 1. (Continued).
Participant
Characteristics Targeted Sleep Problem & Dependent Variable Research Design
Citation (N, Age & Diagnosis) Measures Intervention Components, Dosage and Agent & Rigor Results
Friedman & N = 1 (male) Sleep Problem: Excessive daytime sleeping Components: Environmental arrangement (removing Design: ABAB Effect Sizes: Not calculated
Luiselli51 Agesa: 13:0 Measure: Cumulative duration of daytime stimuli that was slept on), response interruption and experimental Overlap Estimate: Decrease in excessive daytime
Diag: Autism sleeping. redirection, and positive reinforcement. design sleeping (Tau-U = 0.72)
Dosage: Specific data not provided. Intervention Rigor3: Adequate Visual Analysis: Positive. The participants day time
phase N= 14 days. sleeping decreased to zero rates during intervention
Agent: Classroom Staff phases and maintained during follow-up.
Generalization: NR
Maintenance: NR
Social validity: NR
Jin, Hanley, & N = 3 (male) Sleep Problem: Delayed sleep onset, bedtime Components: Comprehensive treatments for each Design: Effect Sizes: Weak decrease in delayed sleep onset (g
Beaulieu42 Agesa: 7:0 to 9:0 (M= disturbance. child included procedures to enhance the Nonconcurrent = 0.29)
8:3) Measure: Sleep onset delay and sleep- establishing operations and discriminative stimuli for MBL2 across Overlap Estimate: Small decrease in delayed sleep
Diag: Autism interfering behaviors. sleep. subjects onset (Tau-U = 0.18)
Other: Typical Two-hour parent training for each treatment plan. Rigor3: Weak Visual Analysis: Positive. Each participant had
developing Dosage: Specific data not provided. Intervention improvements in decreases of sleep-onset and sleep
phase M= 27 nights. interfering behaviors and maintained during follow-
Agent: Parent up.
Generalization: NR
Maintenance: Positive
Social validity: Social validity data indicated parent
satisfaction with the assessment, treatment, and
amount of behavior change.
Johnson, Turner, N= 47 families Sleep Problem: Components: Behavioral parent training including Design: Effect Sizes: d = 0.18, 0.42, and 0.16 and weeks 0, 4
Foldes, Brooks, Agesa: −6 Measure: Parent treatment fidelity, composite video examples. Role play, modelling, problem Randomized and 8 respectively.
Kronk, & Diag: Autism sleep index score, actigraphy solving, and homework. Parents worked with control trial. Overlap Estimate:
Wiggs, 201241 trainers to complete individualized behaviour Rigor3:Adequate Data Analysis: Both groups improved from initial
intervention plan. levels. The improvement was more significant in the
Dosage: 8 weeks BPT group.
Agent: Parents Generalization: NR
Maintenance:
Social validity:
Knight & N = 3 (2 males; 1 Sleep Problem: Parent compliance with Components: Circadian rhythm management, white Design: Non- Effect Sizes: Moderate decrease in night wakings (g =
Johnson52 female) treatment package. noise machine, bedtime routine of calm activities, concurrent MBL2 0.68), sleep onset latency (g = 0.67)
Agesa: 4:0 to 5:0 (M= Measure: Frequency of night wakings and sleep and graduated extinction. across Overlap Estimate: Moderate decrease in knight
4:7) onset delay. Dosage: Specific data not provided. Intervention participants wakings (Tau-U = 0.48), sleep onset latency (Tau-U =
Diag: Autism Night wakings, sleep onset delay, co-sleeping. phase M= 30 nights. Rigor3: Adequate 0.57)
Agent: Parents Visual Analysis: Positive. Each child showed
improvement in frequency of night wakings and
maintained during follow-up sessions. Each
participant showed overall decreases in sleep delay
and maintained during follow-up sessions.
Generalization: NR
Maintenance: Positive
Social validity: Social validity data indicated parent
satisfaction with the treatment and treatment
outcomes.
Loring, Johnston, N = 18 Sleep Problem: Going to bed, falling asleep, Components: Adolescents and their parents received Design: Pre-post Effect Sizes: NA
Gray, Agesa: 11–18 maintaining sleep, reinitializing sleep, returning two individual sleep education sessions. design. Data Analysis: Parents reported larger changes than
Goldman, & Diag: Autism to wakefulness. Dosage: Two 2 hour sessions with telephone calls to Rigor3: Weak adolescents. All components showed positive change
Malow, 201653 Measure: Actigraphy, adolescent sleep wake follow up Generalization: NA
DEVELOPMENTAL NEUROREHABILITATION

scale, Modified Epworth Sleepiness Scale Agent: Parent as trained by psychologist Maintenance: NA
(M-ESS), parent/adolescent absorption scale Social validity: NR
(Continued )
5
6
Table 1. (Continued).
Participant
Characteristics Targeted Sleep Problem & Dependent Variable Research Design
Citation (N, Age & Diagnosis) Measures Intervention Components, Dosage and Agent & Rigor Results
Malow, et al., N = 80 Sleep Problem: Prolonged sleep latency of at Components: Parents randomly placed in Design: Effect Sizes: NR
201454 Agesa: 5.6 ave least 30 minutes. individualized sleep intervention or group training. Randomized
Data Analysis: There was no statistically significant
Diag: Autism Measure: Actigraphy and Sleep Diaries, CSHQ, Dosage: Two 2h sessions a week apart with follow control trial.
difference between individualized and group
Family Inventory of sleep habits, Child Behavior up phone calls Rigor3: Adequate
treatments. Across both groups, significant change
checklist, Repetitive Behavior Scale Revised. Agent: Parents was seen on multiple dimensions of the CSHQ.
A. CARNETT ET AL.

Generalization: NR
Maintenance: NR
Social validity: NR
Malow, N = 10 Sleep Problem: A variety of sleep problems Components: Parents were given access to a manual Design: Pre-post Effect Sizes: NR
MacDonald, Agesa: including night wakings, bedtime resistance, and guided through a sequence of chapters on design Data Analysis: Eight of the 10 families were able to
Fawkes, Alder Diag: Autism and co-sleeping. common sleep strategies. Rigor3:Weak implement the entire intervention. Of the eight
& Katz27 Measure: Change on the Children’s Sleep Habits Dosage: Information not provided. families, six families showed some improvement on
Questionnaire (CSHQ), the Family Inventory of Agent: Nurses targeted areas.
Sleep Habits, actigraphy, and sleep diaries. Generalization: NR
Maintenance: NR
Social validity: NR
McLay, France, N = 1 (male) Sleep Problem: Sleep onset delay, Bedtime Components: Antecedent manipulations: establishing Design: Clinical Effect Sizes: Not calculated
Blampied, Agesa: 3:9 disturbances, night wakings, and sleep consistent bedtime routine with the use of social case with four Overlap Estimate: Small decrease in night wakings
Danna, & Diag: Autism duration. story and teaching replacement behaviors (cuddling treatment phases (Tau-U = 0.27), moderate decrease in bedtime
Hunter55 Measure: Frequency of curtain calls (bids for soft toy). and follow-up. disturbance (Tau-U = 0.47)
attention after told goodnight), frequency of Consequence manipulation: elimination of Rigor3: Weak Visual Analysis: Positive. The child showed overall
breastfeeding, frequency of night wakings, and breastfeeding (extinction), graduated extinction of decreases in frequency of curtain calls, breast
duration of night wakings. co-sleeping, positive reinforcement of alternative feeding, and night wakings, as well as a decrease in
behaviors. duration of night wakings. These results were
Dosage: Specific data not provided. Intervention maintained during follow-up, with the exception of
phase N= 54 nights. curtain calls.
Agent: Parents Generalization: NR
Maintenance: Positive
Social validity: Social validity data indicated
satisfactory level of parent satisfaction with the
treatment and effectiveness, but reported discomfort
due to undesirable side effects and time of the
intervention.
Moon, Corkum, & N = 3 (2 males; 1 Sleep Problem: Sleep onset delay Components: Parent use and training using the Design: Case- Effect Sizes: Moderate decrease in sleep onset latency
Smith56 female) Measure: Sleep onset delay, sleep duration, Better Nights, Better Days: Treatment for Sleep series design (g = 0.60)
Agesa: 8:3 to 9:4 (M= sleep efficiency, and daytime problem Difficulties Parent Handbook (sleep restriction with Rigor3: Weak Overlap Estimate: Moderate decrease in sleep onset
8:9) behavior. positive reinforcement). latency (Tau-U = 0.80)
Diag: Autism Dosage: Specific data not provided. Intervention Visual Analysis: Mixed. For sleep onset delay, an
phase M= 8 weeks. overall reduction was shown at end-of-treatment
Agent: Parent compared to baseline for each participant. For sleep
duration, no significant changes were observed. For
sleep efficiency no clear pattern of change and
a small decreases in daytime behavior problems were
also reported.
Generalization: NR
Maintenance: NR
Social validity: Social validity indicated a satisfactory
level of parent satisfaction with the treatment, would
recommend it to other families, and found the
manual helpful.
(Continued )
Table 1. (Continued).
Participant
Characteristics Targeted Sleep Problem & Dependent Variable Research Design
Citation (N, Age & Diagnosis) Measures Intervention Components, Dosage and Agent & Rigor Results
Piazza, Fisher, & N = 14 (gender not Sleep Problem: Night wakings, sleep duration, Components: Random assignment to either bedtime Design: Effect Sizes: Not calculated
Sherer57 specified) sleep onset delay. scheduling or faded bedtime with response cost A-B design Overlap Estimate: Not calculated
Agesa: 4 to 14 (M= 7:8) Measure: Mean hours of disturbed sleep. (FBRC). Rigor3: Weak Visual Analysis: Positive. Decrease in the mean hours
Diag: Developmental Dosage: Information not provided. of disturbances were observed for both treatments,
disabilities; Autism; Agent: Staff however more significant decreases we observed for
PDD FBRC.
Comorbidities: seizure Generalization: NR
disorder and cerebral Maintenance: NR
palsy Social validity: NR
Reed, McGrew, N = NR, 15–25 families Sleep Problem: General sleep problems as Components: Parents participated in workshops on Design: Pre-post Effect Sizes: NR
Artibee, Agesa: 3 to 10 captured on the CSHQ a variety of sleep-related topics. design Data Analysis: Some improvement was evident on
Surdkya, Diag: Autism Measure: CSHQ, the Parental Concerns Dosage: Six weeks Rigor3: Weak the CSHQ and the PSQ, as well as actigraphy for
Goldman, Questionnaire (PSQ), the Repetitive Behavior Agent: Parent those families with complete data. Most change was
Frank, Wang & Scale Revised, The parenting strees index – not statistically significant. No change was evident
Malow short form (PSI), and actigraphy on the PSI.
(2009)58 Generalization: NR
Maintenance: NR
Social validity: NR
Weiskop, N = 1 (male) Sleep Problem: Co-sleeping, night wakings. Components: Parent training on antecedents and Design: Effect Sizes: Not calculated
Matthews, & Agesa: 5:4 Measure: Frequency of nights the child falls consequence strategies (for target child and sibling), A-B design with Overlap Estimate: large decrease in co-sleeping (Tau-
Richdale32 Diag: Autism asleep in own bed and frequency of nights the establishing a bed time routine, and extinction long term follow- U = 0.90), sleep onset latency (Tau-U = 0.80)
child stays in bed all night. procedures. up. Visual Analysis: Positive. Frequency of nights per
Goal Achievement Scale Dosage: Specific data not provided. Intervention Rigor3: Adequate week that the child fell asleep in his bed increased as
phase N= 7 weeks. well as the number of nights the child staying in his
Agent: Parent bed alone all night and maintained during follow-up.
Generalization: NR
Maintenance: Positive
Social validity: Social validity data indicated parent
satisfaction with the treatment and treatment
outcomes.
Weiskop, N = 13 (10 male; 3 Sleep Problem: Sleep onset delay, bed Components: Parent training involving goal setting, Design: Effect Sizes: Weak decrease in bedtime disturbances
2
Richdale, & female) disturbances, night wakings, co-sleeping. antecedents and consequence strategies, effective Concurrent MBL (g = 0.11)
Matthews59 Agesa: 1:1 to 9:1 (M= Measure: Number of pre-sleep disturbances, instructions and partner support strategies, across Overlap Estimate: Small decrease in bedtime
5:1) number of nights that the child fell asleep in extinction (standard, graduated, and ignoring with participants. disturbances (Tau-U = 0.25).
Diag: Autism; FXS their own bed, average sleep latency, number parent present). Rigor3: Adequate Visual Analysis: Positive. For pre-sleep disturbances,
of night of co-sleeping, and average duration Dosage: Specific data not provided. Intervention falling asleep alone, night wakings, and co-sleeping
of sleep. phase M= 7 weeks. improvement was observed in each participant
Agent: Parent (study 1 and 2) and during follow-up. For sleep
latency and duration of sleep some individuals did
not a significant change as they were not
experiencing problems related these areas or no
change was observed.
Generalization: NR
Maintenance: NR
Social validity: Social validity data indicated parent
satisfaction of the treatment and outcomes.
DEVELOPMENTAL NEUROREHABILITATION

a
Ages expressed as years. months; 2Multiple Baseline Design; 3Based on criteria from Reichow et al., 2008
7
8 A. CARNETT ET AL.

this pooled weighted standard deviation, confidence intervals included sleep terrors (targeted in one study)66, daytime sleeping
were calculated for each study’s effect size, and these were (targeted in one study)51, and sleep duration (targeted in one
graphed in forest plots to allow better comparison between study).55 Group design studies did not report change in indivi-
effect sizes. Forest plots indicate the confidence interval, the dual behaviors but rather change on aggregate sleep quality
effect size, and the weight of the effect. Studies were weighted scales.
based on number of participants and strength of effect and All of the single-case studies included in this quantitative-
this was graphed (Figure 2). Similar graphs were created for analysis included multiple intervention components to
Tau-U values to include single-case studies for which d-esti- address the targeted sleep problem. The most commonly
mates could not be calculated (Figure 1). reported component included the use of extinction or
No group design effect size was included in forest plots a variation of extinction (i.e., graduated extinction), and was
because no group design study included in this review used seven (50%) of the studies.32,39,50–56 Other commonly
reported sufficient data to estimate an effect size, and did used components included the use of sleep restriction and
not report their own effect sizes. schedule wakings, used in four (29%) studies29,31,57,59,66 estab-
lished bedtime routines used in five (36%) studies31,32,50,52,55,
and antecedent and reinforcement strategies, used in five
Results
(36%) studies.32,42,55,56,59 Other intervention components
The 13 single case and six group design studies provided used included response cost57, replacement behavior55, and
sleep-related interventions to a total of approximately 210 circadian rhythm management.52 Lastly, dosage of interven-
participants with ASD (27 participants in single-case design tion, when reported, varied with a range between approxi-
studies and 183 participants from group designs that reported mately 7.5 and 65 weeks. All but two of the included studies
number of participants). In the single-case design studies, 14 were conducted by parents, with the remaining two being
participants were male (47%), six were female (20%), and 10 conducted by classroom teachers (excessive daytime sleeping)
participant’s gender were not reported (33%). In the group and staff at a treatment inpatient facility. Group design studies
design studies, insufficient information was given about par- also used various behavioral strategies, although most
ticipant gender to report. Twenty-seven of the participants in reported the results of a manualized intervention or
single-case studies had a diagnosis of autism, three (8.58%) a piloted manualized intervention making it difficult to iden-
had an additional diagnosis (e.g., seizure disorder, vision tify the individual components.
impairment, cerebral palsy), and three (8.58%) were identified Seventeen (94%) of the studies reported positive results for
as having PDD-NOS or PDD. Of the six group design studies, the treatment of the targeted sleep intervention. One study
100% featured participants with ASD, 33% featured (7%) reported mixed results related to sleep duration.56 Nine
a participant with developmental disabilities, and 33% fea- (64%) of the single-case studies also collected social validity
tured participants with co-morbid disabilities. Across all stu- data.29,31,32,42,52–56,59,66 Results from the social validity data
dies included in this review, participant age ranged from 1:1 indicated acceptable levels of parent satisfaction for treatment
to 18 years (M = 6:5). However, it should be noted that the procedures and behavioral outcomes. Single-case studies were
mean age is an estimate, given not all studies reported exact examined using Reichow and colleagues’ quality index for
ages for each participant. autism intervention studies (2008). Results of this analysis
In the included single-case studies, eleven (85%) of the indicated that six of the 13 studies (46%) received a rating
studies reported conducting some method of assessment of “weak”29,31,42,50,55–57 and seven of the 13 studies received
(e.g., direct or indirect) prior to the intervention.31,32,39,42,50– a rating of “adequate”.32,39,51,52,59,66 No study included in this
56,59
The most common type was a questionnaire (e.g., Albany review received a rating of “strong”.
Sleep Problem Scale; Children’s Sleep Habits Questionnaire; Lastly, eight (57%) of the studies collected follow-up
Sleep Assessment and Treatment Tool) or semi-structured data.32,39,42,51–55,59,66 Of those studies, the mean time for collect-
interview.31,32,56 Five (36%) of the studies used a functional ing follow-up data was 4.7 months after the completion of the
behavior assessment to help identify controlling variables and intervention phase. For two studies, McLay et al.55 and Weiskop,
analyze the contingencies involved.39,42,51,55,59 For the Matthews, & Richdale32 follow-up was collected across two
included group design studies, all studies measured change points of time (i.e., 44 days and 79 days for McLay et al.,55; 3
in participant sleep including sleep questionnaires as above months and 12 months for Weiskop, Matthews, & Richdale.32)
and standardized measures of sleep quality (e.g., composite One study42 did not specify when follow-up was collected.
sleep index).
In the included single-case design studies, sleep interventions
Non-overlap Estimates
related to night wakings were targeted most often, occurring in
nine (69%) of the studies reviewed.29,31,32,39,50,52,55,57,59 For the 12 single-case studies for which Tau-U could be calculated,
The second most commonly targeted sleep problem was sleep Tau-U ranged from 0.18 (small or weak effect) to .81 (large or
onset, and was targeted in seven (54%) of the studies.31,39,42,52–57 strong effect). The pooled Tau-U value was also collected for the
The third most common targeted sleep problem was bedtime three most frequently targeted dependent variables. Sleep onset
disturbances, and was targeted in six (43%) of the delay had the largest pooled non-overlap estimate (i.e., 0.63, range
studies.29,31,42,50,55,57 The fourth most commonly addressed 0.57–0.81) across three studies32,52,56, indicating a moderate to
sleep problem was co-sleeping, targeted in four (29%) of the large effect. The lowest pooled non-overlap estimate across depen-
reviewed studies.32,39,52,59 Other sleep problems addressed dent variables was night waking (0.33, range 0.18–0.48) across five
-0.20 0.00 0.20 0.40 0.60 0.80 1.00 1.20 1.40 1.60
Study Effect Size CI LL CI UL Weight
Christodulu & Durand, 2004a 0.38 0.10 0.66 0.21
Durand 1996a 0.66 0.30 1.02 0.16
Didden et al., 2002 0.81 0.55 1.07 0.22
McClay et al. b 0.47 0.23 0.71 0.24
Weiskopf et al., 2005 0.25 -0.13 0.63 0.16
Bedtime Disruptions
Christodulu & Durand, 2004b 0.48 0.20 0.76 0.23
Durand 1996b 0.39 0.03 0.75 0.14
Jin et al., 2013 0.18 -0.10 0.46 0.23
Knight & Johnson, 2014 a 0.48 0.05 0.91 0.09
McClay et al. a 0.27 0.03 0.51 0.31
Night Wakings
Knight & Johnson, 2014 b 0.57 0.19 0.95 0.60
Moon et al., 2011 0.60 -0.13 1.33 0.16
Weiskopf et al., 2001b 0.81 0.18 1.44 0.24
Sleep Onset Latency
Combined effect size 0.49 0.30 0.68

Figure 1. Forrest plot of d-estimates for single-case design studies.


DEVELOPMENTAL NEUROREHABILITATION
9
10 A. CARNETT ET AL.

-1.00 -0.50 0.00 0.50 1.00 1.50 2.00

Christodulu & Durand, 2002 a 1.29 0.64 1.94 0.48

Weiskopf et al., 2005 0.11 -0.53 0.75 0.52

Bedtime Disruptions

Christodulu & Durand, 2002b 0.38 -0.43 1.19 0.09

Didden et al., 2002 0.99 0.30 1.68 0.12

Jin et al., 2013 0.29 -0.05 0.63 0.50

Knight & Johnson, 2014 a 0.68 0.24 1.12 0.30

Night Waking

Knight & Johnson, 2014 b 0.69 0.31 1.07 0.79

Moon et al., 2011 0.68 -0.07 1.43 0.21

Sleep Onset Latency

Combined effect size 0.60 0.42 0.79

Figure 2. Forrest plot of tau-u overlap indices for single-case design studies.

studies, indicating a weak to moderate effect.31,42,50,52,55 The third g = 0.29 to g = 0.9931,39,42,52 Studies in these three groups had
dependent variable with sufficient studies was bedtime distur- a pooled effect size of g = 0.60, indicating a moderate effect of
bance and it also had a moderate pooled effect (0.52; range the research on behavioral treatments of sleep problems in
0.25–0.81) across the five studies.31,39,50,55,59 Studies not included students with ASD. Following guidelines for the comparison
in the pooled weighted Tau-U did not fall under these categories of effect sizes, there was no significant difference in the
and included sleep terrors66 (0.98) and co-sleeping52 (0.60). Across strength of the evidence across these main groups. The only
the studies included in the three main categories, the weighted study targeting a dependent variable not included in the
omnibus Tau-u was 0.52, indicating a moderate effect of the omnibus effect size was Durand and colleagues, targeting
included interventions (0.52, 0.10–0.92). sleep terrors (g = 3.45).55 While some of the included group
designs would have allowed for calculation of an effect size,
we elected to not compare single case and group design effect
Between Case Effect Size Estimate sizes due to the difference in scale in the resulting effect size
Nine of the 13 included single-case studies included had measurements.
sufficient data to calculate a hedges g effect size estimate.
Hedges g follows the same interpretation guidelines as
a cohen’s d, where 0.2 indicates a small effect, 0.5 represents
Group Design Results
a medium effect and 0.8 a large effect size.63 For the difference
in effect to be significant, there must be a minimum of 0.2 The five included group designs included 147 families of
difference between effect size values.63 This yielded 24 children with ASD. Of the included group designs, most
A-B comparisons entered into the between case effect size took data using a sleep scale including The Children’s Sleep
quantitative-analysis. Across the appropriate studies, the Habits Questionnaire (CSHQ) the Family inventory of sleep
g value ranged from 0.11 to 3.45, indicating evidence to habits and the Modified Epworth Sleepiness Scale. Of the five
suggest a variable effect across studies (weak to very strong). group design studies, all five used actigraphy methods, and
Across the three groups, sleep onset delay had the largest two of the five included parent sleep diaries. All five of the
pooled effect (g = 0.69) with two studies available for included studies used some form of parent training, including
analysis.31,59 Analogous to the Tau-u analysis, studies addres- parent coaching, didactic sleep training, parent workshops, or
sing night waking had the lowest pooled effect of g = 0.50, manualized parent interventions. Overall results of group
indicating a moderate effect across four studies ranging from designs included minimal improvement on targeted sleep
DEVELOPMENTAL NEUROREHABILITATION 11

behaviors, with only two of the five studies showing statisti- significant enough to warrant intervention.69 Thus, the incon-
cally significant change in child sleep. sistencies in the data collected, definitions of sleep problem
behavior, and the recording of the dependent variables pre-
cludes a level of methodological rigor that would be necessary
Discussion
for large-scale statements to be made about evidence-based
Maintaining healthy sleep behavior is an important aspect of practices. Best practices for statistical analysis of effect sizes
daily functioning and intellectual development for all chil- does not include analysis of studies that do not meet mini-
dren. Literature supports the use of behavioral interventions mum rigor requirements. However, on further analysis of the
as effective treatments for the treatment of sleep rigor ratings of those studies rated as “weak”, we identified
problem.33,42,66 As children with autism are more likely to two key limitations of the Reichow rating scale for behavioral
have instances of sleep problems, evaluation of this literature interventions on sleep. First, most of the data in the included
is important to guide treatment and research. The purpose of studies was collected by parents in home environments, mak-
this quantitative-analysis was to synthesize and evaluate the ing inter-observer agreement difficult to collect. Secondly,
preliminary evidences supporting the effectiveness of beha- several studies had variable behavior in baseline, which is an
vioral interventions reported in the literature for the treat- indicator of sleep problems and is to be expected for this data
ment of sleep problems for children with ASD. set. Further statistical analysis, such as moderating effects of
The first aim of this quantitative-analysis was to synthesize additive interventions and interaction effects between specific
the literature to identify the most commonly treated sleep sleep problems would benefit the findings of this review, but
problems. Of the sleep problems addressed in the literature, would require larger groups of homogeneous studies. For
night wakings was most commonly treated problem, followed example, in the current analysis, the majority of studies relied
by bedtime disturbances. Previous reviews as well as parent exclusively on parental reports of sleep problems (i.e., sleep
and practitioner report support these findings.32,67 diaries and questionnaires) to assess treatment outcomes.
The second aim of this quantitative-analysis was to identify Clearer and more precise measurement of dependent vari-
the most common intervention components employed in the ables is needed to ensure objective measurement. Given the
extant literature. For interventions reported in the literature, availability of technology and the advances of equipment used
all the studies utilized multiple treatment components, most for sleep monitoring (e.g., actigraphy and videography) objec-
often including the use of a consistent bedtime routine, and tive measures of data recording can be used to triangulate
an extinction-based procedure. Generally, these findings sup- parental reports of sleep patterns. Furthermore, the use of
port the overall effectiveness of behavioral interventions for such technologies can allow for more accurate detection of
the treatment of sleep problems for this population. Previous sleep problems (e.g., quiet awakenings unknown to parents)
reviews of pediatric sleep interventions including studies tar- and the analysis of parent–child interactions used during the
geting children with autism have identified similar interven- assessment of sleep problem behavior. It is important that
tion components and overall effectiveness ratings68, but to our future research incorporates the use of such technology, and
knowledge no prior study has mathematically examined the also evaluates the feasibility and social validity of such
effectiveness of behavioral interventions for this specific measures.
population. Both a non-parametric overlap estimate (i.e., Tau-U)65 and
The third aim of this quantitative-analysis was to evaluate a between case fixed effect size (i.e., hedges g)46 indicated
the strength of the intervention effects using statistical effect a moderate effect of this body of research. Despite an increase
size calculations. Analysis of these results indicates a moderate of evidence in the use of non-overlap and parametric effect
effect size; however, due to the small number of studies, this is size estimates for the analysis and quantitative-analysis of
limited in comparison. Further, despite the general conclusion single-case design46,65,70, there are still barriers to accurate
related to overall effectiveness, a direct comparison of the description of the effects of single-case design using these
findings is not completely possible because of the variability measures. Non-overlap indexes feature the limitation of not
in the sleep interventions reported and the dependent vari- having a reliable measurement of the magnitude of those
ables. Due to the variety of target behaviors and the different measured change in level or trend typically indicated by
treatment packages between studies, it was not possible to single-case design (e.g., a very small change in level could
identify best practices for particular sleep problems. lead to a 1.0 Tau-U score due to zero rates of overlapping
However, these results suggest preliminary evidence that data). Parametric estimates of the effect also have limitations.
effective interventions are available for the most commonly Single-case designs typically measure behavior change that is
defined sleep problems. the product of intensive, targeted, individualized intervention,
and change in the targeted behavior is measured with direct
behavior measurement (i.e., frequency count, partial interval
Limitations and Directions for Future Research
recording). Effect sizes like hedges g are designed to capture
One of the major limitations of this review is the limited the magnitude of the variance between treatment and control
evaluation of methodological rigor. Due to the age of the groups that are likely more homogeneous, maybe receiving
literature base and variations between studies, our review of a more diffuse standardized intervention, and whose progress
quality is limited. Sleep is necessarily studied in applied con- is generally measured using standardized measures. Further,
texts and is a behavior with high rates of variability, even in although hedges g allows for the estimation of effect size even
typical populations, without sleep interfering behaviors given a small population, single-case designs are smaller n’s
12 A. CARNETT ET AL.

than in typical small n group research. The result is that of these interventions in terms of maintenance of sleep beha-
d-estimates for single case often appear inflated, as with the vior. Since quality sleep is important for overall functioning
d-estimate for Durand and colleagues (g = 3.45)66, which is and development, it would seem highly beneficial for research
outside the parameters of analysis. For these reasons, results to evaluate procedures that yield positive effects over time.
of statistical analysis of single-case design should be inter- Additionally, there may also be the need for analysis of cases
preted with caution and used as a supplement only to gold- where resurgence of sleep problems occurs and effective treat-
standard visual analysis. Future analyses of this literature base ments for such occurrences.
should consider statistical methods that have the technology Lastly, the overall scope of this quantitative is limited in
to compare single case and group designs such as hierarchical that it was focused on the use of behavioral interventions,
linear modeling. In addition, we did not conduct a risk of bias rather than encompassing all available interventions (e.g.,
measure for the included literature base, mostly due to the medical, pharmalogical, alternative medicine) for the treat-
overall low rigor of the included research. Future analyses of ment of sleep problems. Thus, further analyses of the litera-
this literature base as it grows and improves should be mind- ture, such as medical treatments and/or alternative treatments
ful of potential effects of bias on the reporting of findings in should be conducted to evaluate the differential effects. Future
this area. quantitative analyses should consider both the differential
Despite these limitations associated with the statistical effects between medical or behavioral treatments, as well as
analysis, it is important to highlight the overall effectiveness their potential interaction. Pending an updated body of litera-
of behavior analytic assessment and treatment for children ture on this topic, future analyses should also consider the
with ASD. Compared to more common treatments, such as weighted effect of intervention components in order to iden-
the use of pharmaceuticals, there is limited research on the tify the most efficacious treatment package. Finally, grey lit-
clinical guidance needed and long-term efficacy.34 As such, it erature was not included in the current review, so emergent
is important to address limitations within the behavior analy- interventions featured in dissertations or other non-peer
tical research related to sleep problems. First, given the like- reviewed scholarly work could have been overlooked.
lihood that sleep interventions most often occur in home
environments and are implemented by the parents or care-
givers, it is important to evaluate procedures that are feasible References
to implement, as well as the social validity of these proce- 1. Chu J, Richdale AL. Sleep quality and psychological wellbeing in
dures. However, only about half to the studies evaluated in mothers of children with developmental disabilities. Res Develop
this quantitative-analysis have assessed these measures related Disabil. 2009 Jul 10;30(6):1512–22. doi:10.1016/j.ridd.2009.07.007.
to social validity. Even further, to date research has not 2. Richdale AL, Schreck KA. Sleep problems in autism spectrum
assessed issues related to parent stress or other factors that disorders: prevalence, nature, & possible biopsychosocial aetiolo-
gies. Sleep Med Rev. 2009 Dec 1;13(6):403–11. doi:10.1016/j.
might contribute to the offer all effectiveness of interventions smrv.2009.02.003.
conducted in the home environment. Thus, it is important for 3. Singh K, Zimmerman AW. Sleep in autism spectrum disorder and
future research to include parents and caregivers during the attention deficit hyperactivity disorder. Semin Pediatr Neuro.
assessment and development of intervention procedures and 2015 Jun 1;22(2):113–25. doi:10.1016/j.spen.2015.03.006.
to include feedback related to the social validity of the selected 4. Brown CA, Kuo M, Phillips L, Berry R, Tan M. Non-
pharmacological sleep interventions for youth with chronic health
interventions. conditions: a critical review of the methodological quality of the
Second, it is important that adequate assessments are com- evidence. Disabil Rehabil. 2013 Jul 1;35(15):1221–55. doi:10.3109/
pleted that isolated the establishing operations and controlling 09638288.2012.723788.
variables associated to the sleep problem since there is a lack 5. Krakowiak P, Goodlin-Jones BE, Hertz-Picciotto IR, Croen LA,
of consistency within the literature on assessment measures Hansen RL. Sleep problems in children with autism spectrum
disorders, developmental delays, and typical development:
and procedures. For example, a functional analysis is the gold- a population-based study. J Sleep Res. 2008 Jun 1;17(2):197–206.
standard in behavioral interventions for the assessment pro- doi:10.1111/j.1365-2869.2008.00650.x.
blem behavior.70 A functional analysis involves the systematic 6. Buckley AW, Rodriguez AJ, Jennison K, Buckley J, Thurm A,
evaluation of the environmental variables maintaining pro- Sato S, Swedo S. Rapid eye movement sleep percentage in children
blem behavior and has been proven to improve intervention with autism compared with children with developmental delay
and typical development. Arch Pediatr Adolesc Med. 2010 Nov
effects. Some limited research supports the use of functional 1;164(11):1032–37. doi:10.1001/archpediatrics.2010.202.
analysis technology to evaluate sleep as a moderator of day- 7. Cortesi F, Giannotti F, Ivanenko A, Johnson K. Sleep in children
time challenging behavior71,72, but future research might with autistic spectrum disorder. Sleep Med Res. 2010 Aug 1;11
incorporate this technology into the assessment and treatment (7):659–64. doi:10.1016/j.sleep.2010.01.010.
of sleep behaviors. 8. Deliens G, Leproult R, Schmitz R, Destrebecqz A, Peigneux P.
Sleep disturbances in autism spectrum disorders. J Autism Dev
Third, studies included in this review frequently used Disord. 2015 Dec 1;2(4):343–56. doi:10.1007/s40489-015-0057-6.
multi-component treatment procedures. Thus, it is unclear 9. Hodge D, Carollo TM, Lewin M, Hoffman CD, Sweeney DP.
what elements are essential for desired behavior change. Sleep patterns in children with and without autism spectrum
Further, given parents and caregivers are likely to be respon- disorders: developmental comparisons. Res Dev Disabil. 2014 Jul
sible for the implementation of these procedures, this is of 1;35(7):1631–38. doi:10.1016/j.ridd.2014.03.037.
10. Goldman SE, McGrew S, Johnson KP, Richdale AL, Clemons T,
particular importance. Future research is needed to determine Malow BA. Sleep is associated with problem behaviors in children
the most essential and most efficacious intervention compo- and adolescents with autism spectrum disorders. Res Autism Spectr
nents. Further, research should evaluate the long-term effects Disord. 2011 Jul 1;5(3):1223–29. doi:10.1016/j.rasd.2011.01.010.
DEVELOPMENTAL NEUROREHABILITATION 13

11. Gail Williams P, Sears LL, Allard A. Sleep problems in children handbook of infant, child, and adolescent sleep and behavior.
with autism. J Sleep Res. 2004 Sep 1;13(3):265–68. doi:10.1111/ New York (NY): Oxford University Press; 2013. p. 471–94.
j.1365-2869.2004.00405.x. 29. Durand VM, Christodulu KV. Description of a sleep-restriction
12. Hollway JA, Aman MG, Butter E. Correlates and risk markers for program to reduce bedtime disturbance and night waking. J Pos
sleep disturbance in participants of the autism treatment network. Behav Interven. 2004;19:130–39.
J Autism Dev Disord. 2013 Dec 1;43(12):2830–43. doi:10.1007/ 30. Rosen CL, Owens JA, Scher MS, Glaze DG. Pharmacotherapy for
s10803-013-1830-y. pediatric sleep disturbances: current patterns of use and target
13. Liu X, Hubbard JA, Fabes RA, Adam JB. Sleep disturbances and populations for controlled clinical trials. Current Therap Res.
correlates of children with autism spectrum disorders. Child 2002 Jan 1;63:B53–B66. doi:10.1016/S0011-393X(02)80103-5.
Psychiat Hum Dev. 2006 Dec 1;37(2):179–91. doi:10.1007/ 31. Christodulu KV, Durand VM. Reducing bedtime disturbance and
s10578-006-0028-3. night waking using positive bedtime routines and sleep
14. Malow BA, Marzec ML, McGrew SG, Wang L, Henderson LM, restriction. Focus Autism Other Dev Disabil. 2004 Aug;19
Stone WL. Characterizing sleep in children with autism spectrum (3):130–39. doi:10.1177/10883576040190030101.
disorders: a multidimensional approach. J Sleep Res. 2006 Dec 32. Weiskop S, Matthews J, Richdale A. Treatment of sleep problems in a
1;29(12):1563–71. doi:10.1093/sleep/29.12.1563. 5-year-old boy with autism using behavioural principles. Autism.
15. Paavonen EJ, Nieminen-von Wendt T, Vanhala R, Aronen ET, 2001 Jun;5(2):209–21. doi:10.1177/1362361301005002009.
von Wendt L. Effectiveness of melatonin in the treatment of sleep 33. Mindell JA, Kuhn B, Lewin DS, Meltzer LJ, Sadeh A. Behavioral
disturbances in children with Asperger disorder. J Child Adolesc treatment of bedtime problems and night wakings in infants and
Psychopharm. 2003 Mar 1;13(1):83–95. doi:10.1089/ young children. Sleep Res. 2006;29:1263–76.
104454603321666225. 34. Richdale A, Wiggs L. Behavioral approaches to the treatment of
16. Schreck KA, Mulick JA, Smith AF. Sleep problems as possible sleep problems in children with developmental disorders: what is
predictors of intensified symptoms of autism. Res Dev Disabil. the state of the art? Int J Behav Consult Ther. 2005;1(3):165.
2004 Jan 1;25(1):57–66. doi:10.1037/h0100743.
17. Schwichtenberg AJ, Young GS, Hutman T, Iosif AM, Sigman M, 35. Vriend JL, Corkum PV, Moon EC, Smith IM. Behavioral inter-
Rogers SJ, Ozonoff S. Behavior and sleep problems in children ventions for sleep problems in children with autism spectrum
with a family history of autism. Autism Res. 2013 Jun 1;6 disorders: current findings and future directions. J Pediatr
(3):169–76. doi:10.1002/aur.1278. Psychol. 2011 Jul 9;36(9):1017–29. doi:10.1093/jpepsy/jsr044.
18. Doo S, YK W. Sleep problems of children with pervasive develop- 36. Williams CD. The elimination of tantrum behavior by extinction
mental disorders: correlation with parental stress. Dev Med Child procedures. J Abnorm Soc Psychol. 1959 Sep;59:269. doi:10.1037/
Neurol. 2006 Aug;48(8):650–55. doi:10.1017/S001216220600137X. h0046688.
19. Hoffman CD, Sweeney DP, Lopez-Wagner MC, Hodge D, 37. Blampied NM, France KG. A behavioral model of infant sleep
Nam CY, Botts BH. Children with autism: sleep problems and disturbance. J App Behav Anal. 1993 Dec 1;26(4):477–92.
mothers’ stress. Focus Autism Other Dev Disabil. 2008 Sep;23 doi:10.1901/jaba.1993.26-477.
(3):155–65. doi:10.1177/1088357608316271. 38. Blampied NM. Functional behavioral analysis of sleep in infants
20. Meltzer LJ, Mindell JA. Relationship between child sleep distur- and children. In: Montgomery-Downs HE, Wolfson AR, editors.
bances and maternal sleep, mood, and parenting stress: a pilot The Oxford handbook of infant, child, and adolescent sleep and
study. J Fam Psychol. 2007 Mar;21(1):67. doi:10.1037/0893- behavior. New York (NY): Oxford University Press; 2013. p.
3200.21.1.67. 169–88.
21. Foody C, James JE, Leader G. Parenting stress, salivary biomar- 39. Didden R, Curfs LM, van Driel S, de Moor JM. Sleep problems in
kers, and ambulatory blood pressure in mothers of children with children and young adults with developmental disabilities:
Autism Spectrum Disorders. Res Autism Spectr Disord. 2014 Feb home-based functional assessment and treatment. J Behav Ther
1;8(2):99–110. doi:10.1016/j.rasd.2013.10.015. Exp Psychiatry. 2002 Mar 1;33(1):49–58. doi:10.1016/S0005-
22. Meltzer LJ. Factors associated with depressive symptoms in parents 7916(02)00012-5.
of children with autism spectrum disorders. Res Autism Spectr 40. Owens LJ, France KG, Wiggs L. Behavioural and
Disord. 2011 Jan 1;5(1):361–67. doi:10.1016/j.rasd.2010.05.001. cognitive-behavioural interventions for sleep disorders in infants
23. Tilford JM, Payakachat N, Kuhlthau KA, Pyne JM, Kovacs E, and children: a review. Sleep Med Rev. 1999 Dec 1;3(4):281–302.
Bellando J, Williams DK, Brouwer WB, Frye RE. Treatment for doi:10.1053/smrv.1999.0082.
sleep problems in children with autism and caregiver spillover 41. Johnson CR, Turner KS, Foldes EL, Malow BA, Wiggs L.
effects. J Autism Dev Disord. 2015 Nov 1;45(11):3613–23. Comparison of sleep questionnaires in the assessment of sleep dis-
doi:10.1007/s10803-015-2507-5. turbances in children with autism spectrum disorders. Sleep Med
24. Giallo R, Wood C, Jellett R, Porter R. Fatigue, wellbeing and Rev. 2012 Aug 1;13(7):795–801. doi:10.1016/j.sleep.2012.03.005.
parental self-efficacy in mothers of children with an Autism 42. Jin CS, Hanley GP, Beaulieu L. An individualized and compre-
Spectrum Disorder, Autism: international. Autism J Res Prac. hensive approach to treating sleep problems in young children.
2011:465-480. J Appl Behav Anal. 2013 Mar 1;46(1):161–80. doi:10.1002/jaba.16.
25. Davis NO, Carter AS. Parenting stress in mothers and fathers of 43. Reichow B. Overview of quantitative-analyses on early intensive
toddlers with autism spectrum disorders: associations with child behavioral intervention for young children with autism spectrum
characteristics. J Autism Dev Disord. 2008 Aug 1;38(7):1278. disorders. J Autism Dev Disord. 2012 Apr 1;42(4):512–20.
doi:10.1007/s10803-007-0512-z. doi:10.1007/s10803-011-1218-9.
26. Malow B, Adkins KW, McGrew SG, Wang L, Goldman SE, 44. Virués-Ortega J, Julio FM, Pastor-Barriuso R. The TEACCH
Fawkes D, Burnette C. Melatonin for sleep in children with aut- program for children and adults with autism: A
ism: a controlled trial examining dose, tolerability, and outcomes. quantitative-analysis of intervention studies. Clin Psychol Rev.
J of Autism Dev Disord. 2012 Aug 1;42(8):1729–37. doi:10.1007/ 2013 Dec 1;33(8):940–53. doi:10.1016/j.cpr.2013.07.005.
s10803-011-1418-3. 45. Virués-Ortega J. Applied behavior analytic intervention for aut-
27. Malow BA, MacDonald LL, Fawkes DB, Alder ML, Katz T. ism in early childhood: quantitative-analysis, quantitative-
Teaching children with autism spectrum disorder how to sleep regression and dose–response quantitative-analysis of multiple
better: A pilot educational program for parents. Clinic Practice outcomes. Clin Psychol Rev. 2010 Jun 1;30(4):387–99.
Pedia Psych. 2016 Jun;4(2):125. doi:10.1037/cpp0000138. doi:10.1016/j.cpr.2010.01.008.
28. Richdale AL. Autism and other Developmental Disabilities. In: 46. Shadish WR, Hedges LV, Pustejovsky JE. Analysis and
Montgomery-Downs HE, Wolfson AR, editors. The Oxford quantitative-analysis of single-case designs with a standardized
14 A. CARNETT ET AL.

mean difference statistic: A primer and applications. J Sch 59. Weiskop S, Richdale A, Matthews J. Behavioural treatment to
Psychol. 2014 Apr 1;52(2):123–24. doi:10.1016/j.jsp.2013.11.005. reduce sleep problems in children with autism or fragile X
47. Baer DM, Wolf MM, Risley TR. Some current dimensions of syndrome. Dev Med C Neurol. 2005 Feb;47(2):94–104.
applied behavior analysis 1. J Appl Behav Analy. 1968 Mar;1 doi:10.1017/S0012162205000186.
(1):91–97. doi:10.1901/jaba.1968.1-91. 60. Reichow B, Volkmar FR, Cicchetti DV. Development of the eva-
48. Gringras P, Green D, Wright B, Rush C, Sparrowhawk M, Pratt K, luative method for evaluating and determining evidence-based
Allgar V, Hooke N, Moore D, Zaiwalla Z, et al. Weighted blankets practices in autism. Autism Devel Dis. 2008 Aug 1;38
and sleep in autistic children—A randomized controlled trial. (7):1311–19. doi:10.1007/s10803-007-0517-7.
Pedia. 2014 Jul 1:peds–2013;134. 61. DL G, JR L, editors. Single subject research methodology in
49. Williamson AA, Patrick KE, Rubens SL, Moore M, Mindell JA. behavioral sciences. Routledge; 2009.
Pediatric sleep disorders in an outpatient sleep clinic: clinical 62. Lang R, O’Reilly M, Healy O, Rispoli M, Lydon H,
presentation and needs of children with neurodevelopmental Streusand W, Giesbers S. Sensory integration therapy for
conditions. Clinic Prac Pedia Psych. 2016 Jun;4(2):188. autism spectrum disorders: A systematic review. Res Autism
50. Durand VM, Gernert-Dott P, Mapstone E. Treatment of sleep Spec Disord. 2012 Jan;3:1004–18. doi:10.1016/j.
disorders in children with developmental disabilities. J Assoc Pers rasd.2012.01.006.
Severe. 1996 Sep;21(3):114–22. 63. Parker RI, Vannest KJ, Davis JL. Effect size in single-case research:
51. Friedman A, Luiselli JK. Excessive daytime sleep: behavioral A review of nine nonoverlap techniques. Behav Modif. 2011;35
assessment and intervention in a child with autism. Behav (4):303–22. doi:10.1177/0145445511399147.
Modif. 2008 Jul;32(4):548–55. doi:10.1177/0145445507312187. 64. Rakap S, Rakap S, Evran D, Cig O. Comparative evaluation of
52. Knight RM, Johnson CM. Using a behavioral treatment package the reliability and validity of three data extraction programs:
for sleep problems in children with autism spectrum disorders. unGraph, graphclick, and digitizeit. Comput Hum Behav.
Child Fam Behav Ther. 2014 Jul 1;36(3):204–21. doi:10.1080/ 2016 Feb;1(55):159–66. doi:10.1016/j.chb.2015.09.008.
07317107.2014.934171. 65. Parker RI, Vannest KJ, Davis JL, Sauber SB. Combining nonover-
53. Loring WA, Johnston R, Gray L, Goldman S, Malow B. A brief lap and trend for single-case research: tau-U. Behav Ther. 2011
behavioral intervention for insomnia in adolescents with autism Jun 1;42(2):284–99. doi:10.1016/j.beth.2010.08.006.
spectrum disorders. Clin Prac in Ped Psych 2016; 4(2):112. 66. Durand VM. Treating sleep terrors in children with autism.
doi:10.1037/cpp0000141 J Posit Behav Interv. 2002 Apr 4;(2):66–72. doi:10.1177/
54. Malow B, Adkins KW, Reynolds A, Weiss SK, Loh A, Fawkes D, Katz 109830070200400201.
T, Goldman SE, Madduri N, Hundley R, Clemons T. Parent-based 67. Souders MC, Zavodny S, Eriksen W, Sinko R, Connell J, Kerns C,
sleep education for children with autism spectrum disorders. J of Schaaf R, Pinto-Martin J. Sleep in children with autism spectrum
Autism Dev Disord. 2014 Jan;44(1):216-22. doi:10.1007/s10803-013- disorder. Curr Psychiatry Rep. 2017 Jun 1;19(6):34. doi:10.1007/
1866-z s11920-017-0782-x.
55. McLay L, France K, Blampied N, Danna K, Hunter J. Using func- 68. Horner RH, Kratochwill TR. Synthesizing single-case research
tional behavioral assessment to develop a multicomponent treatment to identify evidence-based practices: some brief reflections.
for sleep problems in a 3-year-old boy with autism. Clin Case Stud. J Behav Ed. 2012 Sep 1;21(3):266–72. doi:10.1007/s10864-012-
2017 Jun;16(3):254–70. doi:10.1177/1534650116688558. 9152-2.
56. Moon EC, Corkum P, Smith IM. Case study: a case-series evalua- 69. Mindell JA, Owens JA. A clinical guide to pediatric sleep: diag-
tion of a behavioral sleep intervention for three children with nosis and management of sleep problems. Lippincott Williams &
autism and primary insomnia. J Pediatr Psychol. 2010 Jul 14;36 Wilkins; 2015 May 4.
(1):47–54. doi:10.1093/jpepsy/jsq057. 70. Hanley GP, Iwata BA, McCord BE. Functional analysis of pro-
57. Piazza CC, Fisher WW, Sherer M. Treatment of multiple sleep blem behavior: A review. J Appl Behav Anal. 2003 Jun 1;36
problems in children with developmental disabilities: faded bed- (2):147–85. doi:10.1901/jaba.2003.36-147.
time with response cost versus bedtime scheduling. Dev Med 71. Kennedy CH, Meyer KA. Sleep deprivation, allergy symptoms,
Child Neurol. 1997 Jun 1;39(6):414–18. doi:10.1111/j.1469- and negatively reinforced problem behavior. J Appl Behav Anal.
8749.1997.tb07456.x. 1996 Mar 1;29(1):133–35. doi:10.1901/jaba.1996.29-133.
58. Reed HE, McGrew SG, Artibee K, Surdkya K, Goldman SE, Frank 72. O’Reilly MF. Functional-analysis and treatment of
K, ... & Malow, BA. Parent-based Sleep Education Workshops in escape-maintained aggression correlated with sleep-deprivation.
Autism. J Child Neuro. 2009 Aug;24(8):936–945. J Appl Behav Anal. 1995;28:225–26. doi:10.1901/jaba.1995.28-225.

You might also like