Psychoeducational Interventions For Adults With.1
Psychoeducational Interventions For Adults With.1
Psychoeducational Interventions For Adults With.1
Cogn Behav Neurol Volume 32, Number 3, September 2019 www.cogbehavneurol.com | 139
Davis et al Cogn Behav Neurol Volume 32, Number 3, September 2019
by the most recent version of the Diagnostic and Statistical instruction on improving skills that are deficient, enhanc-
Manual of Mental Disorders (DSM–5; American Psychi- ing best individual abilities, or developing compensatory
atric Association, 2013) as “requiring very substantial mechanisms? These same questions have concerned edu-
support” due to severe impairments in social communi- cators and psychologists studying the ASD–3 population
cation and restrictive/repetitive behaviors. Although the for decades, as have the best potential psychoeducational
scale of severity described in the Diagnostic and Statistical strategies.
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Manual is defined by key functional impairments such as The main aims of this review were to (a) assess the
poor social communication and restricted/repetitive be- psychoeducational interventions that have thus far been
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haviors, it is important to consider a potential heteroge- applied to adults with ASD–3, (b) identify how rigorously
neous set of comorbid impairments, including intellectual these interventions have been applied and assessed (eg,
disability and behavioral impairments, that could also adherence to protocols, data collection, and analyses) us-
require substantial support. Interventions aimed at re- ing quality assessment/risk of bias tools, (c) evaluate the
ducing the impact of these impairments and facilitating efficacy of the interventions used so far within the adult
individuals with ASD in practicing self-care, employment, ASD–3 population, and (d) suggest practical ways to ad-
and participation in recreational activities would argu- vance the field of psychoeducational interventions.
ably significantly improve their quality of life and also
help reduce the enormous cost of social care (Leigh and METHODS
Du, 2015).
To our knowledge, no prior reviews have inves- Search and Information Sources
tigated the availability and quality of psychoeducational We searched articles and reports within the 50-year
interventions for adults with ASD–3. The only reviews we period from 1968, when applied behavioral analysis first
located focused on psychoeducational interventions for appeared in the literature (Baer et al, 1968), to the present.
adults with mixed- and/or higher level ASD, and these Our inclusion criteria were adults (age ≥ 18 years) with a
supported the effectiveness of applied behavioral analysis primary diagnosis of ASD at level 3 on the severity scale
(Aylott, 2000), social skills interventions (Aylott, 2000; (“requiring very substantial support”; American Psychi-
Lorenc et al, 2018; Reichow and Volkmar, 2010), voca- atric Association, 2013).
tional skills interventions (Lorenc et al, 2018; Nicholas We started with relatively broad searches of the
et al, 2015), and video modeling interventions (Reichow PubMed and Google Scholar electronic databases, using
and Volkmar, 2010). One prior review of psychoeduca- the search terms (“1968”[PDAT]: “3000”[PDAT]) AND
tional interventions for mixed-age individuals with ASD–3 (“autism”[All Fields] OR “autistic”[All Fields] OR
also supported the effectiveness of social skills inter- “ASD”[All Fields] OR “PDD”[All Fields]) AND “adult”
ventions using video-based, developmental, peer-mediated, [All Fields] AND (“intervention”[All Fields] OR “treat-
behavioral, and structured teaching approaches (Walton ment”[All Fields] OR “therapy”[All Fields] OR “train-
and Ingersoll, 2013). Several other published reviews of ing”[All Fields]). We used broad search terms because recent
interventions for children and mixed-aged individuals with debates regarding the suitability of specific terminology to
ASD notably highlighted the lack of research into inter- describe severity level (eg, low-functioning, Asperger’s) made
ventions for adults with ASD–3 and called for more it difficult to synthesize search terms that would extract our
attention to the topic (Kasari et al, 2014; Lorenc et al, 2018; specific target population. Also, we wanted to evaluate
Matson, 1996; Shattuck et al, 2012; Taylor et al, 2012; the far-reaching conclusions and impacts of a wide range of
Weiss and Harris, 2001). interventions.
Because our review was limited to studies on psy-
choeducational interventions, we did not include literature
OBJECTIVE reviews, meta-analyses, books, or program reports (see
We conducted this review in order to clarify what Eligibility Criteria and Study Selection), although several
psychoeducational interventions have been shown to work were identified in the electronic search. However, we did
best for what behavioral outcomes within the ASD–3 manually scrutinize the reference lists of literature reviews
category. At present, behavioral training methods that and meta-analyses (Aylott, 2000; Bishop-Fitzpatrick et al,
have proven effective in the treatment of behavioral defi- 2013; Broadstock et al, 2007; Brugha et al, 2015; Com-
cits in children with ASD (Heflin and Simpson, 1998) have mittee on Children With Disabilities, 1998; DeJong et al,
been widely adopted and adapted for adults with ASD. 2014; El Achkar and Spence, 2015; Gates et al, 2017;
Nonetheless, there are considerable controversies as to the Kasari et al, 2014; Lorenc et al, 2018; Matson et al, 2011,
suitability of these methodological adaptations for adults 2016; Millar et al, 2006; Nicholas et al, 2015; Ratto and
with ASD–3, as well as to what knowledge or skills should Mesibov, 2015; Reichow and Volkmar, 2010; Reichow
be taught (Heflin and Simpson, 1998). For instance, given et al, 2013; Rutter, 1996; Schreibman, 1996; Shattuck
the inevitable limitations in time and resources facing et al, 2012; Sinha et al, 2004, 2006; Sturmey, 2012;
clinicians and educators, what skills and competencies Taylor et al, 2012; Tsai, 1999; Vismara and Rogers, 2010;
should be psychoeducationally targeted and how? Should Volkmar et al, 1999, 2014; Walton and Ingersoll, 2013;
we try to improve independent self-care skills, social skills, Weiss and Harris, 2001; Wong et al, 2015), books (Sheridan
and/or work-related competencies? Should we focus and Raffield, 2008; Volkmar et al, 2014), and program
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Cogn Behav Neurol Volume 32, Number 3, September 2019 Psychoeducational Interventions for ASD–3
which to include in our review. Two eligibility reviewers ◦ The published study was not available in English.
(K.S.D. and S.A.K.) screened the studies to make sure they
Regarding terminology, we use the term “psycho-
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We excluded studies based on the following criteria: Risk of Bias Within Studies
Types of participants We assessed each identified study for quality and for
risk of bias using standards outlined in the Cochrane Risk
◦ Participants were children and/or adolescents (ie, of Bias data extraction template for randomized inter-
aged <18 years). ventions (Higgins and Green, 2011), the Cochrane Risk of
◦ Participants were not adults with ASD (eg, partic- Bias in Non-Randomized Studies template for non-
ipants were either children of, or family members randomized interventions (Sterne et al, 2016), and the
and caregivers of, individuals with ASD). Reichow Risk of Bias template for case studies (Reichow
◦ ASD severity was below level 3 (ie, the severity level et al, 2018). Because bias domains in the Cochrane Risk of
was determined to be level 1 or 2, or the level could Bias, Cochrane Risk of Bias in Non-Randomized Studies,
not be determined from the available data). and Reichow Risk of Bias tools overlap, we mapped bias
domains from the Cochrane Risk of Bias in Non-
Types of interventions
Randomized Studies and the Reichow Risk of Bias onto
◦ The study was not a true intervention study (ie, it was the original scoring conventions of the Cochrane Risk of
a systematic review, meta-analysis, prevalence study, Bias. We identified five key bias domains (selection bias,
description of symptoms, follow-up study), and/or it performance bias, detection bias, attrition bias, and re-
was another type of nonempirical study (eg, position porting bias) and design-specific criteria for each domain
paper, book review). (Table 1). We assessed each bias domain as being low risk,
◦ The study consisted of pharmacological or other moderate risk, high risk, or very high risk. Quality
“physical” intervention methods. assessment was carried out independently by S.A.K.,
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Davis et al
TABLE 1. Bias Domain Definitions
Selection Bias Performance Bias Detection Bias Attrition Bias Reporting Bias
Cochrane Random sequence Blinding of participants and Blinding of outcome Incomplete outcome data: Selective reporting: “State how
RoB generation/allocation personnel: “Describe all assessment: “Describe all “Describe the completeness the possibility of selective
concealment: “Describe the measures used, if any, to measures used, if any, to of outcome data for each outcome reporting was
method used to generate blind study participants blind outcome assessors main outcome, including examined by the review
the allocation sequence in and personnel from from knowledge of which attrition and exclusions authors, and what was
sufficient detail to allow an knowledge of which intervention a participant from the analysis. State found.”
assessment of whether it intervention a participant received. Provide any whether attrition and
should produce received. Provide any information relating to exclusions were reported,
comparable groups. information relating to whether the intended the numbers in each
Describe the method used whether the intended blinding was effective.” intervention group
to conceal the allocation blinding was effective.” (compared with total
sequence in sufficient detail randomized participants),
to determine whether reasons for attrition/
intervention allocations exclusions where reported,
could have been foreseen in and any re-inclusions in
advance of, or during, analyses performed by the
enrollment.” review authors.”
Cochrane Allocation bias: “ROBINS-I Performance bias: “Bias may Measurement bias/observer Bias due to missing data: Outcome reporting bias:
ROBINS also addresses time-varying occur when these bias: “Differential “Reasons for missing data “Selective outcome reporting
Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc.
confounding. This only differences arise because of misclassification occurs include attrition (loss to occurs when an effect
needs to be considered in knowledge of the when misclassification of follow up), missed estimate for a particular
studies that partition intervention applied and intervention status is related appointments, incomplete outcome measurement is
follow up time for the expectation of finding a to the outcome or the risk of data collection and selected from among
switch between the
A.D., and K.S.D., and any differences in opinion were Scholar k = 9, no duplicates); our hand search yielded 24
arbitrated by R.L.S. studies. Figure 1 is a flow diagram of our literature search.
We excluded studies for the following reasons: they
Summary Measures were intervention studies in which participants were not
In order to derive treatment effect sizes, we calcu- adults with ASD (ie, children and/or adolescents with
lated standardized mean difference scores and 95% CIs ASD or family members of individuals with ASD,
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using pre- and postintervention scores of the primary k = 2819); they were intervention studies in which partic-
outcome variables for each study (Higgins and Green, ipants did not meet the criteria for ASD–3 or it was im-
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2011; Olive and Smith, 2005). Several of the studies from possible to isolate the results for participants with ASD–3
our electronic and hand searches included some partici- (k = 190); they were studies of pharmacological inter-
pants who were not diagnosed with ASD–3; for those ventions (k = 405); they were nonempirical studies or
studies, we considered only the findings for the partici- quantitative data relevant to the psychoeducational in-
pants with ASD–3. tervention were not provided (k = 907); or the study was
not available in English (k = 4).
Synthesis of Results Ultimately, a total of 56 studies were included in this
Inconsistencies and variability in reporting of inter- review: 46 (82%) from the electronic search and 10 (18%)
vention content and outcome measures made a structured from the hand search. Of the studies identified by elec-
meta-analysis impossible. Thus, we organized our syn- tronic search, 16 were published in the last 10 years, and
thesized results according to behavioral outcome. 40 were published earlier; of the studies identified by hand
search, one was published in the last 10 years, and nine
Risk of Bias Across Studies were published earlier. The 56 studies that ultimately met
In order to rate the certainty of evidence supporting our criteria for inclusion in this review are summarized in
psychoeducational interventions targeting specific behav- Table 2.
ioral outcomes, we used the Cochrane GRADE approach
to rate the literature, taking into account the quality of the
evidence and the magnitude of the effect (Schünemann Risk of Bias Within Studies
et al, 2013). Given the difficulties in studying the ASD pop-
ulation, as well as the evolution of research standards over
RESULTS the last 50 years, we (as expected) found many method-
ological limitations in all of the studies. Considering cur-
Study Selection rent terminology, these limitations raise the possibility of
Our electronic search yielded 4357 studies across biases in the interpretation of the data. Because they were
PubMed and Google Scholar (PubMed k = 4348, Google common between studies, the limitations will be reported
FIGURE 1. Flow diagram of our literature search. ASD–3 = level 3 autism spectrum disorder.
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Davis et al
TABLE 2. Characteristics of Included Studies
Excluded Outcome Standard
Sample From M Age Intervention Timing of Measurement M ASD–3
Reference Design Size Review % Male (Years) Description Measurements Outcome Type Difference Diagnosis
Adelinis and ABAB 1 0 100.0 27 Behavioral intervention for During each Aggressive/ Behavioral 2.30 History
Hagopian aggressive behavior using session Destructive observation [–1.89, 6.50]
(1999) symmetrical ‘‘do’’ and “don’t” behaviors
requests
Baker et al AB 1 0 100.0 45 Intervention for coprophagia by Daily Self-injurious Behavioral 7.13 History
(2005) introduction of highly spiced, behaviors observation [2.76, 11.49]
flavorful food options for each
meal and as snacks
Banda et al ABA 1 1 (child) 100.0 21 Communication skills training using During each Language/ Behavioral NE Confirmed
(2010) video-recorded, individualized session Communication observation by direct
vignettes that showed modeling of skills assessment
requesting of objects
Bebko et al ABCD 1 19 (children 100.0 20 Examined shared communication During each Language/ Behavioral NE History
(1996) or not method, facilitated communication session Communication observation
ASD–3) skills
Bennett et al ABA 2 1 (not 100.0 26 Vocational skills training using During each Vocational Behavioral NE History
(2010) ASD–3) physical and vocal feedback in session skills observation
the form of praise, guidance, and
correction statements delivered via
covert audio coaching
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Boso et al AB 8 0 87.5 30.2 Active musical activities aimed at Postintervention Aggressive/ Rating scale 2.13 Confirmed
(2007) facilitating social engagement, at 26 and 52 Destructive [0.84, 3.45] by direct
improving behavioral problems, weeks behaviors assessment
and enhancing creative music
making (drumming, piano
playing, singing)
et al (2014) in waiting situations, using months functioning observation [–0.56, 4.21]
Duker and ABA 2 3 (children) 100.0 26 Response-contingent During each Stereotypy/ Behavioral 0.76 Confirmed
Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc.
(1991) measures instructional methods on baseline Communication observation by direct
Villamisar controlled trial group: 59.5; group: 31.5; exposure to various preferred postintervention [1.37, 2.48] by
and wait list wait list activities presented in several clinical
Dattilo group: 55.9 group: 30.1 levels of difficulty with evaluation
(2010) associated levels of support
Gaylord- ABCD 2 2 (children) 100.0 19 1: Social skills training using direct During each Language/ Behavioral 7.02 Confirmed
Ross et al instruction, modeling, and role play generalization Communication observation [–1.24; 15.28] by
(1984) with 3 leisure objects (a radio, a probe and at skills clinical
video game, and gum) follow-up evaluation
2: Social skills training using direct (4 months later)
instruction, modeling, and role play
with 3 leisure objects (a radio, a
video game, and gum)
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Davis et al
TABLE 2. (continued)
Excluded Outcome Standard
Sample From M Age Intervention Timing of Measurement M ASD–3
Reference Design Size Review % Male (Years) Description Measurements Outcome Type Difference Diagnosis
Gerber et al Repeated 31 0 74.2 43 1: Autism programme with a Aberrant Behavior Emotional Rating scale 1.51 [0.75, Confirmed
(2011) measures structured method with chosen Checklist: functioning, 2.28]; 0.87 by direct
objectives focused on the 3 months language/ [0.17, 1.57]; assessment
development of autonomy Quality of Live communication 0.09
2: Traditional program for intellectual Inventory: at the skills, stereotypy/ [–0.57, 0.76]
disability (not an autism programme beginning, after mannerisms
with a structured method) based 12 months and at
on systemic pedagogy and a variety the end of the
of approaches, ie, developmental study
and individualized approaches; Child Autism
chosen objectives focused on Rating Scales:
strengthening the processes approximately
of socialization every 12 months
Gilson and ABA 2 1 100.0 21.5 One-on-one coaching through an During each Language/ Behavioral 0.63 History
Carter (not ASD–3) earpiece of social and task- session Communication observation [–3.46, 4.73]
(2016) related skills skills
Goodson ABAB 3 1 (did not 100.0 34.3 1: Skills training using video Twice per week Activities Behavioral 2.77 Confirmed
et al (2007) receive modeling and voice-over per session of daily living observation [–13.03, by direct
intervention) instructions 18,58] assessment
2: Skills training using video
Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc.
3: through restricting access
Jerome et al ABA 2 1 100.0 33 Skills training using errorless During each Activities of Behavioral 4.35 History
Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc.
proprioceptive and vistubular
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Davis et al
TABLE 2. (continued)
Excluded Outcome Standard
Sample From M Age Intervention Timing of Measurement M ASD–3
Reference Design Size Review % Male (Years) Description Measurements Outcome Type Difference Diagnosis
McClean AB 2 3 100.0 23 Positive behavior support in Behavior: Aggressive/ Behavioral 0.63 [–3.45, History
et al (2007) (not ASD–3) community settings as an throughout the Destructive observation 4.72]
intervention for serious physical duration of the behaviors, and rating scale (combined
injury resulting from challenging study self-injurious effect size for
behaviors Mini Psychiatric behaviors aggressive/
Evaluation Scales destructive
for Adults with behaviors
Developmental and self-
Disabilities: at injurious
baseline, and at 6 behaviors)
and 12 months,
and
postintervention
Quality of Life
Questionnaire:
pre- and
postintervention
Medication: every
month Costs: at
baseline and at
Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc.
18 months
McKee et al ABAB 3 0 100.0 30.3 Social skills training using a Daily monitoring Aggressive/ Behavioral 0.20 History
(2007) Snoezelen room of disruptive Destructive observation [–1.81, 1.41];
behaviors and behaviors, 0.08
hourly language/ [–1.52, 1.69]
monitoring of communication
controlled eating with differential 6 month
Saiano et al Between- 3 4 (not 100.0 40 Safety skills training using direct Familiarization: Activities of Behavioral 2.10 Confirmed
Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc.
weeks
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Davis et al
TABLE 2. (continued)
Excluded Outcome Standard
Sample From M Age Intervention Timing of Measurement M ASD–3
Reference Design Size Review % Male (Years) Description Measurements Outcome Type Difference Diagnosis
Van Randomized 6 26 NS 25 Residential program, based on 6-month intervals Activities of Rating scale NE Confirmed
Bourgon- controlled trial (did not the TEACCH model, focused daily living by direct
dien et al receive on improving residents’ ability assess-
(2003) intervention) to function independently ment
within the community
Vuran (2008) AB 2 0 100.0 22 Skills training using verbal During each Activities of daily Behavioral 3.84 History
and physical feedback intervention living observation [–17.98,
session and at 6 25.67]
week follow-up
Wong et al AB 1 0 100.0 31 Subject treated with a During each Self-injurious Behavioral 4.90 History
(1991) differential reinforcement session behaviors observation [–8.65, 10.57]
of other behavior schedule
and compliance training
Averages 5.2 83.5 27.7
Totals ABAB: k = 8; Activities of daily Behavioral History:
AB: k = 17; living: k = 9; observation: k = 31;
ABA: k = 12; aggressive/ k = 51 Con-
ABCD: k = 2; destructive Rating scale: firmed by
ABC: k = 1; behaviors: k = 12; k = 10 direct
Repeated emotional assess-
Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc.
Volume 32, Number 3, September 2019
Naturalistic
exploratory:
k=1
ASD–3 = level 3 autism spectrum disorder. NE = no effect size calculated. NS = not stated.
Cogn Behav Neurol Volume 32, Number 3, September 2019 Psychoeducational Interventions for ASD–3
as general themes rather than discussing them study by We also found that the assessment of interrater re-
study. Figures 2 and 3 depict our bias assessments. liability was often insufficient, which is of particular con-
There was a high risk of bias in terms of the study cern for studies of psychosocial and behavioral
design of the studies we included in our review (Higgins functioning, in which assessment of the outcome is sub-
and Green, 2011; Reichow et al, 2018; Sterne et al, 2016). jective. Missing data were also encountered in many of the
First, only a few of the studies were randomized con- studies in our review; however, few of the studies discussed
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trolled trials; this, by default, introduces selection bias to this issue, and there are no available published analyses of
the sampling strategy. In most of the studies, the par- the possible patterns of missing data that may have con-
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ticipants were selected based on the subjective judgment founded the reported findings and their interpretation.
of the clinicians. It is important to note that it is To assess whether the methodological quality of the
extremely difficult to conduct randomized controlled studies improved over time, we correlated the number of
trials on this population given that few institutions pro- methodological issues identified with the publication date
vide regular access to participants. Other ethical and of each study. The quality of study methods did show a
logistical considerations associated with this population trend toward improvement with time, but this trend was
—including, but not limited to, obtaining consent for not significant (r = –0.211, P = 0.110; Figure 4).
participation, transportation to and from sessions, and
compensation for participation—make this population Descriptive Review of the Findings
relatively challenging to recruit. Nevertheless, we did Despite the aforementioned issues, the studies we
identify four randomized controlled trials containing reviewed did provide useful evidence concerning psycho-
both a treatment group and a control group (Elliott et al, educational interventions for adults with ASD–3. This
1994; García-Villamisar and Dattilo, 2010; Lundqvist evidence will be qualitatively evaluated here by behavioral
et al, 2009; Van Bourgondien et al, 2003). However, the outcome (see Tables 3 and 4, and Figure 5, for a complete
process of randomization was clearly described in only summary of the data).
three of these (Elliott et al, 1994; Lundqvist et al, 2009;
Van Bourgondien et al, 2003), and all four studies pro- Activities of Daily Living
vided inadequate descriptions of the inclusion and ex- Due to a very low proportion of studies that showed
clusion criteria as well as limited information on other a significant effect of the intervention yet showed a very
potential neurodevelopmental factors that may have in- high risk of bias, the quality of the evidence supporting the
fluenced participant performance. Thus, it is difficult to effectiveness of interventions to improve daily activities in
tell how representative the participants in the four studies adults with ASD–3 was very low. This behavioral out-
were of the population as a whole (which, as mentioned, come was investigated in nine studies: Edrisinha et al
is heterogeneous in itself). Moreover, it was impossible to (2011), Goodson et al (2007), Haring et al (1987), Jerome
determine the presence of any confounding factors rele- et al (2007), Saiano et al (2015), Siaperas and Beadle-
vant to the outcomes tested. Only three of the 56 studies Brown (2006), Smith and Belcher (1985), Van Bourgon-
(Carminati et al, 2007; Fava and Strauss, 2010; Gerber dien et al (2003), and Vuran (2008). One study involved a
et al, 2011) blinded the participants, investigators, and social skills training program as the intervention (Siaperas
outcome assessments; the remainder of the included and Beadle-Brown, 2006); the other eight studies focused
studies did not consider the impact of performance and on behavioral techniques as the intervention (Edrisinha
detection bias in their designs. et al, 2011; Goodson et al, 2007; Haring et al, 1987;
FIGURE 2. Risk of bias graph: Review authors’ judgments about each risk-of-bias item presented as percentages across the 56
studies.
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Davis et al Cogn Behav Neurol Volume 32, Number 3, September 2019
1.2
1
0.8
Bias Ratio
0.6
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0.4
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0.2
0
1980 1985 1990 1995 2000 2005 2010 2015 2020
Publication Date
FIGURE 4. Bias ratio by publication date: Ratio of bias domains
judged to have high risk plotted against publication date.
Aggressive/Destructive Behaviors
Due to a very low proportion of studies that showed a
significant effect of the intervention yet showed a high risk of
bias, the quality of the evidence supporting the effectiveness
of interventions to reduce aggressive/destructive behaviors in
adults with ASD–3 was low. This outcome was investigated
in 12 studies: Adelinis and Hagopian (1999), Boso et al
(2007), Carminati et al (2007), Elliott et al (1994), Fava and
Strauss (2010), Kaplan et al (2006), Kennedy (1994),
Lundqvist et al (2009), McClean et al (2007), McKee et al
(2007), McNally et al (1988), and Reese et al (1998). Two
studies used recreational therapies as the intervention (Boso
et al, 2007; Lundqvist et al, 2009), seven used behavioral
techniques (Adelinis and Hagopian, 1999; Carminati et al,
2007; Elliott et al, 1994; Kennedy, 1994; McClean et al,
2007; McNally et al, 1988; Reese et al, 1998), and three used
multisensory rooms (Fava and Strauss, 2010; Kaplan et al,
2006; McKee et al, 2007). Of the 10 studies for which we
could calculate an effect size, two showed a significant pos-
itive effect (Boso et al, 2007: 2.13 [0.84, 3.45]; Carminati
et al, 2007: 1.28 [0.30, 2.26]). Boso et al (2007) included
active musical activities (ie, drumming, piano playing, sing-
ing) for a predominantly male (87.5% male) group of eight
adults (M age = 30.2); Carminati et al (2007) included a
structured applied behavioral analysis approach to a resi-
dential program for a predominantly male (78.9% male)
group of 19 adults (M age = 39).
Emotional Functioning
Due to a moderate proportion of studies that
showed a significant effect of the intervention yet showed a
moderate risk of bias, the quality of the evidence sup-
porting the effectiveness of interventions to improve
emotional functioning in adults with ASD–3 was moder-
FIGURE 3. Risk of bias summary: Review authors’ judgments ate. This outcome was investigated in five studies: Cam-
about each risk-of-bias item for each included study. + = high pillo et al (2014), García-Villamisar and Dattilo (2010),
risk. − = low risk. ? = unclear risk. Gerber et al (2011), Kaplan et al (2006), and Shabani and
Fisher (2006). One study used recreational therapies as the
Jerome et al, 2007; Saiano et al, 2015; Smith and Belcher, intervention (García-Villamisar and Dattilo, 2010), three
1985; Van Bourgondien et al, 2003; Vuran, 2008). Of the used behavioral techniques (Campillo et al, 2014; Gerber
seven studies for which we could calculate an effect size, et al, 2011; Shabani and Fisher, 2006), and one used a
none showed a significant effect. multisensory room (Kaplan et al, 2006). Of the two studies
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[–0.37, 1.25] NE; Breen et al
25.67]
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Davis et al
TABLE 3. (continued)
Aggressive/
Type of Activities of Daily Destructive Emotional Language/ Self-Injurious Stereotypy/
Intervention Living Behaviors Functioning Communication Skills Behavior Mannerisms Vocational Skills
Recreational Boso et al (2007), García- Lundqvist et al
Therapy 2.13 [0.84, 3.45]; Villamisar and (2009), 0.57
Lundqvist et al Dattilo (2010), [–0.33, 1.47]
(2009), 0.00 1.93 [1.37, 2.48]
[–0.88, 0.88]
Behavioral Adelinis and Campillo et al Gerber et al (2011), Baker et al (2005), Duker and
Techniques for Hagopian (2014), 1.83 0.09 [–0.57, 0.76]; 7.13 [2.76, 11.49]; Schaapveld
Problem (1999), 2.30 [–0.56, 4.21]; Rehfeldt and Carr et al (1997), (1996), 0.76
Behavior [–1.89, 6.50]; Gerber et al Chambers (2003), 0.55 [–7.30, 8.41]; [–3.95, 5.46];
Reduction Carminati et al (2011), 1.51 2.40 [–1.39, 6.19] Elliott et al (1994), Elliott et al (1994),
(2007), 1.28 [0.75, 2.28]; NE; Hagopian NE; Gerber et al
[0.30, 2.26]; Shabani and et al (2011), 1.96 (2011), 0.87 [0.17,
Elliott et al Fisher (2006), [–4.31, 8.23]; 1.57]; Hanley et al
(1994), NE; NE Kennedy (1994), (2000), 1.60
Kennedy (1994), 0.91 [–6.70, 8.52]; [–7.68, 10.88];
0.91 [–6.70, Kuhn et al (1999), Kennedy (1994),
Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc.
Volume 32, Number 3, September 2019
(1991), 4.90
[–8.65, 10.57]
Multisensory Fava and Strauss Kaplan et al McKee et al (2007), Fava and Strauss
Room (2010), 0.17 (2006), 0.08 0.08 [–1.52, 1.69] (2010), 0.07
[–8.61, 8.95]; [–25.78, 25.93] [ –8.71, 8.84]
Kaplan et al
(2006), 0.32
[–16.65, 17.30];
McKee et al
(2007), 0.20
[–1.81, 1.41]
95% CIs reported. Citations bolded to indicate significant effect size.
NE = no effect size calculated.
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Goodson et al (2007) 3 2.77 [–13.03, 18,58] NS 1 Very high
156 | www.cogbehavneurol.com
Davis et al
TABLE 4. (continued)
Number of Significance of Proportion Significance Bias Quality of
Domain Study Participants Effect Size Effect Size Significant Effect Level Total Bias Level Evidence
Cividini-Motta and 1 2.04 [–6.75, 10.83] NS 0.8 Very high
Ahearn (2013)
Elliott et al (1991) 23 NE 0.6 High
Gaylord-Ross et al 2 7.02 [–1.24; 15.28] NS 1 Very high
(1984)
Gerber et al (2011) 31 0.09 [–0.57, 0.76 NS 0.4 Moderate
Gilson and Carter 2 0.63 [–3.46, 4.73] NS 1 Very high
(2016)
Graff and Gibson 1 5.73 [1.42, 10.03] S 1 Very high
(2003)
Lee et al (2002) 1 0.53 [–8.02, 9.09] NS 1 Very high
Liu et al (2013) 14 0.44 [0.01, 0.88] S 1 Very high
McKee et al (2007) 3 0.08 [–1.52, 1.69] NS 0.6 High
Rehfeldt and 1 2.40 [–1.39, 6.19] NS 0.8 Very high
Chambers (2003)
Sheehan and Matuozzi 1 NE 1 Very high
(1996)
Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc.
Volume 32, Number 3, September 2019
McClean et al (2007) 2 0.63 [–3.45, 4.72] NS 0.8 Very high
McKeegan et al (1987) 1 2.36 [–1.34, 7.90] NS 0.8 Very high
McNally et al (1988) 1 NE 0.8 Very high
Smith (1986) 1 NE 0.8 Very high
Smith (1987) 1 2.55 [–2.72, 7.82] NS 0.8 Very high
Smith and Coleman 3 NE 0.8 Very high
(1986)
Wong et al (1991) 1 4.90 [–8.65, 10.57] NS 0.8 Very high
Stereotypy/ 53 0.25 Low 0.75 High Low
Mannerisms
Duker and Schaapveld 2 0.76 [–3.95, 5.46] NS 1 Very high
(1996)
Elliott et al (1994) 6 NE 0.6 High
Fava and Strauss 9 0.07[–8.71, 8.84] NS 0.2 Low
(2010)
Cogn Behav Neurol Volume 32, Number 3, September 2019 Psychoeducational Interventions for ASD–3
Very high
Very high
Very high
Very high
Moderate
0.8
0.9
0.8
0.8
0.8
Language/Communication Skills
1
1
1
1
1
NS
NS
NS
NS
S
6 [–0.01, 12.02]
[–6.70, 8.52]
[–2.90, 8.42]
[0.61, 8.68]
NE
3
3
14
3
Liu et al (2013)
(1986)
Self-injurious Behaviors
Due to a very low proportion of studies that showed a
Vocational skills
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Davis et al Cogn Behav Neurol Volume 32, Number 3, September 2019
60.00
50.00
40.00
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30.00
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20.00
Effect Size
10.00
0.00
Edrisinha et al 2011
Goodson et al 2007
Haring et al 1987
Jerome et al 2007
Saiano et al 2015
Siaperas & Beadle-Brown 2006
Smith & Belcher 1985
Vuran 2008
Adelinis & Hagopian 1999
Boso et al 2007
Carminati et al 2007
Fava & Strauss 2010
Kaplan et al 2006
Kennedy 1994
Lundqvist et al 2009
McClean et al 2007
McKee et al 2007
Reese et al 1998
Campillo et al 2014
Garcia-Villamisar & Dattilo 2010
Gerber et al 2011
Kaplan et al 2006
Breen et al 1985
Cividini-Motta & Ahearn 2013
Gaylord-Ross et al 1984
Gerber et al 2011
Gilson & Carter 2016
Graff & Gibson 2003
Lee et al 2002
Liu et al 2013
McKee et al 2007
Rehfeldt & Chambers 2003
Siaperas & Beadle-Brown 2006
Baker et al 2005
Carr et al 1997
Hagopian et al 2011
Kennedy 1994
Kuhn et al 1999
Lundqvist et al 2009
McClean et al 2007
McKeegan et al 1987
Smith 1987
Wong et al 1991
Duker & Schaapveld 1996
Fava & Strauss 2010
Gerber et al 2011
Hanley et al 2000
Kennedy 1994
McKeegan et al 1984
Moore 2009
Hume & Odom 2007
Lattimore et al 2008
Lattimore et al 2009
Smith & Coleman 1986
-10.00
-20.00
-30.00
-40.00
FIGURE 5. Forest plot of comparison: Pre- versus postintervention scores, effect sizes (ie, standardized mean differences).
Lundqvist et al (2009), McClean et al (2007), McKeegan et al was a structured behavioral program (autism program with
(1987), McNally et al (1988), Smith (1986, 1987), Smith and a structured method) that focused on the development of
Coleman (1986), and Wong et al (1991). One study used autonomy in a mixed-gender (74.1% male) group of 31 adults
recreational therapies as the intervention (Lundqvist et al, (M age = 43 years), and McKeegan et al (1984) used a non-
2009), and 13 used behavioral techniques (Baker et al, 2005; exclusionary time-out procedure on one 28-year-old man.
Carr et al, 1997; Elliott et al, 1994; Hagopian et al, 2011;
Kennedy, 1994; Kuhn et al, 1999; McClean et al, 2007; Vocational Skills
McKeegan et al, 1987; McNally et al, 1988; Smith 1986, 1987; Due to a very low proportion of studies that showed
Smith and Coleman, 1986; Wong et al, 1991). Of the 10 a significant effect of the intervention yet showed a very
studies for which we could calculate an effect size, one showed high risk of bias, the quality of evidence supporting the
a significant positive effect: Baker et al, 2005: 7.13 [2.76, effectiveness of interventions to improve vocational skills
11.49]. Baker et al (2005) reduced coprophagia in one 45-year- in adults with ASD–3 was very low. This outcome was
old man by introducing flavorful meal options. investigated in six studies: Bennett et al (2010), Hume and
Odom (2007), Lattimore et al (2008, 2009), Liu et al
Stereotypy/Mannerisms (2013), and Smith and Coleman (1986). One of the studies
used a social skills program as the intervention (Liu et al,
Due to a low proportion of studies that showed a sig-
2013), and five used behavioral techniques (Bennett et al,
nificant effect of the intervention yet showed a high risk of
2010; Hume and Odom, 2007; Lattimore et al, 2008, 2009;
bias, the quality of evidence supporting the effectiveness of
Smith and Coleman, 1986). Of the four studies for which
interventions to reduce stereotypy/mannerisms in adults with
we could calculate an effect size, only one showed a sig-
ASD–3 was low. This outcome was investigated in eight
nificant positive effect (Smith and Coleman, 1986: 5.12
studies: Duker and Schaapveld (1996), Elliott et al (1994),
[0.06, 10.18]). The intervention in that study involved on-
Fava and Strauss (2010), Gerber et al (2011), Hanley et al
the-job training with role play, token economies, and
(2000), Kennedy (1994), McKeegan et al (1984), and Moore
differential reinforcement of behaviors for three adult
(2009). Seven studies used behavioral techniques as the in-
males (M age = 26 years).
tervention (Duker and Schaapveld, 1996; Elliott et al, 1994;
Gerber et al, 2011; Hanley et al, 2000; Kennedy, 1994;
McKeegan et al, 1984; Moore, 2009), and one used a multi- DISCUSSION
sensory room (Fava and Strauss, 2010). Of the seven studies To our knowledge, this is the only review of the
for which we could calculate an effect size, two showed a efficacy of available psychoeducational interventions for
significant positive effect (Gerber et al, 2011: 0.87 [0.17, 1.57]; adults with ASD–3. This is not altogether surprising, as
McKeegan et al, 1984: 4.64 [0.61, 8.68]). Gerber et al (2011) published studies into this research niche are rare in
158 | www.cogbehavneurol.com Copyright © 2019 The Author(s). Published by Wolters Kluwer Health, Inc.
Cogn Behav Neurol Volume 32, Number 3, September 2019 Psychoeducational Interventions for ASD–3
comparison to the extensive body of literature on inter- several treatment modalities that are commonly used in
ventions for children with ASD or adults with ASD–1 and children with ASD (including floortime/developmental, in-
–2. Following a broad search, we found only 56 relevant dividual-differences, relationship-based therapy [Solomon et al,
studies, published in the past 50 years, that attempted to 2007], speech-language therapy, occupational therapy, and
quantitatively test the effects of psychoeducational inter- physical therapy) have also not yet been modified for and
ventions on adults with ASD–3. tested in adults with ASD–3. Finally, in our search, we
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Of the seven outcome domains studied (activities of came across studies reporting treatments for caregivers of
daily living, aggressive/destructive behaviors, emotional adults with ASD–3 as the primary participants; although
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functioning, language/communication skills, self-injurious these were outside the scope of our review, these types of
behaviors, stereotypy/mannerisms, and vocational skills), treatments could also be expected to be influential in
only moderately reliable evidence, per Cochrane criteria, improving the lives of individuals with ASD–3.
existed to support the effectiveness of interventions
designed to improve emotional functioning in adults with Considerations for Future Methodologies
ASD–3; reliability of evidence for all other domains was Based on our review, it is clear that studies of psy-
assessed as low or very low. Despite the general lack of choeducational interventions for adults with ASD–3
reliable evidence to support specific interventions, we should be designed to be conducive to and report high-
propose that this review is useful as a guide for future quality evidence, even when investigating this challenging
intervention studies involving the ASD–3 population participant population (for a summary, see Figure 6).
because it provides some insight into how such studies
should be better designed. Study Design
Although randomized controlled trials are the gold
Considerations for Future Research Topics standard in intervention research, they may not be feasible
Other Cultural/Gender Groups for the early evaluation of interventions. Other study de-
The studies reviewed here involved males from signs such as clinical case reports, within-subjects and
Western countries, particularly the United States, almost between-groups experimental studies, and pilot studies
exclusively. Females with ASD–3 have been suspected as may provide a good balance between investigative rigor
having different clinical phenotypes and psychosocial and practicality (Skolasky, 2016; Smith et al, 2007).
factors from males with the same diagnosis (Lai et al,
2015) and arguably are exposed to different sociocultural Data Analysis and Reporting of Results
environments and expectations in most cultures (Lips, There are several ways in which future studies of
2017). Gender, ethnic/cultural, and socioeconomic differ- interventions for adults with ASD–3 could minimize issues
ences are, therefore, likely to be worthwhile factors for relating to the analysis and reporting of results. First, it
investigation, specifically in terms of how these differences would be helpful if studies used both visual and quanti-
may moderate intervention efficacy (Gerber et al, 2017; tative analyses to gain the most accurate inferences from
Singh and Bunyak, 2019). the data and to allow for broader conclusions to be made
across studies (Skolasky, 2016).
Other Outcomes Second, future studies should attempt to be more
The studies we reviewed did not explore inter- rigorous in their handling of missing data. Relevant to
ventions that target a wide range of behavioral outcomes. this, the ASD–3 population presents a number of logistical
Interventions should be studied that target skills crucial to challenges, including, but not limited to, issues related to
daily life, which include food preparation, cleaning and transportation, which increase the likelihood of missing
household chores, personal hygiene and grooming, home data. However, it is important that future studies make
and community safety awareness, budgeting and banking, careful attempts to minimize missing data and address
medication management, shopping, and managing ap- whatever issues arise using rigorous statistical methods
pointments; vocational skills, which include applying for (Skolasky, 2016). At the very least, missing data are
jobs, learning to select professional attire, collaborating a limitation that should be acknowledged. We are opti-
and interacting with coworkers, and managing job stress; mistic that future initiatives will more effectively reduce
and neurocognitive skills, which include attention/executive the impact of missing data, as widely used statistical
functioning, psychomotor abilities, and learning/memory. packages such as R (R Core Team, 2018) allow re-
searchers with no statistical background to effectively
Other Intervention Methods analyze challenging data sets, including those involving
The majority of studies we reviewed tested only small sample sizes and case studies with missing data
applied behavioral analysis and cognitive-behavioral (Skolasky, 2016).
techniques; thus, other intervention methods warrant future Third, although interventions may not show a sig-
study. Many well-known treatments based on behavioral nificant effect size, they may still lead to meaningful
principles (including pivotal response treatment [Koegel behavior change. Thus, future studies should include
et al, 1999], verbal behavior intervention [Skinner, 1957], mixed quantitative and qualitative methodologies so that
and relationship development intervention [Gutstein, 2009]) any clinical impact (even if not statistically significant)
have not yet been studied in adults with ASD–3. Moreover, may be comprehensively reported.
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Cogn Behav Neurol Volume 32, Number 3, September 2019 Psychoeducational Interventions for ASD–3
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