Sleep Medicine 53 (2019) 141e152

Contents lists available at ScienceDirect

Sleep Medicine
journal homepage: www.elsevier.com/locate/sleep

Review Article

Efficacy of parent training incorporated in behavioral sleep

interventions for children with autism spectrum disorder and/or
intellectual disabilities: a systematic review
Bernadette Kirkpatrick*, Julia S. Louw, Geraldine Leader
National University of Ireland Galway, Galway, Ireland

a r t i c l e i n f o a b s t r a c t

Article history: Objective: Behavioral sleep interventions are regularly used to improve sleep problems experienced by
Received 28 March 2018 children with autism spectrum disorder (ASD) and/or intellectual disability (ID). Recent developments
Received in revised form have seen the introduction of parent sleep education and healthy sleep practice training to sleep in-
8 August 2018
terventions. This article aims to systematically review the evidence on the efficacy of parent training that
Accepted 29 August 2018
Available online 11 October 2018
is incorporated within recent sleep interventions for children with ASD and/or ID.
Method: Electronic databases and manual searches of reference lists identified 11 studies (n ¼ 416
children) that met the inclusion criteria.
Keywords:
Autism spectrum disorder
Results: The evidence presented in this systematic review would suggest that the inclusion of parent
Intellectual disabilities training within behavioral sleep interventions for children with ASD and/or ID is generally effective and
Sleep disorders valued by parents. Nine of the 11 studies reviewed reported a reduction in sleep problems.
Behavioral interventions Conclusion: The literature conveys an emerging evidence-based practice that could contribute to future
Parent training behavioral sleep research and guide best-practice decisions to support effective parent training to
Sleep education improve sleep outcomes for children with ASD and/or ID.
© 2018 Elsevier B.V. All rights reserved.

1. Introduction [20]. Noterdaeme and Euders [35] found that individuals with ASD
can be diagnosed with or without an ID. It is evident from previous
Autism spectrum disorder (ASD) is defined as a pervasive neu- research that a person can have a diagnosis of ASD and/or ID.
rodevelopmental disorder which can be characterized by a triad of Considering that several sleep studies include children with ASD
persistent impairments with core deficits in social interaction, lan- and/or ID, sleep interventions within the current context were
guage and communication, as well as restrictive, repetitive thoughts, reviewed with a potential comorbidity between ASD and/or ID.
routines and behavior patterns [4]. Individuals with ASD can often Sleep is an essential aspect of life, and a high percentage of
experience several comorbid medical conditions including sleep children with ASD [26] and ID [40] experience sleep problems. It is
disorders [46]. An intellectual disability (ID) is defined as a disability reported that 40e80% of children with ASD and/or ID (ASD/ID)
which is characterised by an intellectual impairment that may present with sleep disturbances [38] and that 58% of children with
significantly impede mental capacity such as learning, reasoning, mild to profound ID experience sleep problems [40]. The abbre-
understanding which can affect social functioning and practical skills viated term “ASD/ID” used hereafter represents children with
[3]. Previous research has suggested there may be an overlap with either a single diagnosis of ASD or ID or a combined diagnosis of
intellectual disabilities and those diagnosed with ASD [27]. However, ASD and ID. Within the context of the current review, the terms
this may not always be the case, as researchers have found that due ‘sleep difficulties’, ‘sleep problems’, and ‘sleep disturbance’ are
to the broad and varied nature of the spectrum of autism, some in- interchangeable and represent the sleep difficulties commonly
dividuals with ASD may not have impaired intellectual functioning reported by parents of children with ASD/ID. These include
adhering to bedtime routines, sleep initiation, frequent nighttime
waking, early morning waking, and co-sleeping with parents.
* Corresponding author. Irish Centre for Autism and Neurodevelopmental Sleep disturbances are distressing for the child and their families
Research, School of Psychology, National University of Ireland, Galway, Ireland. and often result in reduced daily functioning [25]. Sleep
E-mail address: Bernadette.kirkpatrick@nuigalway.ie (B. Kirkpatrick).

https://doi.org/10.1016/j.sleep.2018.08.034
1389-9457/© 2018 Elsevier B.V. All rights reserved.
142 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152

deprivation can exacerbate the symptoms of ASD/ID, which may training and behavioral strategies with three children with ASD. It
result in challenging daytime behaviors [23]. There is a correlation could be argued that knowledge of sleep education, behavioural
between sleep problems and increased aggression, noncompli- strategies and guidance on how to implement them within the
ance, increased social skills deficits, an increase in emotional be- home environment was invaluable to achieving the positive out-
haviors, deficits in daily life skills [41], and a decrease in comes reported by Moon et al., [31]. Vriend et al., concluded that
communication skills [19]. Insufficient sleep has a profound there was evidence to suggest that some behavioral interventions
negative effect on daily activities and social inclusion for those focusing on sleep problems were possibly effective for children
affected. Disrupted sleep patterns experienced by children with with ASD/ID. This conclusion led them to advocate the need for
ASD/ID and the subsequent negative impact on their families can further research to examine the effectiveness of evidence-based
result in a reduced quality of life for the family unit. Furthermore, sleep interventions for children with ASD/ID, using larger samples
sleep problems in children with or without ASD/ID are associated within rigorously controlled designs.
with increased parental stress, reduced parental sense of compe- Malow et al., [24] published a review on the identification,
tence, and poor physical health for the family unit [32]. evaluation, and management of insomnia in children and adoles-
Individuals with ASD/ID require effective user-friendly sleep in- cents with ASD. They included behavioral, alternative, and phar-
terventions that meet their diverse needs. “User-friendly” within the macological publications dated from 2000 to 2011. Only one of
current context refers to well-designed individualized behavioral these studies [37] was relevant to the current review. Reed et al.,
strategies that parents or carers/caregivers find easy to implement [37] had a within-subject design that implemented a multi-
within the home and family routines. Sleep interventions can be component behavioral parent training programme to improve a
broadly categorized into three approaches: namely, pharmacological, wide range of sleep problems experienced by children with ASD.
behavioral, or combined pharmacological and behavioral. Pharma- They [37] evaluated the use of three face-to-face group workshops
cological interventions aim to increase sleep duration and to with three to five families per session, in which they used a stan-
decrease sleep latency. However, sleep-promoting medications may dardized training programme with opportunities for individuali-
have numerous side effects and could have an adverse interaction zation. Sleep improvements were assessed using questionnaires
with other medications [12]. It is suggested that melatonin may be an and actigraphy. Results demonstrated a reduction in the target
effective pharmacological approach for children with neuro- behaviors of bedtime resistance, initiating sleep, night waking, or
developmental disorders with minimal adverse effects [5]. Mela- early waking in many of the children. These findings may suggest
tonin treatment may benefit children with developmental disorders that behavioral parent training, if utilized and maintained, could be
such as ASD/ID to decrease sleep onset latency and to increase total a suitable and effective addition to enhance parents’ confidence
sleep time; nonetheless, it is argued that it will not decrease night- and ability to apply behavioral sleep intervention procedures.
time awakenings [9]. As the current review was not investigating the Malows et al., concluded that parent sleep education training
efficacy of pharmacological sleep treatments, those studies that were combined with behavioral techniques should be the first approach
purely pharmacological were excluded. to improving sleep difficulties in children with ASD.
Research has suggested that behavioral interventions are A third review, conducted by Meltzer and Mindell [28], on
effective for improving a range of sleep problems experienced by behavioral interventions for paediatric insomnia focused mainly on
individuals with developmental disorders [18,23,39]. Behavioral typically developing children and included publications from 1985
interventions can include a range of procedures to include sleep to 2013. Although their review included two studies with a special
hygiene, reinforcement of desirable behavior, ignoring undesirable needs population, only one of these studies [1] was applicable for
behaviors, and environmental changes. They encourage the initia- inclusion within the current systematic review. In conclusion,
tion and maintenance of sleep and are recommended as the first Meltzer and Mindell highlighted the need for further evidence on
approach to resolve sleep disturbances in children [29]. Behavioral the efficacy of behavioral interventions for pediatric insomnia.
interventions aim to increase the frequency of behaviors that are Considering the evidence presented in the three previously
crucial to improving sleep and to decrease the behaviors that are summarized reviews, the time frame for the current systematic
detrimental to sleep. Many sleep interventions for children with or review was set between 2010 and 2016. Comprehensive electronic
without ASD/ID are implemented within the home environment by database searches for the six-year time frame for the current re-
parents. Sleep interventions can be time consuming and involve view identified 11 new publications that included parent training.
ignoring unwanted bedtime behaviors which the parent may find Notably, the number of studies published had increased since the
stressful, such as the child crying out. It is important that parents Vriend et al., [47] review, in which, over a similar time frame of six
are given the knowledge, support, and appropriate training on how years from 2004 to 2010, five publications were cited that included
to deal with undesirable bedtime behaviors (eg, how to change the an element of parent training. This could possible signify a change
environment and how to deliver timely reinforcement to enhance in the trend towards an increase in behavioral sleep interventions
the effectiveness of the behavioral sleep intervention). Behavioral that include parent training.
interventions incorporating parent training are increasing, and in
recent years parent training has been introduced to enhance the 1.1. Rationale and objectives for current review
treatment of sleep problems for children with ASD/ID [6,32,42,45].
Several reviews have been published relating to insomnia in There is a need for the identification and synthesis of out-
children with ASD/ID and atypically developing children. However, comes from evidence-based behavioral practices using parent
only three of the most recent reviews included a few studies that training to improve sleep in children with ASD/ID. It is equally
were relevant to the current systematic review inclusion criteria of important to evaluate the quality and mode of delivery and to
parent training. Vriend et al., [47] published a review on the reflect on the variations in the methods employed within these
effectiveness of 15 behavioral interventions for sleep problems in interventions. In doing so, knowledge, impact, and societal value
children with ASD with studies dating from 1964 to 2010. Five of are all enhanced, enabling clinicians and families to make
the studies included by Vriend et al., published between 2004 and informed choices. Further to this, the current review may inform
2010, incorporated an element of parent training or sleep educa- and guide future research directions and clinical practice in the
tion. One study [31] was of interest to the current review, wherein management of pediatric sleep problems. This article presents the
the researchers conducted sleep interventions using parent first systematic review to focus on parent training incorporated
 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152 143

within behavioral sleep interventions for children with ASD/ID. In Index [13]. The Downs and Black Quality Index [13] has been shown
addition, it updates the existing review literature by including to provide a profile of randomised and non-randomized studies. It
studies published between 2010 and 2016. Thus, the goal of this has been extensively used by reviewers to highlight the method-
article was to identify and to systematically review the evidence ological strengths and weaknesses of health care studies. The scale
on the efficacy of current behavioral interventions that include consists of five subscales to include reporting (10 items), external
parent training in the treatment of sleep problems experienced validity (three items), bias (seven items), confounding (six items),
by children with ASD/ID. and power (one item). The items are answered as ‘yes’, ‘no’,
‘partially’, or ‘unable to determine’ depending on the subscale.
2. Method Responses are scored 0, 0, or 1, except for question 5 (reporting
subscale) which can be scored 0, 1, or 2, and question 27 (power
2.1. Eligibility criteria subscale), which can be scored 0, 1, 2, 3, 4, or 5. A maximum overall
score of 32 can be achieved, with higher scores indicating higher
Upon receipt of ethical approval from the NUI Galway Research quality. The index can also be scored and summed to produce five
Ethics Committee, the following inclusion criteria were imple- subscale scores. Internal consistency for the Quality Index is high
mented: (1) included a sample that was representative of children (KR-20 ¼ 0.89) with RCTs (KR-20 ¼ 0.92) and non-RCTs (KR-
with ASD and/or ID who experienced sleep problems; (2) com- 20 ¼ 0.88). Testretest reliability (r ¼ 0.88, p ¼ 0.90), with RCTs
bined behavioral strategies with parent training, parent-mediated (r ¼ 0 0.76, p ¼ 1.00) and non-RCTs (r ¼ 0.7, p ¼ 0.84) and interrater
intervention, or parent sleep education in the treatment of sleep reliability (r ¼ 0.75, p ¼ 0.056) of the Quality Index are good [13].
problems; (3) research designs included single-group design Interrater reliability (IRR) for the current review was calculated for
(SGD), randomized and nonrandomized controlled trials (RCTs and each subscale (reporting 93% IRR, external validity 96% IRR, internal
non-RCTs, respectively), single-subject research designs (SSRDs), validity bias 91% IRR, confounding and selection bias 97% IRR, and
and no experimental case studies using pre and post measure- power 100% IRR), and the resulting overall quality score (96% IRR)
ments; (4) published between January 2010 and November 2016; for each study (see Table 1 for reviewers' independent scores).
(5) published in peer-reviewed journals; and (6) published in the Although the reviewers' scores varied slightly on occasion, their
English language. Exclusion criteria were as follows (1) purely overall quality assessment score agreed as to which methodological
pharmacological sleep treatments; (2) studies that did not include quality category matched the study (100% IRR). The results indi-
parent sleep education, training, or parental mediated in- cated that nine studies were of high quality, three studies were of
terventions; (3) if the sample was not identified as ASD/ID; (4) if good quality, and that no studies were rated as of adequate or poor
the sleep intervention was implemented with an adult population; quality.
and (5) all reports, reviews, dissertations, and poster presentations
were excluded. 2.5. Data extraction

2.2. Search procedures Data extraction for all included studies was completed by the
first and second authors using an adapted version of the best-
During 2016, the three electronic databases of PsycINFO, practice data collection form for reviews incorporating a range of
PubMed, and Web of Science were searched to identify po- methodological designs (downloaded from http://epoc.cochrane.
tential studies. Keyword search terms included three areas org.resources) [14]. Data extraction included the number of par-
related to autism (autis* OR pervasive develop* OR Asperger* ticipants, age range, gender, sample population, that is, ASD and/or
AND learning disabilities OR intellectual disability*), sleep ID, diagnosis criterion, setting, recruitment method, and type of
disorders (sleep problems* OR sleep disorders OR sleep diffi- sleep problem reported. Along with study design (SGD, RCTs, non-
culties), and interventions (behavioral intervention AND RCTs, SSRDs, or case studies using pre and post measurements),
training OR parent* OR education OR mediated). The database type of parent involvement (parent training, sleep education, or
searches resulted in 2765 potential publications. Five additional parent-mediated intervention) and mode of training delivery
studies were identified by manually scanning the reference (telephone, face-to face, group, individual, workshop, or leaflets)
lists of the full-text publications which met the inclusion were noted. Data for attrition rates, treatment integrity, measure-
criteria for the current review. ments employed, and intervention outcomes were also extracted.
Interrater reliability for data extraction was high, at 94%. A sum-
2.3. Study selection mary of the extracted data are presented in Table 2.

After exporting all publications to a reference management 3. Results

software, 602 duplicated articles were removed and the remaining
2163 titles and abstracts were screened for eligibility. This resulted 3.1. Narrative synthesis of participants and study design
in the removal of a further 1749 studies based on the exclusion
criteria listed earlier. The remaining 416 full-text publications were Across the 11 studies, a total of 416 children and 414 parents
assessed for eligibility, and 11 studies were identified as meeting participated; in the eight studies that specified the child's gender,
the inclusion criteria listed earlier (see Fig. 1 for a flow diagram 188 of 250 children (75%) were male. Stuttard et al., [42] included
summarizing this process). The first and second authors indepen- young children aged 1e4 years with a mean age of three years.
dently screened the 11 studies using the inclusion and exclusion Most of the studies provided data on children with ages ranging
criteria for this review, and there was 100% agreement for the in- from 2 to 11 years with a mean age of five years, with exception of
clusion of all 11 studies. [32] and [42], which included children and adolescents aged 8e17
years with a mean age of 12 years, and 5e15 years with a mean age
2.4. Methodological quality of nine years, respectively. Ten of the studies included children
ASD/ID, whereas Allen et al [2] study was specific to Angelman
The first and second authors independently assessed the syndrome. Four of the 11 studies also included children with ID,
methodological quality of the 11 included studies using the Quality Down syndrome, cerebral palsy, learning disability, and decreased
144 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152

Fig. 1. Flow diagram depicting the identification and selection process for included review studies.

intellectual functioning with an IQ score of 70. A professional and training mode of delivery. Nonetheless, nine of the 11 studies
assessment and diagnosis were provided for all children included had similar aims to reduce sleep difficulties experienced by chil-
in the current review studies. dren with ASD/ID by means of behavioral sleep interventions
Of the 11 studies, four (36.3%) were RTCs, of which two included implemented by parents who were supported with parent training
waitlist control groups and two involved different treatment and/or sleep education materials.
groups. Of the remaining studies, three (27.3%) were single-group
design, two (18.2%) were comparison-group designs, one (9.1%) 3.2. Parent training programs and sleep outcomes
was a single-subject design, and one (9.1%) was a case series design
(Table 2). Methodological quality assessment indicated that nine In Adkins et al., [1], parents of children (n ¼ 36) with ASD, As-
studies were of high quality and three studies were of good quality, perger's and Pervasive Development Disorder-Not Otherwise
as summarized in Table 1. Specified (PDD-NOS) who experienced sleep onset latency of
Studies included in this review differed considerably in their 30 min or more were randomly stratified by their child's age into
experimental design, sample population, study duration, parent the treatment group (n ¼ 18) and wait list control group (n ¼ 18). A
contact time with professional support, parent training programs, sleep education pamphlet developed by the Autism Treatment
 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152 145

Table 1
First and second authors’ independent scores for the Downs & Black Quality Assessment Scale for included studies.

Authors Reporting Reporting External External Internal Internal Confounding Confounding Power Power Total Total
validity validity validity validity and selection and selection Quality Quality
ebias ebias bias bias Rating Rating
Score [13] Score [13]

First Second First Second First Second First Second First Second First Second

Moon et al. 10/11 11/11 3/3 3/3 4/7 4/7 4/6 4/6 1/5 1/5 22/32 Good 23/32
[31] Good
Adkins et al. 10/11 10/11 3/3 3/3 4/7 4/7 3/6 3/6 5/5 5/5 25/32 High 25/32
[1] High
Allen et al. 9/11 9/11 1/3 1/3 3/7 2/7 4/6 3/6 2/5 2/5 19/32 Good 17/32
[2] Good
Austin et al. 10/11 11/11 3/3 2/3 3/7 3/7 2/6 2/6 5/5 4/5 23/32 Good 22/32
[6] Good
Johnson et al. 11/11 11/11 3/3 3/3 6/7 7/7 6/6 6/6 5/5 5/5 31/32 High 32/32
[21] High
Malow et al. 11/11 11/11 3/3 3/3 6/7 7/7 6/6 6/6 5/5 5/5 31/32 High 32/32
[23] High
Moss et al. 10/11 11/11 3/3 3/3 6/7 6/7 6/6 6/6 5/5 5/5 30/32 High 31/32
[32] High
Fawkes et al. 10/11 11/11 3/3 3/3 6/7 7/7 5/6 6/6 5/5 5/5 29/32 High 32/32
[15] High
Stuttard, 10/11 11/11 3/3 3/3 4/7 5/7 3/6 3/6 5/5 5/5 25/32 High 27/32
Beresford High
et al. [42]
Stuttard, 10/11 11/11 3/3 3/3 5/7 5/7 4/6 4/6 5/5 5/5 27/32 High 28/32
Clarke et al. High
[42]
Veatch et al. 10/11 11/11 3/3 3/3 5/7 7/7 5/6 6/6 5/5 5/5 28/32 High 32/32
[45] High

First, first author; second, second author.

Network Sleep Committee was distributed to parents in the treat- to enhance the sleep environment, modify sleep schedules, and
ment group. Treatment group parents were instructed to read the alter parentchild interactions during bedtime routines and
pamphlet. No further support was given to the parents during the throughout the night resulted in immediate improvements in the
study. It was reported that the use of the pamphlet did not child's sleep patterns.
conclusively improve sleep within this sample. Feedback from Moon et al., [31] evaluated the effectiveness of the ‘Better
parents suggested that the pamphlet was informative but lacked Nights, Better Days’ parent handbook to reduce sleep onset latency
guidance on how to put the information into practice. in children (n ¼ 3) with ASD. The mean sleep onset latency
Two studies Moss et al., [32] and Austin et al., [6] investigated decreased for all three children; although the improvements were
the ‘Sleepwise’ program, with children (n ¼ 26) with ASD, Angel- relatively small, they were maintained at the follow-up assessment.
man Syndrome, Down Syndrome and ID [32] and with children (n However, for two of the three children, the average duration of
¼ 8) with ASD or PDD-NOS [6]. Moss et al., [32] used an RTC design sleep decreased slightly.
to compare a parent training workshop (n ¼ 13) with a wait list
control group (n ¼ 13), and reported improvements in sleep
problems for both groups, although the improvements were 3.3. Training delivery mode and professional support
significantly greater for the ‘Sleepwise’ treatment group than for
the wait list control group. In contrast, Austin et al., used a single- Within the current sample of studies, the parent training de-
group design to evaluate the same program and reported signifi- livery mode included group (n ¼ 4), individual (n ¼ 5), or self-
cant sleep improvements [6]. directed (n ¼ 2). Ten of the studies offered varying degrees of
Managing Your Child's Behavior to Promote Better Sleep professional support/contact throughout the intervention; how-
(MCBPBS) training manual [42], and the Parent Education Curric- ever, one study, by Adkins et al., offered no further support to
ulum Training program [23,45] were associated with positive out- parents after the distribution of the sleep education pamphlet [1].
comes. All three studies reported significant success in improving In Adkins et al., the self-directed support approach revealed that
sleep outcomes for their sample group. Stuttard et al., [42] further parents found the pamphlet informative but lacked guidance on
observed that parents' attitudes and confidence had improved after how to put the information into practice.
training. In addition, Veatch et al., [45], reported that the parent Fawkes et al., [15] compared the data from two previously
sleep education training resulted in closer correlations between the published studies that used different protocols. The first study used
subjective and objective sleep measures employed during the brief verbal training during an informed consent visit for children
study. (n ¼ 20) with ASD experiencing sleep problems [37]. The second
Allen et al., [2] found that a Behavioral Treatment Package (BTP) study delivered a 1-h structured hands-on training prior to
was effective for all children (n ¼ 5) with Angelman syndrome who commencement of the sleep intervention for children (n ¼ 80) with
experienced prolonged sleep difficulties. Each child learned to ASD, Asperger's disorder, and PDD-NOS [23]. Fawkes et al., focused
initiate falling asleep independently, and the duration of sleep was primarily on improving the scoring on the actigraphy and sleep
increased on average by 30 min per night. Parents also reported diary measurements, which are essential elements for ascertaining
that there was a marked improvement in their child's disruptive the efficacy of sleep interventions [15]. Although Fawkes et al., did
bedtime behaviors. Allen et al., concluded that enabling the parents not measure sleep outcomes directly, the study did produce
146 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152

Table 2
Summary of research studies included in the current systematic review.

Author/s Participants' age/ Design Sleep issues Measurement Intervention Results Quality Rating [13]
gender/diagnosis

Moon et al. Parents (n ¼ 3) & Case series Sleep onset Pre, Post & Follow Five weeks' self- Mean sleep onset Good
[31] children (n ¼ 3: 8 design. latency, sleep up. Actigraphy, delivered: Better latency was
e9 years: 2 males & duration & Sleep diaries, CSHQ, Nights, Better Days: reduced for all 3
1 female). Autism sleep efficiency. CBC & Parent Treatment for sleep children.
Spectrum Disorder. Satisfaction difficulties parent Maintained at 12
Questionnaire. handbook. weeks follow up.
Telephone support. Parents satisfied.
Behavioral Parent handbook
strategies: Faded considered to be
bedtime, Response effective.
Cost & Positive
reinforcement.
Adkins et al. [1] Parents (n ¼ 36) & RCT Sleep onset Pre, Post & Follow Distribution of Use of the High
children (n ¼ 36: 2 Sleep Education latency, sleep up. Actigraphy, Sleep education pamphlet did not
e10 years: 24 Pamphlet duration, sleep CSHQ & Sleep pamphlet conclusively
males and 12 (n ¼ 18) efficiency diaries, Stanford- developed by improve sleep
females). Autism Control Group &wake time Binet 5, Mullen Autism Treatment within this sample.
Spectrum Disorder, No Pamphlet after sleep Scales of Early Network Sleep Feedback from
Asperger's & (n ¼ 18). onset. Learning. Committee. Parents parents indicated
Pervasive received no further information was
Development support throughout valuable but
Disorder -Not the duration of the guidance on how to
Otherwise study. implement the
Specified. strategies would be
beneficial.
Allen et al. [2] Parents (n ¼ 5) & Multiple Sleep onset Pre, Post & Follow Parent mediated The BTP was found Good
children Baseline latency, sleep up. Actigraphy, Behavioral to be effective for
(n ¼ 5: 2e11years; SSRD. duration, sleep CSHQ & Sleep Treatment Package all 5 children who
3 females & 2 efficiency diaries, (BTP). To include had experienced
males). &wake time Developmental sleep environment, prolonged sleep
Angelman after sleep Behavior Checklist, sleep-wake problems.
syndrome. onset. Initiate Abbreviated schedule & parent/
sleep Acceptability child interactions.
independently. Rating Profile. Professional
support throughout
via telephone or
video contact.
Austin et al. [6] Parents (n ¼ 6: 2 Single group Night waking, Pre, Post & Follow A 15-week Sleepwise program Good
males & 4 females) (n ¼ 6) for Bedtime up. CSHQ, DBC-P, Sleepwise program was effective with
& children (n ¼ 8: 3 parent training resistance, co- Sleep diaries, consisted of 3 x 2-h all 8 children
e7 years: 8 males). & SSRD for sleeping, early Sleepwise Sleep workshops making significant
Autism Spectrum sleep waking & Disturbance Index, focusing on sleep improvements in
Disorder, Pervasive interventions. irregular sleep- Caregivers education and sleep problems.
Development wake cycle. Acceptance of behavioral
Disorder & PDD- Treatment Survey strategies. Along
Not Otherwise (CATS). with ongoing
Specified. support during
sleep interventions.
Johnson et al. Parents (n ¼ 40) & RCT Sleep onset Pre, 4 weeks & 8 Five individual Mixed results with Good
[21] children (n ¼ 40: 2 Behavior Parent delay, bedtime weeks. Actigraphy, sessions (60 the actigraphy not
e6 years). Gender Training resistance, Composite Sleep e90 min) over 8 detecting a
not reported. (n ¼ 20) night Index, Sleep diaries, weeks were significant
Autism Spectrum Psycho- awakening & Treatment fidelity delivered by 2 difference between
Disorder, Pervasive educational early morning checklist, Parent Board Certified the two groups.
Development training awakening. satisfaction. Behavior Analysts Although the BPT
Disorder -Not (n ¼ 20). and the third group indicated via
Otherwise therapist was the Composite
Specified. training with the Sleep Index that
BACB. The BPT there was a
included specific significant
bedtime and sleep improvement in
management bedtime and sleep
training whereas behaviors.
the PE training
related to ASD
diagnosis and
developmental
issues.
 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152 147

Table 2 (continued )

Author/s Participants' age/ Design Sleep issues Measurement Intervention Results Quality Rating [13]
gender/diagnosis

Malow et al. Parents (n ¼ 80) & RCT Sleep onset Pre & Post. Group (n ¼ 2 to 4 Improvements High
[23] children (n ¼ 80: 2 Individual delay, Sleep Actigraphy, Sleep parents: 2 x 2-h observed in sleep
e10 years: 64 Education efficiency & diaries, CSHQ, FISH, sessions over 2 problems and
males & 16 Program wake time after CBCL, RBS-R, Peds weeks) & behavioral
females). Autism (n ¼ 40) & sleep onset. QL, PSOC & End of Individual one x 1- measures
Spectrum Disorder. Group Education Session h session. Parent regardless of the
Asperger's, PDD- Education Survey. education mode of parent
NOS & Intellectual Program curriculum training education.
Disability. (n ¼ 40). program [37].
Telephone follow
-up. Behavioral
strategies included
graduated
extinction paired
with rewards,
bedtime pass,
visual schedules &
sleep hygiene.
Moss et al. [32] Parents (n ¼ 26: 29 RCT Night waking, Pre, Post & Follow- A 10-week Sleepwise program High
e61 years) & Sleepwise sleep duration, up. CSHQ, DBC-P, Sleepwise program was effective. Both
children (n ¼ 26: 8 treatment settling to PSI-SF, Consumer (adapted for older groups reduced
e17 years). Gender Group (n ¼ 13) sleep, excessive Satisfaction using a children & sleep problems
not reported. & Wait List daytime semi structured teenagers) however the
Autism Spectrum Control Group sleepiness, co- interview, CATS & consisted of 3 x 2-h improvements
Disorder, Angelman (n ¼ 13). sleeping and Goal Attainment workshops (n ¼ 5 were significantly
Syndrome, Down early waking. Scale. e7 parents) greater for the
Syndrome, focusing on sleep treatment group
Intellectual education and compared to the
Disability & behavioral WL.
Blindness. strategies.
Professional
facilitators
supported the
program
throughout within
the workshops;
home observations
and visits.
Fawkes et al. Study 1: Pilot study Comparison Specific sleep Pre & Post Comparing two A higher number of High
[15] Parents (n ¼ 20) & group design problems were Actigraphy methods of parent nights were scored
children (n ¼ 20: 4 between study not reported in Sleep diaries training on the use on the actigraphy
e10 years) Gender 1 (n ¼ 20) Ref. [15]. of the actigraphy and sleep diary data
not reported. conducted by and sleep diaries. for study two. This
Autism Spectrum Ref. [37] Brief verbal training was attributed to
Disorder [37]. & study 2 during study one's the structured
Study 2: Parents (n ¼ 80) informed consent educational hands
(n ¼ 80) & children Conducted by visit versus 1-h on parent training
(n ¼ 80: 2e10 Ref. [23]. structured hands delivered during
years). Gender not on actigraphy and study two.
reported. Autism sleep diary training
Spectrum Disorder, visit before study
Asperger's & two commenced.
Pervasive
Development
Disorder -Not
Otherwise
Specified [23].
Stuttard, Parents (n ¼ 23) & Single group Bedtime Pre, Post & Follow Group delivered Improvements in High
Beresford children (n ¼ 22: 5 design (n ¼ 23). resistance; up at 3 & 6 months. (n ¼ 4e9 parents) child's bedtime
et al. [42] e15 years: 13 night CSHQ, PSCO, in one of the 4  3 h resistance and
males & 9 females). awakening and Implementation sessions over 5 night awakenings
Autism Spectrum night-time self- fidelity checklist. weeks. Two were observed.
Disorder; settling. learning disability Parent's beliefs,
Intellectual nurses form attitudes and
Disability CAMHS-LD team confidence
delivered the improved.
Managing your
child's behavior to
promote better
sleep (MCBPBS)
manual.
(continued on next page)
148 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152

Table 2 (continued )

Author/s Participants' age/ Design Sleep issues Measurement Intervention Results Quality Rating [13]
gender/diagnosis

Stuttard, Clarke Parents (n ¼ 15) & Qualitative Sleep problems. Pre, Post & Follow Specialist health Irrespective of the High
et al. [42] children (n ¼ 15: 1 analysis. up at 2e3 months. visitors provided delivery mode (HV
e4 years: 11 males Comparison Semi structured Sleep strategy or TC) parents
and 4 females). groups: home interviews. implementation reported benefits
Autism Spectrum visits (HV support (SSIS) to from receiving SSIS
Disorder, Cerebral n ¼ 7) & deliver an Intensive to implement the
Palsy, telephone behavioral sleep IBSMI. TC was
Developmental contact (TC management viewed to be an
Delay, Sensory n ¼ 8) & SSRD intervention acceptable and
Impairments & for sleep (IBSMI). Individual convenient mode to
Learning Disability. interventions. HV (3e9weeks for deliver SSIS.
30 min to 1 h) or TC
(3e10 weeks for 10
and 30 min).
Veatch et al. Parents (n ¼ 80) & Single group Sleep duration, Pre & Post. Parent education Parent sleep High
[45] children (n ¼ 80: 2 design. bedtime Actigraphy, CSHQ, curriculum training education resulted
e10 years: 64 resistance, sleep dairies. program (Malow in improvements in
males & 16 night time et al., [23]). Good correlations
females) awakenings. habits that between objective
Autism Spectrum contribute to good and subjective
Disorder. sleep patterns, measurements of
appropriate timing sleep problems.
of sleep, visual
schedules, healthy
child/parent
interactions during
night time
awakenings.

CBC, Child Behavior Checklist; CSHQ, Child Sleep Habit Questionnaire; RCT, randomized control trial; SSRD, single-subject research design.
Note. RCT e Randomized Control Trials, CSHQ e Child Sleep Habit Questionnaire, DBC-P e Developmental Behavior Checklist-Parent version, RBS-R e Repetitive Behavior
Scale-Revised, FISH e Family Inventory of Sleep Habits, CBC e Child Behavior Checklist, PedsQL e The Parent Proxy-Report of the Pediatric Quality of Life Inventory, PSOC e
Parents Sense of Competence Scale, PSI-SF e The Parenting Stress Index: Short Form.
Note. RCT e Randomized Control Trials.
Note. SSRD e Single Subject Research Design, CSHQ e Child Sleep Habit Questionnaire, PSOC e Parents Sense of Competence Scale, CAMHS-LD e Child and Adolescents Mental
Health Service e Learning Disability.

evidence to support the suggestion that hands-on extended parent trained sleep therapist in a clinic setting [21]. In comparison, par-
training was more valuable than the brief verbal instructions [15]. ents participating in the Malow et al., [23] study were supported by
Stuttard, Clarke, Thomas and Beresford, [42] conducted a trained sleep educators, with the group education program
study with children (n ¼ 15) with ASD, Cerebral Palsy, Devel- providing two 2-h sessions over two weeks compared to a single 1-
opmental Delay, Sensory Impairments and Learning disability h session provided by the individualized program. All parents
which implemented an individualized intensive behavioral sleep received two follow-up phone calls.
management intervention. Parents received continued support Moss et al., [32] and Austin et al., [6] (as detailed in parent
either through home visits (n ¼ 7) or telephone calls (n ¼ 8). training programs and sleep outcomes above), offered parent
Even though Stuttard et al., did not measure sleep as an outcome, training using a group mode of delivery which consisted of three 2-
they did provide some evidence of the effectiveness of the in- h sessions. However, the duration of the training differed, with
dividual delivery mode irrespective of the type of professional Austin et al., [6] running the program for 15 weeks as opposed to
contact [42]. It was found that training was beneficial and valued Moss et al., [32] running it for 10 weeks. Professional facilitators
by the parents implementing sleep interventions. supported both programs throughout, within the workshops and
Two further studies evaluating the mode of delivery of parent with home observations and visits. Both studies reported positive
training programs were Malow et al., [23] for children (n ¼ 80) with outcomes from the parent training program.
ASD, Asperger's, PDD-NOS, and ID, and Johnson et al., [21] for
children (n ¼ 40) with ASD and PDD-NOS. Malow et al., [23] 3.4. Follow-up and maintenance
compared individual parent education training (n ¼ 40) with
group parent education training (n ¼ 40), whereas Johnson et al., Five of the 11 studies reported follow-up data on sleep out-
[21] compared individual delivery mode of behavior parent training comes; these follow-up periods ranged from one month to six
(BPT, n ¼ 20) with individual psycho-educational parent training months postintervention. All five studies reported that improve-
(PE, n ¼ 20). Malow et al., [23] observed improvements in sleep ments in sleep outcomes were maintained at a one-month follow-
outcomes regardless of the delivery mode of parent education up [6], after two months [32], from one to three months [2], after
training. In contrast, Johnson et al., [21] reported mixed results. three months [31], and for repeated follow-ups at three and six
Measurements obtained using the actigraphy indicated no differ- months [42].
ences between the BPT and PE groups, although measurements
provided by the Composite Sleep Index indicated that there was a 3.5. Treatment fidelity and social validity
significant improvement in bedtime and sleep behaviors for the
BPT group in comparison to the PE group. In terms of professional Several of the studies included in the current review, with the
support, participating parents in the Johnson et al., study attended exception of Fawkes et al. [15] and Veatch et al. [46], reported either
five individually administered 60- to 90-min sessions with a treatment fidelity and/or social validity data. Stuttard, Beresford et
 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152 149

al., [42] reported implementation fidelity data for the content of the behaviors for several of the children. There are numerous factors
training workshops which were rated 100% consistent, relevant, that may have contributed to the meaningful sleep outcomes re-
beneficial and effective. Parent attendance was generally good, ported in the studies included in this review. Parent training pro-
with 11 parents attending all four workshops, and the acceptability grams included across the 11 studies incorporated a multi-element
of the training workshops was considered promising. approach with a combination of the following components of sleep
In a study by Johnson et al., [21], treatment fidelity measured the hygiene practice, sleep education, reinforcement, extinction, faded
delivery of treatment as intended and parent adherence to deliv- bedtime, response cost, sleepwake schedule, parentchild
ered treatment across the two treatment groups of BPT and PE. nighttime interaction, and psychoeducation. It is possible that the
Results were high for both groups, with BPT at 98% for treatment as improvements were assisted by the parent training program con-
intended and 93% for parent adherence to delivered treatment; the tent, the duration and mode of delivery of the parent training, along
PE was 99% for treatment as intended and 98% for parent adherence with professional support received, for example, face-to-face
to delivered treatment. Interrater reliability for treatment as consultation versus written information or group versus individ-
intended was reported as 98% for BPT group and 99% for the PE ual contact.
group, and parent adherence to the delivered treatment was 91% Notably, four of the studies included in the current review
for the BPT group and 98% for the PE group. A total of 30 parents [1,6,31,32] used written parent training materials. The content of
provided social validity data by completing a parent's satisfaction the Sleepwise manual employed by Moss et al., and Austin et al., as
questionnaire, on which 90% satisfaction was reported for the BPT well as the Better Nights, Better Days manual used by Moon et al.,
group and 88% for the PE group. [31] included sleep education and behavioral strategy topics similar
Of the remaining seven studies that provided social validity data to those presented in the standardized pamphlet for insomnia in
[2], found that parents evaluated the BTP as highly acceptable and children with ASD used by Adkins et al., [1]. However, Adkins et al.
effective, and stated that they intended to continue to use the BTP. reported negative results and Moss et al., Austin et al., and Moon
The parents who participated in the [32] study were satisfied with et al. reported positive results. Adkins et al. found the pamphlet to
the intervention outcomes and indicated that the Sleepwise pro- be ineffective for insomnia in children with ASD, whereas Moss
gram was acceptable, valuable, and effective. The Sleepwise pro- et al., [32] Austin et al., [6] and Moon et al., [31] reported significant
gram was also evaluated in the [6] study, and parents found that the positive sleep outcomes for children with developmental disabil-
resources used, training delivered, and outcomes were beneficial ities and ASD, respectively. Albeit not conclusive, it is possible that
and effective. In addition, parents stated that peer support was an the different delivery modes implemented, and the level of pro-
influential factor throughout the Sleepwise program. Parent satis- fessional support received, could have contributed to the reported
faction measured in the Moon et al., study indicated that parents outcomes. Moss et al., [32] and Austin et al., [6] provided profes-
were satisfied with the sleep intervention and that the manual was sional facilitators who supported the program with home visits and
helpful [31]. Adkins et al., [1] received feedback from parents that observations, and all parents attended three 2-h workshops which
suggested that the pamphlet was informative but lacked guidance focused on sleep education and behavioral strategies. Moon et al.,
on how to put the information into practice. In the Malow et al., [31] supported the parents via weekly telephone calls for the
[23] the parent's evaluation of the overall programme and the ed- duration of the study. In contrast, Adkins et al., [1] distributed the
ucators’ training delivery mode indicated a high level of satisfaction sleep education pamphlet and the parents received no further
with 75% for the group delivery sessions and 86% for the individual support for the duration of the study. The difference in the delivery
sessions. In total, 91% of parents agreed that there were improve- mode and/or the insufficient professional contact/support may be
ments in sleep habits. Parents’ reported views in the Stuttard, an explanation for the negative results reported by Adkins et al., [1]
Clarke et al., [42] revealed that parents felt the telephone contact These results would suggest that factors such as the delivery mode,
was considered acceptable and less time consuming nonetheless advice, and ongoing support are essential to maximize treatment
the telephone calls were considered very impersonal. Parents also outcomes.
expressed that the initial home visit to instruct the TC group on Further evidence presented from studies evaluated within the
how to implement the sleep intervention strategies was essential to current review would suggest that training delivered either in a
the success of the intervention. It was expressed that more face-to- group or in individual format produced similar positive treatment
face contact would have been valuable. outcomes [23]. Three studies also produced positive outcomes by
delivering parent training sessions ranging from two to five weeks
4. Discussion with durations varying from 1 to 4 h [21,23,42]. In addition, parent
training that was delivered by professionals and that contained
The aim of the present review was to evaluate and to synthesize similar sleep education and behavioral intervention content pro-
the effectiveness of parent training programs incorporated in duced positive treatment outcomes [21,23,42]. The synthesis of
behavioral sleep interventions for children with ASD and/or ID. In these studies has provided useful information on the program
the limited research currently available, it is evident that parent content, mode of delivery, and professional sleep educators’ input
training programs have the potential to reduce prevalent sleep into the implementation of evidence-based parent training that can
problems such as initiating and maintaining sleep, early morning improve sleep interventions for children with ASD/ID.
and night awakenings, and bedtime resistance. Future research In addition, two comparison group studies conducted by Fawkes
should build on this evidence to first clarify which elements of et al., [15] and Stuttard, Clarke, et al., [43] did not report evidence
parent training improve acquisition of healthy sleep practice for sleep outcomes. However, these authors did provide additional
knowledge, and second, to identify the challenges that may exist evidence to support the efficacy of parent training for sleep prob-
which prevent parents from implementing sleep knowledge into lems experienced by children with ASD/ID. This evidence offered
healthy sleep practice. information regarding the level of professional support options
which could be successfully integrated into behavioral sleep in-
4.1. Current evidence terventions. Fawkes et al., produced evidence from two previously
published research articles to support the suggestion that hands-on
Eight of the nine studies that measured sleep behaviors as an extended parent training was more valuable than brief verbal in-
outcome repeatedly demonstrated a reduction in the target structions [15]. As for Stuttard, et al., [14] they found that parent
150 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152

training was beneficial irrespective of the type of support offered treatment fidelity [42], measured the content of the training
during training and home implementation stage, for instance, workshops, and [21] measured the delivery of the behavioral
home visits (HV) or telephone calls (TC). The defining difference parent training as intended and parent adherence to training
between the two studies was that Stuttard, et al., [14] provided all delivered. Both studies received favorable feedback in relation to
parents with an intensive behavioral sleep management interven- the parent training provided which demonstrated that the training
tion, with the HV group receiving 30- to 60-min sessions for 3e9 was standardized, consistent, and effective. Fidelity feedback
weeks and the TC group receiving 10e30 min for 310 weeks within the current context could ultimately increase the replication
before delivering the home sleep strategy implementation support. of the training, promote dissemination of the research, and
In Fawkes et al., [15] data from previous research were compared encourage generalization of the training to other settings. Recent
wherein one group of parents were offered face-to-face extended research suggests that performance feedback is the most effective
training and the other group of parents were offered brief verbal way to improve treatment fidelity [8], and that this feedback is
instructions. It would appear from the evidence presented in these crucial to the successful transition of evidence-based interventions
two studies that face-to-face, good-quality training surpasses brief into practice. This has implications for the evidence presented in
verbal instructions and that parents did not differentiate between the current review, as only two of the 11 studies reported treatment
HV and TC and viewed both as acceptable methods of support. It is fidelity data. This has the potential of weakening the identification
always difficult to rule out alternative explanations for conflicting of the parent training components which may have been either
evidence, and this is one area of research that could benefit from ineffective or effective within behavioral sleep interventions.
future investigation. Future research should attempt to investigate these components
and investigate under which conditions parent training obtains
4.2. Quality of evidence optimal outcomes, to consider and understand what motivates
parents’ behavior and their application of the sleep knowledge
Methodological quality was assessed using the Downs and Black obtained from parent training programs.
[13] assessment criteria, which revealed that eight of the included In contrast, nine of the studies included in the current review
studies were of high quality and three were of good quality. The received positive social validity feedback; this would suggest that
benefits of using the Downs and Black assessment was that it parents found behavioral sleep interventions to be effective,
provided a more objective approach to the quality of the evidence acceptable, supportive, and relevant to their child's sleep problems.
presented within the current review. Rather than solely relying on Social validity plays a significant role in assisting researchers to
each of studies’ reported successful outcomes, the strength of the generate ongoing evaluation and to implement improvements to
evidence was also based on the quality of report writing, external the intervention strategies employed. This was a promising
validity, internal validity, confounding and selection bias, and sta- outcome; nonetheless, social validity should not be considered as a
tistical power for each study. Studies with high methodological replacement for the measurement of treatment fidelity.
quality will enhance the evidence and thus help to establish and
promote the approach as evidence-based practice for effective 4.5. Strengths and limitations
behavioral sleep interventions for children with ASD/ID.
It is possible that insufficient inclusion of methodological pro-
4.3. Follow-up and maintenance cedures relating to parent sleep education and behavioral in-
terventions within existing research has contributed to the small
Not all the studies included in the current review reported number of studies included in the current review. Each study
follow-up and maintenance data on treatment outcomes. Within within the current review was examined separately and then
the current review, five of the 11 studies provided short-term to compared on common features. Based on the evidence, it is
relatively long-term data on sleep outcomes. Each of the five apparent that parent training and professional support have a vital
studies reported that improvements in sleep outcomes were role to play in the success of behavior sleep interventions. As such,
maintained at a one-month follow-up [6], after two months [32], future behavioral sleep interventions should consider the addi-
from one to three months [2], after three months [31], and for tional resources required to provide additional professional sup-
repeated follow-ups at three and six months [43]. The number of port and guidance. Even though the extent of the evidence
studies that did not report follow-up efficacy on treatment out- presented is scant, it nonetheless strengthens the support for the
comes was a concern highlighted by the current review. To consider implementation of parent training programs within behavioral
an intervention as effective, it is imperative that the posttreatment sleep interventions. It is also important to clarify that the low level
long-term positive effects are maintained. The current review was of evidence may be a result of the substantial number of studies
unable to conclusively offer a strong synthesis of the maintenance excluded based on the exclusion criteria to not include pharma-
data. From an effective treatment perspective, future research cological interventions rather than the lack of research into the
should consider incorporating data collection on treatment treatment of sleep problems for children with ASD/ID. A further
outcome maintenance into research procedures. Considering the strength of the current review was the inclusion of a range of
evidence that parenting practices contribute to the improvement of research designs, diverse sample populations, a variety of parent
sleep problems [2,6,21,23,31,32,42,43]. An additional topic of in- training programs, and a range of outcome measures to maximize
terest should be the follow-up assessment of the maintenance of the use of the evidence available. This approach not only enabled an
parental knowledge/practice, as this can have implications for evaluation and comparison of the evidence presented, it also
either retaining sleep improvements or further enhancements in enabled a synthesis of the studies included in the current review.
sleep outcomes. However, this also created a limitation, as the studies included in
the review were significantly heterogeneous, and therefore it was
4.4. Treatment fidelity and social validity not appropriate to conduct a meta-analysis. Depending on the
variability within the research, a meta-analysis may be used to
Several of the studies included in the current review, with combine and to directly compare quantitative results, providing
exception of Fawkes et al., [15] and Veatch et al., [45] reported additional information such as the statistical efficacy of parent
either treatment fidelity and/or social validity data. In terms of training programs. As research into parent training and behavioral
 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152 151

sleep interventions for children with ASD/ID advances and more The ICMJE Uniform Disclosure Form for Potential Conflicts of
homogeneous studies exist, future research would benefit from the Interest associated with this article can be viewed by clicking on the
inclusion of a meta-analysis within systematic reviews. Even following link: https://doi.org/10.1016/j.sleep.2018.08.034.
though the studies in the current review produced results that
were too different to combine in a meta-analysis, the formal and
rigorous systematic approach enabled a level of comparability of References
the distinct aspects of information presented within each study and [1] Adkins KW, Molloy C, Weiss SK, et al. Effects of a standardized pamphlet on
provided an opportunity to assess the consistency of the evidence. insomnia in children with autism spectrum disorders. Pediatrics
It would appear, regardless of the variability within the current 2012;130(Suppl. 2):S139e44.
[2] Allen KD, Kuhn BR, DeHaai KA, et al. Evaluation of a behavioral treatment
studies, that parent training was an effective component of
package to reduce sleep problems in children with Angelman syndrome. Res
behavioral sleep interventions. The narrative synthesis offered Dev Disabil 2013;34:676e86.
within the current review should be considered with the previously [3] American Association on Intellectual and Developmental Disabilities. Intel-
lectual Disability. Available at: http://aaidd.org/intellectual-disability/
stated limitations and strengths in mind. In addition, the current
definition#.WPi8mIWcE2w. [Accessed 20 September 2016].
findings may not generalize to other clinical populations. [4] American Psychiatric Association. Diagnostic and statistical manual of mental
disorders. 5th ed. Arlington, VA: American Psychiatric Association; 2013.
[5] Andersen IM, Kaczmarska J, McGrew SG, et al. Melatonin for insomnia in
children with autism spectrum disorders. J Child Neurol 2008;23:482e5.
5. Conclusion [6] Austin KL, Gordon JE, O'Connell A. Preliminary evaluation of Sleepwise pro-
gram for children with sleep disturbance and developmental delay. Child Fam
The current review conveys an emerging evidence-based prac- Behav Ther 2013;35:195e211.
[8] Breitenstein SM, Gross D, Garvey CA, et al. Implementation fidelity in com-
tice that could contribute to future behavioral sleep research and
munity-based interventions. Res Nurs Health 2010;33:164e73.
guide best-practice decisions to support effective parent training to [9] Bruni O, Alonso-Alconada D, Besag F, et al. Current role of melatonin in pe-
improve sleep outcomes for children with ASD and/or ID. Quality diatric neurology: clinical recommendations. Eur J Paediatr Neurol 2015;19:
122e33.
research should be conducted to demonstrate the efficacy and
[12] Doran SM, Harvey MT, Horner RH. Sleep and developmental disabilities:
appropriateness of parent training incorporated in behavioral sleep assessment, treatment, and outcome measures. Ment Retard 2006;44:13e27.
interventions. The evidence presented in the current review may [13] Downs SH, Black N. The feasibility of creating a checklist for the assessment of
inform and guide future clinical practice in the management of the methodological quality both of randomised and non-randomised studies
of health care interventions. J Epidemiol Commun Health 1998;52:377e84.
pediatric sleep problems and enable clinicians and families to make [14] Effective Practice and Organisation of Care (EPOC). Data collection form. EPOC
informed choices. Future behavioral sleep interventions would Resources for review authors. Oslo: Norwegian Knowledge Centre for the
benefit from increased availability and accessibility of parent Health Services; 2013. Available at: http://epoc.cochrane.org/epoc-specific-
resources-review-authors. [Accessed December 2016].
training programs, along with additional professional support to [15] Fawkes DB, Malow BA, Weiss SK, et al. Conducting actigraphy research in
promote meaningful treatment outcomes. It would be beneficial if children with neurodevelopmental disordersda practical approach. Behav
future studies increased the sample size, as this could have the Sleep Med 2015;13:181e96.
[18] Hiscock H, Sciberras E, Mensah F, et al. Impact of a behavioral sleep inter-
potential of demonstrating outcomes more clearly with greater vention on symptoms and sleep in children with attention deficit hyperac-
statistical power. Future large-scale RCTs could investigate and tivity disorder, and parental mental health: randomised controlled trial. BMJ
identify the effective components of parent training programs for 2015;350:h68.
[19] Hollway JA, Aman MG, Butter E. Correlates and risk markers for sleep
children with ASD/ID. It is equally important that future studies
disturbance in participants of the autism treatment network. J Autism
incorporate a measurement of treatment fidelity, as this could Develop Disord 2013;43:2830e43.
provide the knowledge required to shed more light on the training [20] Hurley AD, Levitas AS. The importance of recognizing autism spectrum dis-
orders in intellectual disability. Ment Health Aspects Dev Disabil 2007;10:
content, duration, mode of delivery, level of professional support,
157e62.
and training delivered as intended. Regarding immediate and [21] Johnson CR, Turner KS, Foldes E, et al. Behavioral parent training to address
postintervention effects and maintenance of improved sleep be- sleep disturbances in young children with autism spectrum disorder: a pilot
haviors, researchers should be mindful of the parents’ ongoing role trial. J Sleep Med 2013;14:995e1004.
[23] Malow BA, Adkins KW, Reynolds A, et al. Parent-based sleep education for
of supporting the positive outcomes in the months following children with autism spectrum disorders. J Autism Dev Disord 2014;44:216e28.
treatment. Future research designed to evaluate the immediate and [24] Malow BA, Byars K, Johnson K, et al. A practice pathway for the identification,
postintervention effects would be of significant value, as analyses of evaluation, and management of insomnia in children and adolescents with
autism spectrum disorders. Pediatrics 2012;130(Suppl. 2):S106e24.
follow-up data enable an understanding of the retention of the [25] Mannion A, Leader G. Sleep problems in autism spectrum disorder: a litera-
outcomes and could be vital to evaluating the overall efficacy of ture review. Rev J Autism Devel Disord 2014;1:101e9.
parent training programs. There is a necessity for further evaluation [26] Mannion A, Leader G, Healy O. An investigation of comorbid psychological
disorders, sleep problems, gastrointestinal symptoms and epilepsy in children
studies, as sleep problems are extremely prevalent and have sig- and adolescents with autism spectrum disorder. Res Autism Spect Disord
nificant negative consequences for the individual who experience 2013;7:35e42.
sleep problems and for the families who support them. [27] Matson JL, Shoemaker M. Intellectual disability and its relationship to autism
spectrum disorders. Res Dev Disabil 2009;30:1107e14.
[28] Meltzer LJ, Mindell JA. Systematic review and meta-analysis of
behavioral interventions for pediatric insomnia. J Pediatr Psychol 2014;39:
Source of funding 932e48.
[29] Mindell JA, Emslie G, Blumer J, et al. Pharmacologic management of insomnia
in children and adolescents: consensus statement. Pediatrics 2006;117:
This research is supported by funding from the charity RESPECT e1223e32.
and the People Programme (Marie Curie Actions) of the European [31] Moon EC, Corkum P, Smith IM. Case study: a case-series evaluation of a
Union's Seventh Framework Programme (FP7/2007e2013) under behavioral sleep intervention for three children with autism and primary
insomnia. J Pediatr Psychol 2010;36:47e54.
REA grant Agreement no PCOFUND-GA-2013-608728. [32] Moss AH, Gordon JE, O'Connell A. Impact of Sleepwise: an intervention for
youth with developmental disabilities and sleep disturbance. J Autism Dev
Disord 2014;44:1695e707.
Conflict of interest [35] Noterdaeme M, Enders A. Autistic disorders and mental retardation. How to
recognise and differentiate? Tagliche Praxis 2009;50:111e21.
[37] Reed HE, McGrew SG, Artibee K, et al. Parent-based sleep education work-
The authors have no conflict of interest to declare. shops in autism. J Child Neurol 2009;24:936e45.
152 B. Kirkpatrick et al. / Sleep Medicine 53 (2019) 141e152

[38] Reynolds AM, Malow BA. Sleep and autism spectrum disorders. Pediatr Clin [43] Stuttard L, Clarke S, Thomas M, et al. Replacing home visits with telephone
North Am 2011;58:685e98. calls to support parents implementing a sleep management intervention:
[39] Richdale AL. Sleep problems in autism: prevalence, cause, and intervention. findings from a pilot study and implications for future research. Child Care
Dev Med Child Neurol 1999;41:60e6. Health Dev 2015;41:1074e81.
[40] Richdale A, Francis A, Gavidia-Payne S, et al. Stress, behavior, and sleep [45] Veatch OJ, Maxwell-Horn AC, Malow BA. Sleep in autism spectrum disorders.
problems in children with an intellectual disability. J Intellect Dev Disabil Curr Sleep Med Rep 2015;1:131e40.
2000;25:147e61. [46] Veatch OJ, Reynolds A, Katz T, et al. Sleep in children with autism spectrum
[41] Sikora DM, Johnson K, Clemons T, et al. The relationship between sleep disorders: how are measures of parent report and actigraphy related and
problems and daytime behavior in children of different ages with autism affected by sleep education? Behav Sleep Med 2015;14(6):665e76.
spectrum disorders. Pediatrics 2012;130(Suppl. 2):S83e90. [47] Vriend JL, Corkum PV, Moon EC, et al. Behavioral interventions for sleep
[42] Stuttard L, Beresford B, Clarke S, et al. A preliminary investigation into the problems in children with autism spectrum disorders: current findings and
effectiveness of a group-delivered sleep management intervention for parents future directions. J Pediatr Psychol 2011;36:1017e29.
of children with intellectual disabilities. J Intellect Disabil 2015;19:342e55.