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567579

research-article2015
CCP0010.1177/1359104514567579Clinical Child Psychology and PsychiatryKok et al.

Review
Clinical Child Psychology
and Psychiatry
The effectiveness of psychosocial 1­–16
© The Author(s) 2015
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a psychiatric disorder and mild DOI: 10.1177/1359104514567579
ccp.sagepub.com
intellectual disability to borderline
intellectual functioning: A systematic
literature review and meta-analysis

Lidwien Kok1, Anne van der Waa1, Helen Klip2


and Wouter Staal2,3
1Department of Mild Intellectual Disabilities, Karakter Child and Adolescent Psychiatry, The Netherlands
2KarakterChild and Adolescent Psychiatry University Centre, The Netherlands
3Department of Cognitive Neuroscience, Donders Institute for Brain, Cognition and Behaviour, Radboud University

Medical Centre, The Netherlands

Abstract
Children with intellectual disability frequently have difficulties in adapting to their environment.
The extent of the experienced problems does not only depend on cognitive functioning but is
influenced by other factors, such as the presence of a psychiatric disorder or other brain disorders,
or adverse environmental factors. Several epidemiological studies show that children with
intellectual disabilities are at an increased risk to develop psychiatric disorders. This is also true
for youth with a mild intellectual disability and even those with borderline intellectual functioning
(mild to borderline intellectual disability (MBID)). Psychiatric disorders are often overlooked
because behavioral problems are rather attributed to the intellectual disability. Consequently,
effective psychiatric interventions, which are needed to improve the level of functioning, are
not applied. This review aimed to systematically evaluate the currently available, qualitatively
sound research concerning the effectiveness of psychosocial interventions, specifically directed
at psychiatric disorders in children with MBID. Assessed for eligibility were 1409 unique reports,
and the review ultimately included only 12 reports. Review of the results and meta-analyses
showed that the majority of studies suffer from multiple limitations and that methodological
variations between studies are extensive. This possibly reflects the high variance of factors that
may be involved in MBID. It will be important in future research to address multi-causality.

Keywords
Mild intellectual disability, psychiatric disorder, psychosocial intervention, parent training,
borderline intellectual functioning

Corresponding author:
Lidwien Kok, Department of Mild Intellectual Disabilities, Karakter Child and Youth Psychiatry, Utrechtseweg 320, 6862
BC Oosterbeek, The Netherlands.
Email: l.kok@karakter.com

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2 Clinical Child Psychology and Psychiatry 

Background
According to the Diagnostic and Statistical Manual of Mental Disorders–Fifth Edition (DSM-V)
(American Psychiatric Association, 2013), an intellectual disability (ID) is a neurodevelopmental
disorder that arises early during an individual’s development. It encompasses not only impairments
in intellectual functioning but also adaptive deficits in conceptual, social, or practical domains.
Deficits in intellectual functions, such as reasoning, problem-solving, planning, abstract thinking,
judging, academic learning and learning by experience, and practical understanding, have to be
confirmed by clinical assessment and by individualized, standardized intelligence testing. The
score on the intelligence tests is generally approximately 2 standard deviations (SDs) below the
population mean, that is, <70–75. Different levels of cognitive impairment are defined. Mild intel-
lectual disability (MID) is defined by IQ scores ranging from 50 to 55 to approximately 70, while
scores ranging from 70 to 85 are commonly referred to as borderline intellectual functioning (BIF).
Deficits in adaptive functioning in MID and BIF are defined as a failure to meet developmental and
sociocultural standards for personal independence and social responsibility. This means there is an
ongoing need for support. At the same time, specific interventions are often needed to target loss
of functioning due to psychiatric disorders, neurological disorders and other medical conditions,
and adverse environmental factors.
With respect to psychiatric disorders, it is important to note that these are highly prevalent in
patients with ID (e.g. Dekker & Koot, 2003; Emerson, 2003), while at the same time, they are often
overlooked because the presence of psychiatric symptoms is more likely to be attributed to the ID
than to a psychiatric disorder (Nezu & Nezu, 1994). Prevalence studies have estimated that approx-
imately 38.6% of children with a full-scale IQ score between 30 and 80 have an additional
Diagnostic and Statistical Manual of Mental Disorders–Fourth Edition (DSM-IV) diagnosis
(Dekker & Koot, 2003), compared to approximately 21.5% in the general population (Verhulst,
van der Ende, Ferdinand, & Kasius, 1997). These estimated prevalences are comparable with esti-
mates found in other studies (Emerson, 2003; van Nieuwenhuijzen, Orobio de Castro, & Matthys,
2006; Stromme & Diseth, 2000). In line with these observations, higher rates of emotional and
behavioral problems are found in studies using a dimensional approach. Approximately 50% of
children with (IDs) score within the clinical range on the total problems score on the Child Behavior
Checklist (CBCL) compared to 18% of children from the general population (Dekker, Koot, van
der Ende, & Verhulst, 2002). These observations also hold true for MID and BIF. Both groups
show elevated rates of both externalizing and internalizing problem behavior (Dekker et al., 2002;
Einfeld & Tonge, 1996) and are faced with elevated risks for the development of psychiatric disor-
ders (e.g. Dekker & Koot, 2003; Emerson, 2003).
In the light of the high incidence of psychiatric disorders and related emotional and behavioral
problems experienced by children with an ID, it is highly important to provide the field with an
overview of interventions designed specifically for, or adjusted to, this target group. The principles
of treatment of psychiatric disorders in children with an ID may essentially be the same as in the
general population—predominantly either pharmacological treatments and/or psychosocial inter-
ventions. In this review, we specifically focus on psychosocial interventions, specifically directed
at psychiatric disorders in children with a MID or BIF.
Numerous reports have been written on the elements and concepts that may be effective in treat-
ing individuals with an ID and behavioral problems. Consensus appears to exist that within this
population, it is important to engage the family in the treatment program to ensure generalization
to other contexts (Hurley, 1989; Matson, Mahan, & LoVullo, 2009) and to involve the broader
social context of the individual in the therapeutic process (Jahoda, Dagnan, Jarvie, & Kerr, 2006).
In addition, literature has shown that flexibility should be maintained in using different protocols

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Kok et al. 3

(Hurley, 1989) and that interventions should be adjusted to the abilities and needs of the individual
patient (Szymanski & King, 1999; Hurley, 1989; Willner, 2005) but also to the needs and compe-
tencies of the patient’s family (Gavidia-Payne & Hudson, 2002). Furthermore, the use of tech-
niques involving applied behavior analysis prior to treatment appears to be effective in reducing
problem behavior in children with an ID (e.g. Brosnan & Healy, 2011; Didden, Duker, & Korzilius,
1997; Sturmey, 2012). Even though these reports provide a global idea of the type of treatments
that may be efficacious in reducing symptoms of psychiatry within this target group, a systematic
meta-analysis of the effectiveness of specific treatment programs is lacking. To address this issue,
the current systematic meta-analysis was conducted. This systematic review addresses the follow-
ing research questions:

1. What is the quality of research with respect to the effectiveness of psychosocial interven-
tions for psychiatric disorders in children with mild to borderline intellectual disability
(MBID)?
2. What can be concluded from these studies with regard to effect sizes when compared to a
control condition?

Methods
Systematic search
A literature search was performed in PsycINFO, EMBASE, PubMed, and MEDLINE using com-
binations of search terms of methodology, intellectual disability, and treatment. The interventions
of interest were psychosocial interventions aimed at decreasing or diminishing symptoms of psy-
chiatric disorders that did not involve medication. Studies that met the following criteria were
included : (1) participants were younger than 22 years of age, (2) participants had IQ scores between
50 and 85, (3) participants were diagnosed with any psychiatric disorder, (4) the studies were ran-
domized controlled trials (RCTs) or studies at least contained randomization and blindness, and (5)
the studies were published in a peer-reviewed journal. No language restrictions were applied.
Search terms were adapted to each database. Specific search terms used for the different databases
are provided in the Appendix (can be accessed via the website http://www.karakter.com/
media/20723/appendices-systematic-review-mid-and-psychiatric-problems-ccpp.pdf).

Review of the studies


Two independent reviewers—the first two authors (L.A.M.K. and A.C.W.)—assessed all studies
identified through the electronic searches for inclusion in the review. Reference lists of the selected
relevant articles were reviewed to ensure that no relevant articles had been missed. Titles, abstracts,
and full texts of the identified studies were subsequently reviewed using a study inclusion form to
assess the eligibility of the trials. The quality of the studies was assessed by the same two reviewers
using standardized forms developed by the Dutch Cochrane Centre and included an assessment of
the randomization, blindness, and lost to follow-up. The questions included were as follows: Was
the allocation of participants randomized? Were the participants themselves and the assessors of
effect blind to the allocated condition? Was the professional providing the treatment aware of group
assignment? Were the groups comparable at the start of the intervention? Was selective loss-
to-follow-up sufficiently controlled for? Were all participants analyzed in the group to which they
were assigned? Were the groups treated similarly apart from the treatment? In addition, the studies
were scored using Jadad criteria (Jadad et al., 1996). Jadad and colleagues developed a rating system

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4 Clinical Child Psychology and Psychiatry 

to assess the quality of RCTs that contains assessment of elements such as blinding, randomization,
and dropouts. The instrument consists of three questions yielding a total score between zero and
five. For this current review, a score of 1 out of 5 or higher justified inclusion in the review. In case
of disagreement between the two review authors, the third author (H.K.) was asked to resolve the
dispute.

Data collection and analyses


Data from eligible studies were collected by two independent reviewers using a data extraction
form designed specifically for this review. Extracted data included details of the quality and design
of the study, details of the intervention being studied in each trial, characteristics of the participants
(age, diagnosis, severity of ID), and scores on outcome parameters. All data were entered into
RevMan 5.2 (The Nordic Cochrane Centre, The Cochrane Collaboration, 2012). If included studies
turned out to be sufficiently homogeneous, both clinically and methodologically, meta-analyses
were performed. Where suitable, forest plots were obtained including calculations of chi-square in
order to assess statistical heterogeneity. Where possible, sensitivity analyses were performed to
assess whether the findings were robust against decisions made during the reviewing process.
Funnel plots were produced, plotting the effects found by the inverse of their variance to explore
the presence of publication bias. As a rule of thumb, effect sizes of .2 were considered a small
effect, .5 as a moderate effect, and .8 was considered a large effect (Cohen, 1988). If more than 10
studies were included in the meta-analysis, tests for funnel plot asymmetry were performed, as
recommended by Higgins and Green (2011).

Results
Results of the search
The electronic search yielded a total number of 1825 unique titles, and 16 reports were retrieved
through the screening of reference lists of other eligible studies. After removing duplicates, 1409
reports were considered for screening. Of those, 1331 reports were excluded on the basis of inap-
propriate titles or abstracts; full texts were retrieved of the remaining 78 reports. An additional 58
reports were excluded because the full text showed that they did not meet inclusion criteria.
Reasons for exclusion included more severe ID, the absence of a psychiatric disorder or behavioral
symptoms of psychiatry, or the intervention not meeting inclusion criteria for psychosocial treat-
ments. Another eight reports were excluded because the quality of the research was inadequate. As
a result, the remaining 12 reports of 11 different studies were deemed appropriate for analysis, of
which only 10 could be included in the quantitative analyses. Figure 1 depicts the flow of exclusion
of records in this review.

Description of included studies


A total of 12 reports of 11 unique studies were included in the review, each assessing a different inter-
vention. Characteristics of the included studies are presented in Table 1. The reported outcome meas-
ures, also described in Table 1, vary substantially across studies with very little overlap. Outcome
measures varied from observations of parent–child interaction to reports of perceived parental stress.
Not all included reports were eligible for inclusion in quantitative analyses due to missing data such as
SDs and post-treatment scores for the control group. Change scores accompanied by their SDs were

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Kok et al. 5

Figure 1.  Study flow diagram: process of excluding reports.

not reported in any of the included studies. For this reason, analyses were run on post-intervention
scores. Furthermore, only 6 of the 12 studies meeting inclusion criteria were eligible for pooling of
results in meta-analyses. Standardized mean differences (SMD), rather than mean differences, were
obtained because the studies each used different scales. The SMD is expressed as the effect size in a
study, relative to the variability observed in that study (Higgins & Green, 2011).
Overall, the assessed reports suffer from limited reporting of methods used to reduce the influ-
ence of potential sources of bias, justifying a judgment of “Unclear risk of bias.” In all of the
included reports, blinding of participants and personnel (i.e. implementers of the intervention) was
either not possible or reported unclearly. This had two clear reasons. First, in most interventions,
parents were both implementers of the intervention and assessors of effect. Second, in almost all

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6 Clinical Child Psychology and Psychiatry 

Table 1.  Characteristics of included studies.


Study ID Intervention N Age range Intellectual disability Diagnosis Outcome Jadad
measure score
Ntreatment Ncontrol

Bagner Parent–child 10 12 3–6 years Mild–moderate ODD CBCL 3/5


(2007) interaction
therapy
Dawson Early start 24 23 18– Mint: 61.0, (SD: 9.2)a ASD ADOS 2/5
(2010) Denver model 30 months Mcon: 59,04,
(SD: 8.6)a
Hand Parents Plus 16 13 6–12 years Mild intellectual – SDQ 2/5
(2012) Children’s disability
Programme with
special needs
modifications
Hudson Signposts for 26 26 4.6–19.4 Mostly moderate – PHS 1/5
(2003) Building Better (49%) and mild (47%)
Behavior intellectual disability
Jocelyn Care-giver based 16 19 24– Mint: 58.4, (SD: 27.5)b ASD ABC 3/5
(1998) intervention 72 months Mcon: 67.1,
(SD: 27.5)b
McIntyre Incredible Years 21 23 2–5 years Developmental delay – CBCL 1/5
(2008) Parent Training (VABS composite
Program score 45–85)
Mitchell Cognitive 18 20 13– M: 76.25 (SD: 8.47)c – YSR 2/5
(2011) behavior therapy 18 years
Nestler Social 31 30 15– Borderline – AKI 2/5
(2011) competence 22 years intellectual
training functioning
Rickards Home-based 16 18 3–5 years Mint: 60.2, (SD: 20.0)d ASD PBCL 2/5
(2007) intervention Mcon: 60.6, (SD:
program 21.8)d
Rickards Home-based 34 3–5 years Mint: 60.2, (SD: 20.0)d ASD PBCL 2/5
(2009) intervention Mcon: 60.6,
program (SD: 21.8)d
Roberts Behavioral family 17 15 2–7 years Mint: 61.19, – DBC 2/5
(2006) intervention (SD: 14.64)e
Mcon: 63.79,
(SD: 18.58)e
Smith Intensive early 28 – 18– Total IQ scores ASD CBCL 2/5
(2000) intervention and 42 months 35–75
parent training

SD: standard deviation; ABC: Autism Behavior Checklist; ADOS: Autism Diagnostic Observation Schedule; AKI: Ob-
servational Category System; ASD: autism spectrum disorder; CBCL: Child Behavior Checklist; DBC: Developmental
Behavior Checklist–Parent Version; ODD: Oppositional Defiant Disorder; PBCL: Preschool Behavior Checklist; PHS:
Parenting Hassles Scale, Child behavior subscale; SDQ: Strengths and Difficulties Questionnaire; YSR: Youth Self-Report;
VABS: Vineland Adaptive Behavior Scale; WASI: Wechsler Abbreviated Scale of Intelligence; WPPSI-R: Wechsler Pre-
school and Primary Scale of Intelligence–Revised.
aMullen IQ.
bLeiter IQ and Stanford–Binet Intelligence Scale–Fourth edition.
cWASI IQ risk of bias in included studies.
dWPPSI-R IQ.

studies, the control condition was a wait-list control group, and participants were therefore auto-
matically aware of their treatment condition. Allocation concealment and selective reporting bias
were among the best covered topics in the included reports, resulting in either a judgment of low
risk of bias or unclear risk of bias.

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Kok et al. 7

Table 2.  Forest plot of comparison 1.1—parent training versus treatment as usual: total problem
behavior.

Study Treatment Control  

M SD M SD  
Bagner (2007) 51.9 27.87 83.83 20.44
Roberts (2006) 49.65 24.54 67.2 27.36
McIntyre (2008) 57.62 9.03 91.96 12.25
Hand (2012) 16.44 3.78 16.15 5.13
SMD (95% CI) −0.48 [−0.84, −0.12]
Test for overall effect Z = 2.62 p < .05
Heterogeneity χ2: 0.00 df = 1 p = .16 I2: 43%

SD: standard deviation; SMD: standardized mean differences; CI: confidence interval; df: degree of freedom.

Parent training programs


Total scores of child behavior problems.  As shown in Table 2, four studies were identified assessing
the effect of a parent training program based on behavioral therapeutic principles compared to a
wait-list control group or care as usual on the degree of problem behavior displayed by the child
(Bagner & Eyberg, 2007; Hand, Raghallaigh, Cuppage, Coyle, & Sharry, 2012; McIntyre, 2008;
Roberts, Mazzucchelli, Studman, & Sanders, 2006). Meta-analysis was possible for scores on
dimensional problem behavior rating scales. The means and SDs displayed in Table 2 refer to the
means and SDs reported post-intervention on total problem behavior scores on the used behavior
rating scales, for each of the studies. Each study investigated the effectiveness of a different parent-
ing training program, and the scales used to assess the improvement of the children’s challenging
behavior differed across studies (CBCL, Developmental Behavior Checklist (DBC) and Strengths
and Difficulties Questionnaire (SDQ)). All four studies reported statistically significant reductions
in challenging behavior such as conduct problems, disruptive behavior, hyperactivity, and opposi-
tional behaviors. In addition, two of the four studies (Bagner & Eyberg, 2007; Roberts et al., 2006)
reported an increase in positive behaviors of the child, such as obedience and observed appropriate
behavior. Furthermore, all four studies provided some indication of clinically relevant change and
reported higher proportions of clinically significant change in problem behavior in the treatment
groups than in controls and more children scoring within a low needs range. Meta-analysis of the
results showed a small and statistically significant overall treatment effect for parent training inter-
ventions compared to care as usual. Compared to children in the control group, children of families
that received parent training showed less problem behavior (SMD: −0.48 (95% confidence interval
(CI): [−0.84, −0.12]; Analysis 1.1) n = 64 treatment, n = 63 controls). Sensitivity analysis showed
that adding the results of Hand et al. (2012) to the meta-analysis reduced the effect from moderate
to small; the SMD changed from −0.65 (CI: [−1.06, −0.24]) to SMD: −0.48 (CI: [−0.84, −0.12]).
Apparently, the effect of parent training compared to the control group found in Hand et al. was
smaller than that found in the other three studies. Interestingly, Hand et al. (2012) did conclude a
significant group by time interaction. This incongruence may have been caused by the fact that the
intervention group scored higher on total problems prior to the intervention and therefore showed
a larger reduction in problem behavior. A funnel plot was created, but because of the small amount
of studies included in the meta-analysis, tests for funnel plot asymmetry were not performed.
Smith, Groen, and Wynn (2000) compared parent training to an intensive early intervention in
families with a child with an autism spectrum disorder (ASD) and also used scores on the CBCL

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8 Clinical Child Psychology and Psychiatry 

Table 3.  Forest plot of comparison 1.2—parent training versus treatment as usual: child autism
symptoms.

Study Treatment Control  

M SD M SD  
Dawson (2010) 6.5 1.5 7.3 2.1
Jocelyn (1998) 48.5 18.4 56.5 20.1
SMD (95% CI) −0.42 [−0.86, 0.02]
Test for overall effect Z = 1.88 p = .06
Heterogeneity χ2: 0.00 df = 1 p = .95 I2: 0%

SD: standard deviation; SMD: standardized mean differences; CI: confidence interval; df: degree of freedom.

to assess effectiveness. There was no no-treatment control group, but participants were randomly
allocated to either one of the treatment arms, leading to a high risk of bias. Results of the study
showed that the intensive early intervention was more effective in improving the child’s cognitive
abilities and language development. However, there was no difference in follow-up scores on the
CBCL between the intensive intervention group and the less intensive parent training group.

Antisocial and disruptive behavior.  Hudson et al. (2003) assessed the effectiveness of the Signposts
Building Better Behavior program. They compared three different modes of delivery—group sup-
port, telephone support, and self-directed—to a wait-list control group. The design was semi-
randomized because parents were allowed to switch groups if they had difficulty traveling the
distance. The effect of the parenting program was assessed on the antisocial behavior subscale and
the disruptive behavior subscale of the DBC. Meta-analysis of the results was not possible because
SDs were not reported and because the assessment of problem behavior was only done prior to the
intervention and at follow-up. Therefore, results could not be compared to the control group that
also received the intervention after the post-intervention assessment. The authors concluded that
the intervention was effective in reducing antisocial behavior and disruptive behavior and that
there were no differences between the different modes of delivery.

Child autism symptoms.  Two studies were identified that investigated the effect of a parent training
program, compared to care as usual, that assessed its influence on child autism symptoms (Table 3).
Both studies reported an RCT design, and assessment of the quality of reporting of the methods used
resulted in Jadad scores of 2 and 3. Overall, randomization was conducted appropriately and asses-
sors of effects were blind to the condition of the participants. In both studies, autism was diagnosed
according to Diagnostic and Statistical Manual of Mental Disorders–Third Edition, Revised (DSM-
III-R) or DSM-IV criteria, and the presence of a MID was not one of the inclusion criteria but
reported mean IQ scores fell within the 50–85 range.
Both studies (Dawson et al., 2010; Jocelyn, Casiro, Beattie, Bow, & Kneisz, 1998) concluded
that the parent training effectively stimulated the development of these children. For example, they
found that the intervention group children developed better language and communicative skills,
better daily living skills, and significantly higher levels of adaptive functioning. However, both
studies reported no significant results on specific instruments for autism-related behavior or repeti-
tive behavior. The authors did, however, report a trend toward reduction of autistic behavior.
Dawson et al. (2010) found that a statistically significantly larger proportion of the intervention
group improved from an autism disorder diagnosis to a diagnosis of Pervasive Developmental
Disorder–Not Otherwise Specified (PDD-NOS) when compared to the control group.

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Kok et al. 9

Table 4.  Forest plot of comparison 1.3—parent training versus treatment as usual: Preschool Behavior
Checklist (PBCL).

Study Treatment Control  

M SD M SD  
Rickards (2007) 13.7 8.3 21.2 6.7
SMD (95% CI) −7.50 [−12.61, −2.39]
Test for overall effect Z = 2.88 p < .01

SD: standard deviation; SMD: standardized mean differences; CI: confidence interval.

Meta-analysis of the results revealed a treatment effect approaching statistical significance of


parent training compared to care as usual, on scores on two different autistic behavior rating scales
(SMD: −0.42 (95% CI: [−0.86, 0.02]; Analysis 1.2) n = 40 treatment, n = 42 controls). The two
studies were highly similar in the effects found: χ2(degree of freedom (df) = 1): 0.00; p = .95; I2: 0%.
A funnel plot was created, but because of the small amount of studies included in the meta-analy-
sis, tests for funnel plot asymmetry were not performed.

Preschool behavior.  Two reports from one study meeting the inclusion criteria (Rickards, Walstab,
Wright-Rossi, Simpson, & Reddihough, 2007; Rickards, Walstab, Wright-Rossi, Simpson, & Red-
dihough, 2009) assessed the influence of a home-based intervention program for children with
autism and developmental delay and compared it to children only receiving a center-based pro-
gram (Table 4). The effects were assessed on a preschool behavior rating scale. The authors ana-
lyzed the children with and without a diagnosis of an ASD separately. The range of IQ scores of the
sample was unclear, but the reported mean IQ scores fell within the 50–85 range for both develop-
mental delay children and ASD children.
Rickards et al. (2007) concluded that the additional home-based intervention significantly
improved cognitive ability of the children. Children in the intervention group had increased IQ
scores, whereas controls had decreased scores. In addition, they found a significant improvement
in preschool behavior (interaction with other children, anxiety, emotional control, etc.) for children
with a developmental delay. Current analysis of the results did show a significant effect size (SMD:
−7.50 (CI: [−12.61, −2.39]; Analysis 1.3) n = 16 treatment, n = 18 controls). At the follow-up 1 year
after completion of the intervention, the effect on cognitive development was maintained, but the
effect on preschool behavior had disappeared (Rickards et al., 2009).

Parental stress.  Meta-analysis summarizing the results on parental stress before and after treatment
was not possible because of the different scales used to assess the levels of perceived parental
stress. Few studies report significant reductions in perceived parental stress levels (Hand et al.,
2012), increased sense of being able to manage their role as a parent and the feeling that their needs
as a parent were met (Hudson et al., 2003). Other studies targeting families of children with MBID
and problem behavior suggest that parental stress is not reduced significantly as a result of the par-
ent training interventions (Bagner & Eyberg, 2007; McIntyre, 2008; Rickards et al., 2007; Roberts
et al., 2006). According to some authors, this may be due to the fact that pre-intervention levels of
reported parental stress were already low (Bagner & Eyberg, 2007; Roberts et al., 2006).

Social competence training


Only one study that met our inclusion criteria assessed the effectiveness of a social competence
training in children with an ID compared to standard care (Table 5). The training was designed

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10 Clinical Child Psychology and Psychiatry 

Table 5.  Forest plot of comparison 2.1—social competence training versus treatment as usual:
observation of socially competent behavior.

Study Treatment Control  

M SD M SD  
Nestler (2011) 11.88 3.57 10.99 2.68
SMD (95% CI) 0.89 [−0.69, 2.47]
Test for overall effect Z = 1.10 p = .27

SD: standard deviation; SMD: standardized mean differences; CI: confidence interval.

specifically for adolescents with borderline intelligence. Nestler and Goldbeck (2011) used an
RCT, and assessors of effect were blind to the participants’ experimental condition. Assessment of
the quality of the design by the review authors yielded a Jadad score of 2 out of 5. The study
included participants with total symptom scores above average (T-scores above 60) on one of the
global scores of the CBCL. The social competence training focused on three domains of social
competence: cognitive, behavioral, and emotional aspects. Nestler and Goldbeck concluded that
the training was effective in attaining individually set goals such as staying calm when someone
criticized them. In addition, the training was effective in improving cognitive aspects of social
competence. No treatment effect was found on observed socially competent behavior, for instance,
during role play. Perceived social anxiety and contact anxiety were also not affected by the
training.
A non-significant effect of treatment was found for social competence training on socially com-
petent behavior compared to care as usual (SMD: 0.89 (CI: [−0.69, 2.47]; Analysis 2.1) n = 31
treatment, n = 30 controls). The authors reported a significant time effect on observed socially
competent behavior, but there was no difference in effect between the treatment group and control
group.

Cognitive behavior therapy


One study investigated the effectiveness of a cognitive behavioral therapy (CBT) for adolescent
offenders compared to care as usual (Table 6). This study received a Jadad score of 2 out of 5 based
on the quality of the reported methods. The randomization of this multi-center RCT was done
appropriately, and assessors of effect were blind to the experimental condition of the participants.
The presence of a MID was not one of the inclusion criteria, but the mean total IQ scores were
within the 50–85 range. All participants in the treatment arm met International Statistical
Classification of Diseases and Related Health Problems–10th Revision (ICD-10) criteria for con-
duct disorder; five participants also had post-traumatic stress disorder (PTSD), and two were diag-
nosed with attention-deficit hyperactivity disorder (ADHD). Mitchell et al. (2011) concluded that
the intervention was not effective in reducing the number of mental health needs. However, CBT
was shown to be effective in reducing externalizing problems, as reported by the adolescents on the
Youth Self-Report (YSR). The effect of the cognitive behavioral intervention on self-reported total
problems, when compared to care as usual in this study, was non-significant (SMD: −1.12 (CI:
[−19.36, 17.12]; Analysis 3.1) n = 18 treatment, n = 20 controls). With respect to the outcome meas-
ures used, Mitchell et al. concluded that they may not reliably reflect real changes in behavior
because they rely on the adolescents’ ability to self-reflect. The lack of significant effects found in
this study was attributed by the authors to the use of inappropriate outcome measures, the long time

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Kok et al. 11

Table 6.  Forest plot of comparison 3.1—cognitive behavior therapy versus treatment as usual: YSR total
problem score.

Study Treatment Control  

M SD M SD  
Mitchell (2011) 55.83 33.83 56.95 21.46
SMD (95% CI) −1.12 [−19.36, 17.12]
Test for overall effect Z = 0.12 p = .90

YSR: Youth Self-Report; SD: standard deviation; SMD: standardized mean differences; CI: confidence interval.

period between baseline and follow-up assessments, and the treatment not being focused enough
on internalizing problems such as PTSD, anxiety, and depression.

Discussion
The aim of this systematic review and systematic meta-analyses was to evaluate the effectiveness
of psychosocial interventions in children with MBID and a psychiatric disorder. The vast majority
of the included studies investigated the effectiveness of a parent training intervention compared to
care as usual. The remaining studies focused on psychosocial training programs for the children
and adolescents. Parent training programs focus on improving parent–child interactions, increasing
parents’ understanding of their child’s behavior, and the application of behavioral techniques to
reduce problem behavior. In this systematic review, seven different parent training programs were
assessed in a total of 243 participants with varying degrees of psychopathology. The overall results
appear to show a tendency toward reduced problem behavior and an increase in child positive
behavior.
The effectiveness of the parent training programs was explained through the increase in positive
parent–child interactions (Bagner & Eyberg, 2007), decrease in negative parenting behaviors
(McIntyre, 2008), and decrease in observed over-reactivity in mothers (Roberts et al., 2006). Some
studies also assessed the clinical significance of the results and showed positive changes such as a
decrease in diagnostic severity (i.e. from autistic disorder to PDD-NOS). Results on parental stress
levels are currently equivocal, with some authors arguing parental stress was already low at the
start of the intervention and others demonstrating reduced stress levels. Because of the relatively
small amount of included studies, extensive subgroup analyses were not possible. However, there
do not appear to be major differences in effect sizes between individual training or training in
groups.
Only two studies were identified that investigated the effectiveness of psychosocial training
programs for the children and adolescents themselves, administered either in groups or individu-
ally. The interventions appear to have an effect on specific domains of problem behavior, although
definitive conclusions cannot be drawn at this moment.
In this meta-analysis, over 1400 studies were assessed for eligibility, and one might expect that
this would result in clear recommendations for the daily clinical practice. Unfortunately, it has to
be concluded that the overall quality of the studies assessed was relatively poor, creating a high risk
of bias. Even though most studies report to have used a randomized controlled design, randomiza-
tion was not always conducted properly, and none of the studies were performed in a double-blind
manner. It should also be noted that the risk of bias often remained unclear due to poor reporting
of methods employed to reduce the risk of bias. Previous reviews have also reported low quality of

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12 Clinical Child Psychology and Psychiatry 

evidence in this field of research, typically marked by a lack of control groups and small-sized
intervention groups (Beavis, Kerr, Marson, & Dojcinov, 2011; van Nieuwenhuijzen et al., 2006;
Prout & Nowak-Drabik, 2003; Roberts, Mazzucchelli, Taylor, & Reid, 2003).
Across studies, the number of dropouts strongly varied, from studies with no dropouts to studies
with an attrition rate of 43%. While the amount of dropouts was often mentioned, the reasons for
loss to follow-up were rarely reported. Moreover, the differences between participants who
remained in the intervention and those who were lost to follow-up were often not analyzed. In addi-
tion, an intention to treat analysis was performed in only three studies. In some studies, the authors
merely stated that families that dropped out of the study differed in baseline characteristics—such
as being more likely to have an autistic child (Hudson et al., 2003) or having lower levels of family
functioning (Rickards et al., 2009)—but the consequences of this bias were not explored.
Furthermore, in this meta-analysis, it was not possible to control for positive publication bias. This
was due to the small amount of studies which made it impossible to test for funnel plot asymmetry,
although visual inspection of the funnel plots did not suggest such a bias.
Our results are in line with previous studies that investigated the effectiveness of psychosocial
interventions in children with ID. For instance, Harvey, Boer, Meyer, and Evans (2009), in their
update of a study conducted by Scotti, Evans, Meyer, and Walker (1991, as described in Harvey
et al., 2009), found effect sizes (SMDs) of single treatment approaches between .44 and .57.
Furthermore, Heyvaert, Maes, and Onghena (2010) found a combined effect size for all included
interventions of .67 (ranging between .22 and 1.41). Prout & Nowak-Drabik (2003) reported
“moderate” effect sizes of psychotherapy in adults and children with an ID. Overall, smaller effects
of therapy were found than were previously reported in children from the general population.
Kazdin (2003) reported rather large effect sizes of therapy of approximately .70.
This review has some limitations, which mainly relate to the fact that during the conduct of this
review, it was decided to apply the inclusion criteria less strictly. This made the data more hetero-
geneous and affected the quality of included studies. The reason behind applying more flexible
inclusion criteria was to be able to include a larger amount of studies, which would better reflect
the research initiatives in the field. With respect to this decision, it is important to realize that con-
ducting a systematic review is an iterative process, dependent on the quality of identified studies
(Moher, Liberati, Tetzlaff, Altman, & The PRISMA Group, 2009). One of the most important
effects of using broader inclusion criteria was that more heterogeneous data with respect to intel-
lectual abilities and psychiatric diagnoses were collected. IQ ranges between studies and subjects
were wide, and the presence of problem behavior was often not properly defined (such as a cut-off
score on a rating scale). In addition, because exact data on full-scale IQ scores was often not
reported, studies were included that reported a sample mean, rather than a range, of IQ scores up
to 85. As a result of this, children with average IQs (above 85) were also included in the review.
Another limitation concerns the pooling of interventions that were based on the same therapeutic
principles but may in practice have differed from one another.
Furthermore, studies were included even though randomization or blinding was not performed
completely according to common standards, yielding a higher risk of bias. In at least one study, it
was clear that the control group and intervention group differed in the amount of problem behavior
prior to the start of the intervention.
Despite these limitations, our study was, to the best of our knowledge, the first formal meta-
analysis that focused on children with a MID to BIF (i.e. IQ scores between 50 and 85) and a
psychiatric disorder. Previous studies have not been dedicated to psychosocial treatments or have
not used systematic search strategies. Also, previous studies have included wider ranges of (intel-
lectual) disabilities. The most salient result of this review was that it was highly difficult to find
studies that strictly met the set out inclusion criteria. The way in which intellectual or

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Kok et al. 13

developmental disabilities and problem behavior were defined differed markedly across studies,
and only one study (Bagner and Eyberg) included subjects with a formally diagnosed psychiatric
disorder. The fact that psychiatric disorders were rarely diagnosed in the different studies is
remarkable given the high prevalence of psychopathology in children with an ID. This may be
due to the fact that very few diagnostic instruments are currently available which are specifically
appropriate for this population.
One of the practice parameters, as formulated by the American Academy of Child and Adolescent
Psychiatry (Szymanski & King, 1999), is that treatment techniques need to be modified to meet the
needs of patients with an ID. This may especially be true for families in which parents also cope
with an ID. Parents should be addressed appropriately, taking into account their own level of intel-
lectual functioning as well. This appears vital not only when the intervention of choice concerns a
parent training program but may also be important in other categories of treatment (de Koning &
Collin, 2007; de Wit, Moonen, & Douma, 2011). In this systematic review, almost all of the
included studies assessed interventions that were either designed specifically for intellectually
disabled children or were modified to fit their needs. Only Bagner and Eyberg (2007) studied a
non-modified parent training program and assessed its applicability for children with a comorbid
ID. Their results indicate that modifications might not be necessary for the intervention to be effec-
tive. This result needs replication.
Practical implications of the present review include that parent training interventions do appear
to have a positive effect in reducing problem behavior in children who have an ID and a psychiatric
disorder. It would therefore appear to be worth considering using these treatments prior or in addi-
tion to other forms of treatment, such as pharmacological interventions. This is especially interest-
ing, given the high rates of reported side effects of pharmacological interventions in children with
an ID (e.g. Antochi, Stavrakaki, & Emery, 2003; Handen & Gilchrist, 2006).
A number of directions for future research can be deduced from our review. Adequately con-
trolled studies with large sample sizes will have to be conducted. These should allow the identifica-
tion of factors that determine treatment outcome and account for the fact that the population of
children with an ID, is highly heterogeneous (e.g. de Koning & Collin, 2007; Janssen & Schuengel,
2010). Analysis of how individual characteristics, such as specific symptoms and level of impair-
ment, that go beyond classification of disease, relate to the efficacy of interventions should be con-
ducted. This will require that participants should be diagnosed properly, both with regard to ID and
with regard to psychiatric disorders, but it will also be required to address psychopathology that is
not adequately conceptualized as a discrete disorder, or even comorbid diagnoses. The level of func-
tioning in different domains should be assessed systematically, and this assessment should contrib-
ute to the diagnosis of the presence of an ID. Furthermore, diagnostic instruments for psychopathology
that are commonly used in the general population should be either adjusted to the intellectually disa-
bled population or tested for validity in this population. Researchers should keep in mind that self-
report may be of limited value in case of ID and that parent ratings scales may introduce bias.
In conclusion, we interpret that the research efforts so far show the complexity of ID and the
difficulty to conduct research in this field. Nevertheless, some promising psychosocial interven-
tions for children with MBID are available. Building upon these interventions and addressing
multi-causality are likely to be of great value. This will require the conduct of studies with large
numbers of subjects, which can only be achieved in collaborations between multiple high exper-
tise centers.

Funding
This research received no specific grant from any funding agency in the public, commercial, or not-for-profit
sectors.

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14 Clinical Child Psychology and Psychiatry 

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Author biographies
Lidwien Kok is a Child- and Adolescent sychiatrist working at Karakter Child and Adolescent Psychiatry in
the Netherlands. She specialised in diagnosing and treating children with Mild Intellectual Disability and
behavioral disorders.
Anne van der Waa is a child psychologist and junior researcher at Karakter Child and Adolescent Psychiatry,
specialized in conducting field research and treating and diagnosing children and adolescents with borderline
intellectual functioning to mild intellectual disabilities and psychiatric disorders.
Helen Klip is epidemiologist at Karakter Child and Adolescent Psychiatry in the Netherlands. She is the pro-
jectmanager for numerous research projects and is specialized in Routine Outcome Monitoring and Evidence
Based Medicine.
Wouter Staal is a Child- and Adolescent psychiatrist working at the department of psychiatrie of the Radboud
University Medical Hospital, The Netherlands. He also works at Karakter, center for Child and Adolescent
Psychiatry and the Donders Institute for Neurosciences.

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