Title page

Emotion recognition training in Autism Spectrum Disorder: a systematic review of

challenges related to generalizability

Steve Berggren1,2, Sue Fletcher-Watson3, Nina Milenkovic1, Peter B Marschik1,4 Sven Bölte1,2 and Ulf

Jonsson1,2,5

1Center of Neurodevelopmental Disorders (KIND), Pediatric Neuropsychiatry Unit, Department of

Women’s and Children’s Health, Karolinska Institutet, Stockholm, Sweden;2Child and Adolescent

Psychiatry, Child and Adolescent Psychiatry, Center of Psychiatry Research, Stockholm County Council,

Sweden; 3Moray House School of Education, University of Edinburgh, Edinburgh, UK.; 4Institute of

Physiology, Research Unit iDN (interdisciplinary Developmental Neuroscience), Medical University of

Graz, Austria; 5Department of Neuroscience, Child and Adolescent Psychiatry, Uppsala University,

Sweden.

Conflict-of-interest notification

All authors declare: no support from any organization for the submitted work; no financial relationships

with any organizations that might have an interest in the submitted work in the previous three years; no

other relationships, activities or financial interests that could appear to have influenced the submitted

work. Sven Bölte has authored the Frankfurt Test and Training of Facial Affect Recognition (FEFA)

mentioned in this review. The FEFA is distributed by the Center of Neurodevelopmental Disorders at

Karolinska Institutet (KIND), headed by Sven Bölte, against compensation, which is used for

maintenance and support of the instrument.

Corresponding Author:

Steve Berggren, Center of Neurodevelopmental Disorders at Karolinska Institutet (KIND), CAP Research

Center, Gävlegatan 22, 11330 Stockholm, Sweden; E-mail: Steve.Berggren@ki.se

Emotion recognition training in Autism Spectrum Disorder: a systematic review of

challenges related to generalizability

Abstract

Objective: To assess the generalizability of findings from randomized controlled trials (RCTs) evaluating

emotion recognition (ER) training for children and adolescents with Autism Spectrum Disorder (ASD).

Methods: We present a systematic review and narrative synthesis of the determinants of external validity

in RCTs on ER training. Generalizability of the findings across situations, populations, settings, treatment

delivery, and intervention formats was considered.

Results: We identified 13 eligible studies. Participants were predominantly boys with ASD in the

normative IQ range (IQ>70), with an age span from 4 to 18 years across studies. Interventions and

outcome measures were highly variable. Several studies indicated that training may improve ER, but it is

still largely unknown to what extent training effects are translated to daily social life.

Conclusion: The generalizability of findings from currently available RCTs remains unclear. This

underscores the importance of involving children with ASD and their caregivers in informed treatment

decisions.

Key words

Neurodevelopmental disorders, autism, emotion, training, technology, intervention, Asperger syndrome,

child psychiatry

Introduction

Autism Spectrum Disorder (ASD) is a neurodevelopmental condition with a heterogeneous phenotypical

presentation. ASD is defined by impairments in social communication and interaction alongside repetitive,

stereotypic activities and interests, as well sensory processing alterations [1]. Current international,

epidemiological research suggests that between 1 and 2% of the general population present with ASD

[2,3]. Psychiatric and somatic comorbidity is common in ASD, with intellectual disabilities, ADHD and

epilepsy being amongst the most frequent co-existing complications [4]. There are a number of therapeutic

approaches which aim to alleviate the challenges experienced by individuals with ASD and improve

outcomes [5].

Emotion recognition (ER) plays an important role in social communication. The ability to attend to socio-

emotional cues (e.g., facial expressions, tone of voice, and body language), interpret them correctly and

respond to them appropriately is vital for successful everyday social interaction. This ability, in turn,

depends on social orientation/attention, mentalizing/theory of mind (ToM, the ability to understand one’s

own and other’s thoughts, beliefs, and internal states) [6], empathy, and social awareness. Difficulties in

ER are related to various mental disorders, but are particularly prominent in ASD [6,7]. Varying degrees

of ER alterations have been demonstrated along the spectrum, although some problems might appear mild

or insignificant owing to compensatory strategies [8,9]. Difficulties include recognition of emotions from

facial expressions [10,11], vocal intonation patterns [12], gestures and body language [13], and the

integration of multimodal emotional information in context [14,15]. Attentional distractibility has been

reported to be associated with facial affect recognition [16]. ER difficulties among children with ASD

might decrease with age, but remain present to a certain extent [17]. Alterations of ER in ASD are a well-

recognized clinical difficulty and can be a significant risk factor for social exclusion, peer rejection,

bullying or isolation [18]. Deficits in ER may also contribute to adverse long-term outcomes such as low

occupational attainment, poor social relationships, and psychiatric comorbidity [19].

Training of ER skills towards the typical range on a behavioral and neurobiological level is a viable

intervention strategy [20]. A wide range of different interventions has been developed, aiming to improve

these abilities in people with ASD. Most, but not all, of the currently available ER training programs are

computer-based. Computerized environments are assumed to provide a favorable setting for users with

ASD, as they can be programmed to be predictable, consistent, and free from immediate social stressors.

Users can work at their own pace and level of understanding and lessons can be repeated over and over

again, until mastery is achieved. Interest and motivation are usually more easily maintained by

individually selected computerized rewards [21]. Computer-based ER training programs tend to be rather

specific (e.g., focusing only on recognition of facial expressions of photographs), and low budget [22].

Among the ER training programs that are used in current clinical practice are The Frankfurt Test and

Training of Facial Affect Recognition, FEFA (training of emotion recognition from photographs of facial

expressions and strips of the eye region) [23], the Emotion Trainer (teaches emotion recognition of

emotions from facial expressions) [24], Let’s Face It! (teaches emotion and identity recognition from

facial expressions) [25], and Mind Reading (an interactive guide to emotions that teaches recognition of

emotions and mental states) [11,22].

Attempts to train emotion processing, predominantly facial affect recognition [23, 26], have yielded mixed

results. A systematic review by Fletcher-Watson et al. [27] on ER and ToM programs evaluated the

effects of 22 randomized controlled trials (RCTs) including a total of 695 participants. Studies were highly

variable in sample size, age-range of participants, intervention delivery type, outcome measures, and

country of origin. A meta-analysis of a subset of four studies showed that interventions targeting ER

across age groups and within the average range of intellectual ability had a positive effect on the target

skill, measured by a test using photographs of faces (mean increase of 0.75 points, 95% confidence

interval (CI) 0.22 to 1.29 points, N = 105). These preliminary results are promising and indicate that

individuals with ASD could benefit from increased access to ER training. However, the authors note that

there was little evidence of generalization of training beyond the taught skill, or to real world settings.

We are currently at a point in time where scientific support for ER training is emerging and programs

gradually become available in routine clinical practice. However, successful implementation and judicious

use of the interventions requires reliable judgments about generalizability across different populations,

training programs, and settings. It is also of great importance to clarify to what extent the training effects

are translated into social skills in real-life situations, which presumably is the main objective for the

person undertaking the training. Generalizability is a major challenge when designing and using training

tools for individuals with ASD in general. In a recent review of cognitive training technologies in the

treatment of ASD, Wass and Porayska-Pomsta [28] reported several promising findings. However, they

concluded that observed improvements within the computerized training paradigm often fail to generalize

to altered behavior in more naturalistic settings, why the external validity of the accumulated research is

challenged.

While internal validity (i.e., the results are valid for the specific study population and setting) often has

high priority in primary research and systematic reviews [29], external validity (i.e., generalization across

populations and situations) is often neglected [30]. External validity is usually sensitive to factors such as

the setting of the study, selection of participants, characteristics of included participants, differences

between the protocol and routine practice, choice of outcome measures, follow-up timing, providers’

characteristics and training, treatment fidelity, number of participants, and trainer ratio. An overview of

these aspects could both inform clinical practice and help to identify future directions in this research

field.

The current review examined aspects related to the generalizability of RCTs on ER training in children

and adolescents with ASD. Close examination of these aspects was not within the scope of the recent

systematic review by Fletcher-Watson et al. [27]. We decided to focus exclusively on RCTs for two

reasons. First, the high demands on internal validity in RCTs can lead to less focus on external validity.

Second, results from RCTs are frequently generalized across populations and contexts even when the

external validity is limited. Our overarching aim was to draw clinical and scientific attention to the

importance of external validity in RCTs, at a time when several ER training programs already have been

evaluated in controlled efficacy studies. We did so in two ways:

1. We used a check-list [31] to fine-map determinants of external validity (treatment intervention,

treatment provider, population, context, and outcome) in available RCTs.

2. We investigated to what extent the study results indicated that improved ER generalized to daily social

skills.

Methods

Eligibility criteria

Types of study: RCTs published in English.

Participants: Children and adolescents up to 18 years of age with a diagnosis of ASD, including autism,

atypical autism, Asperger’s syndrome (AS), and pervasive developmental disorder not otherwise specified

(PDD-NOS), according to either International Classification of Diseases, tenth edition (ICD-10) [37],

Diagnostic and Statistical Manual of Mental Disorders, fourth (DSM-IV) [38] or fifth edition (DSM-5)

criteria [1].

Interventions: Any intervention or part of a broader intervention approach for which the authors explicitly

state at least one of the following: The study (1) was designed to teach emotion recognition, exclusively or

in combination with other skills; (2) was designed to teach precursor skills of emotion recognition; (3) was

based on common emotion recognition theories of autism; (4) aimed to test common emotion recognition

theories of autism.

Comparator: Any of the following comparators: (1) Treatment-as-usual/wait-list control; (2) ‘placebo’

interventions, for example a ‘contact control’ such as watching Thomas the Tank Engine DVDs; (3)

intervention with no therapeutic content, (e.g. group leisure activities); (4) any active treatment.

Outcome: Any behavioral outcome related to ER or social skills.

Information sources

Cochrane (Wiley); Medline (Ovid); Embase (Elsevier); Cinahl (Ebsco); Psychinfo (Ovid); Social Services

Abstracts (Proquest); ERIC (Ovid) were searched up to November 2015.

Search strategy

Electronic searches were conducted using Medical Subject Headings (MeSH) and relevant text word

terms. The search strategy used by Fletcher-Watson and colleagues [27] in their Cochrane review of

interventions based on ToM cognitive model for ASD was repeated. However, in order to limit the search

to the ER interventions the search was combined with a filter for emotion expression/recognition (see

Appendix 1 for the full search strategy in Medline).

Study selection

Two authors independently screened titles and abstracts of all the citations identified by the search

strategy. Potentially relevant articles were screened for eligibility in full text. Any disagreements were

resolved by discussions. Reference lists and systematic reviews were screened for additional relevant

studies.

Data collection process

From each included study data were extracted and inserted in an extraction sheet by two authors. Any

disagreements were resolved by discussion. If no agreement could be reached, it was planned a third

author would decide in case of a disagreement. The third author arbitration was never required.

Data items

1. A checklist of items relevant to external validity was used, compiled by a recent review of social skills

group interventions for children and adolescents with ASD [31]. Included items were clustered into

overarching themes related to external validity: treatment intervention, treatment provider, population,

context, and outcome. All items from this extensive checklist were extracted, and the items most relevant

for this specific review were included in Table 1 and 2.

2. In order to investigate the extent to which the study results indicated that the training generalized to

daily social skills, mean and standard deviation for each group at post-treatment were extracted on

outcome measures related to ER and social skills.

Planned methods of analysis

Two types of analyses were performed:

1. The extracted information on determinants of external validity was summarized in a narrative synthesis

for each separate theme. For each theme, we also briefly summarize our judgement about generalizability.

2. To evaluate how the ER training generalized to daily social skills, we examined for each separate study

to what extent improvements in ER was accompanied by an improvement in social skills. We looked at

interventions specifically targeting ER separate from interventions combining ER training and social skills

training. Thereby, it would be possible to examine if specific ER training resulted in improved social

skills, or if additional social skills training was required for this. For each separate study, the standardized

mean difference (Hedge’s g) and 95% confidence interval were calculated for all measures of ER and

social skills. The mean and standard deviation for each group at post-treatment (and at follow-up when

available) was used for these analyses, which were conducted with Review Manager (RevMan) Version

5.3.4.

Results

Study selection

The search provided 3521 unique citations. Of these, 3493 were discarded because they did not meet the

eligibility criteria. The large number of excluded citations does not reflect strict eligibility criteria, but

rather that we used an over-inclusive search strategy in order not to miss relevant studies. The remaining

28 articles were examined in full-text. A total of 13 studies presented in 14 publications were found to be

eligible [15,24,25,34-44] (see Figure 1). The 14 remaining were excluded, as they did not meet our

eligibility criteria for population (n=8), intervention (n=2), design (n=1) or publication type (n=3)

(Appendix 2).

Figure 1 about here

Treatment intervention

Narrative synthesis: The evaluated interventions were variable regarding treatment components and

format (Table 1). Four of the 13 eligible studies evaluated programs combining ER training with other

treatment components. Two of these studies [38,42] evaluated a comprehensive summer program, which

included a face-emotion recognition curriculum as an integral part of a 5-week social treatment program;

one study evaluated a computerized multi-component social skills intervention program, The Junior

Detective Computer Program, designed for children with AS [34]; and one study evaluated the Social

Adjustment Enhancement Curriculum, a group intervention targeting ER, ToM, executive functions, and

conversational skills [41].

The remaining nine studies evaluated programs primarily targeting ER. Two studies evaluated the “The

Transporters”, an animated three-dimensional series designed to enhance emotion comprehension in

children with ASD [15,44]. Two studies tested “FaceSayTM”, a computer-based social skills training

program with various games designed to teach specific face-processing skills for social cognition [37,39].

One study tested “Mind Reading (MR)”, an interactive software program designed to teach recognition of

simple and complex emotions [43]. “Emotion Trainer”, another computer program designed to help

people with autism learn to recognize and predict emotions in others, was tested in one study [24]. "Let's

Face It!”, an intervention comprised of seven interactive computer games, was evaluated in one study

[25]. Finally, two therapist-led interventions were evaluated: an ER training intervention consisting of four

sessions [40], and an intervention designed to teach emotion understanding [35,36].

The duration of the interventions varied from 8 days [35,36] to 20 weeks [41]. Four studies had fewer than

10 sessions [34-36,40], two studies used a high intensity training with daily treatment cycles 5 days a

week for several weeks [38,42], while the frequency of the intervention in the remaining studies ranged

from 10 [39] to 24 sessions [43]. Length of each session (intervention intensity) ranged from 10-25

minutes [37] to 2 hours per session [34]. None of the reports included information about costs associated

with the treatment.

Most studies used waitlist controls or no intervention as comparator. However, the two “FaceSayTM”

studies [37,39] and one of the studies testing “The Transporters” [44] used comparators designed to be

similar to the intervention but without the active components. One study used teaching of belief

understanding and play as comparators [36] (see Table 1 for more detailed information about the

interventions and the comparators in each included study).

Generalizability: Most interventions are well described, and several are standardized, favoring

implementation. The heterogeneity of the interventions regarding format, duration, and components

suggests that various interventions are feasible, but also make generalization of effects across

interventions uncertain. Each intervention had been tested in only one or a few RCTs. Thus, conclusions

about which specific intervention might be best suited for a specific individual or group of individuals are

not possible at this time.

Table 1 about here

Treatment provider

Information about the trainers was generally limited. The qualifications of the trainers included post-

graduate, graduate, or undergraduate students delivering the intervention, while some studies also used

clinical psychologists, psychiatrists, social workers, pediatricians and speech and language pathologists.

The experience of the trainers varied across studies. In total, five studies reported that providers received

specific training in delivering the intervention [38,39,42-44], while others did not provide such

information. Four studies reported that the trainers were supervised [39,41-43]. Treatment fidelity was

controlled in 10 studies [15,25,34,38-44].

Generalizability: The interventions were delivered by multiple professions with varying clinical

experience. It is likely that the trainer’s qualifications play a subordinate role in more standardized

training programs.

Populations

Narrative synthesis: An ASD diagnosis (AS, autism, or PDD-NOS) was a criterion for eligibility in all

included studies (Table 2). In most studies, the diagnosis was confirmed by standardized diagnostic tools

or rating scales, such as the Autism Diagnostic Observation Schedule (ADOS-2) [45], Autism Diagnostic

Interview–Revised (ADI-R) [46], and Social Responsiveness Scale (SRS) [47,48]. Although the studies

mainly included participants with ASD without intellectual disability, one study included separate

analyses of a subsample with autism and low IQ [37]. None of the studies included information about

psychiatric comorbidity among the included participants.

A minimum IQ or verbal IQ level (IQ > 70 or IQ > 85 respectively on the Wechsler Intelligence Scale for

Children) was an additional criterion for inclusion in six studies [24,34,38,41-43]. In two studies, also a

minimum score on the Comprehensive Assessment of Spoken Language was required for inclusion

[38,42]. In some studies, additional exclusion criteria were listed, such as vision problems [25], good

performance on emotion recognition tasks [40], and serious conduct problems [41].

The sample sizes were small to moderately large, ranging from 18 to 79 individuals. All studies except

two [37,42] provided some information about recruitment procedures. Three studies used public

announcements [34,38,43] whereas the remaining nine studies recruited from academic centers, clinics,

local or special schools and local organizations. Of the 13 studies, seven reported the number of screened

and randomized participants, as well as the number of participants who completed the study [24,37-

40,42,43], while some of this information was omitted in the remaining studies. No information about the

participants’ expectations and preferences was included in any of the studies.

Ten studies included children ranging from 4 to 13 years of age across studies. Two studies focused on

both children and adolescents up to 15 years of age [37,40], and one study focused mainly on adolescents

[24]. Eleven reports provided information about male/female ratio, one of which [41] reported an all-male

sample. Only a minority of females was included in the studies (0 to 23%), and no study presented gender-

specific results. Two studies [24,44] didn’t provide any information about gender ratio (see Table 2 for

detailed information about key variables related to the populations).

Generalizability: Females and individuals outside the typical IQ range (IQ < 70) were poorly represented

in the study populations, and information on comorbidity was missing in the reports. Future studies will

require large, well-characterized, and heterogeneous samples in order to determine potentially moderating

effects of characteristics such as gender, age, comorbidity, symptom severity, and IQ.

Table 2 about here

Context of included studies

Narrative synthesis: Seven studies were conducted in the United States [25,37-39,41-43], three in the UK

[15,24, 35, 36], two in Australia [34,44], and one in Ireland [40] (Table 2). Type of setting was described

briefly in the majority of studies: five studies were conducted at universities [34,38,41-43], three in

schools or after-school facilities [24,37,39], and three reported that the intervention took place in the

participant’s homes [15,25,44]. Three reports did not provide explicit information about setting

[35,36,40].

Incentives for participation were reported in six studies [15,25,48,42-44]. These included monetary

compensation for parents, reinforcers, points that led to rewards, or free copies of intervention DVDs for

children.

Generalizability: The included studies were conducted in a limited number of geographical and cultural

regions. The studies were not conducted in real-world clinical settings. It is possible, however, that

standardized training of basic skills such as ER is less sensitive to cultural differences and specific setting,

compared to more complex psychological interventions.

Outcome

Narrative synthesis: The effects were measured post-treatment, with the exception of two studies that also

included follow-up assessments after 5 weeks [43] and 6 months [35,36], respectively. Measures designed

to capture ER skills and changes in those skills were included in all studies. These measures generally

assessed the ability to recognize emotions from facial expression, voices or postures. Social skills were

measured in seven of the included studies. While questionnaires such as the SRS [47,48] were used in

most studies, also observations of social interaction were included in three studies [37,39,44] (Table 3).

Nine studies evaluated interventions specifically targeting ER (Table 3). These studies used a variety of

outcome measures intended to measure ER, including the affect recognition subtest of the NEPSY-II [49],

the Face Task [50], and photographs from Ekman’s Pictures of Facial Affect [51]. All nine studies

indicated a significant improvement on at least one ER measure, compared to the control group. The effect

sizes were generally large (see Table 3). Four of these studies also included measures of social skills

[47,39,43,44], enabeling examination of the extent to which improvement in ER was accompanied by

improvement in social skills. Three of the studies did not find a significant effect on social skills as

measured with parent ratings or observations [39,43,44]. The fourth study, which evaluated the program

“FaceSayTM”, reported the results separately for participants with low- and high-functioning ASD [37]. A

significant improvement as measured with Social Skills Rating System (SSRS) [52] was reported for the

low IQ<70 group, while blinded observations indicated improved social skills for the average to IQ>70

group. However, it should be noted that there was a substantial pre-treatment difference between the

intervention group and the control group on two of the outcome measures for the participants with high

functioning autism, indicating risk of bias.

The four remaining studies evaluated programs combining ER training with social skills training (Table

3). Three of these studies [38,41,42] measured the effects on ER with the Diagnostic Analysis of

Nonverbal Accuracy 2 (DANVA2) [53], which is designed to assess the ability to identify simple

emotions in faces displayed on a computer. The fourth study [34] used an outcome measure designed to

assess perception of emotions from photos of facial expressions [54] and postures [55]. The standardized

mean differences post-treatment did not indicate a significant effect on ER in any of these studies,

although a separate publication [56] with pooled analyses with data from two of these studies, both of

which evaluated a comprehensive summer program [38,42], suggested that the treatment group had better

encoding accuracy post treatment on one of the tested emotion. Three of the studies also included

measures of social skills. All suggesting a significant effect favouring the intervention, measured with the

Behavior Assessment System for Children, Second Edition (BASC-2) [57] in two studies and the Social

Skills Questionnaire (SSQ) [58] and Emotion Regulation and Social Skills Questionnaire (ERSSQ) [34] in

one study. (See Table 3 for detailed information about the outcome measures and effects.)

Generalizability: The lack of long-term follow-up assessments and sparse use of observations in real-life

situations limits the external validity of the findings. For interventions specifically targeting ER, there

were few indications that the training effects were generalized to daily social interaction. For

interventions combining ER training and social skills training, on the other hand, the results indicated

improvement in social skills. Although the available studies do not permit firm conclusions, these results

might indicate that combining ER training with applied training of skills is required for generalization of

the training effects.

Table 3 about here

Discussion

This review focused on the determinants of external validity in RCTs on ER training in ASD. A variety of

interventions – spanning from brief computerized ER training to comprehensive programs with multiple

components – have been developed and evaluated in RCTs. Each specific ER intervention has so far only

been evaluated in small to moderately large efficacy studies. Most of these studies suggest beneficial

effects. While these studies are important first steps, judgements about generalizability of findings from

these initial RCTs are compromised by samples with restricted coverage of the source population.

Insufficient information about the participants’ characteristics (e.g., psychiatric comorbidity) further

complicates such judgements. Moreover, it is unclear to what extent training effects are transferred to real-

life social interaction. The latter stands out as particularly important, given that people with ASD often

experience difficulty in generalizing across situations.

Meaningful change in the client’s daily life is arguably an essential goal for skills training in general.

When the training is remote from daily activities it seems particularly important to confirm that such a

change is accomplished. The overall pattern of results of the ER studies does not suggest a clear link

between increased ER and improved social skills. In fact, most studies evaluating specific training

programs for ER do not report significantly improved social skills, despite improvement in ER itself. The

only exception was a study evaluating the computerized program “FaceSayTM” [37], in which blinded

post-intervention observations indicated improved social interaction in children with ASD in the typical

IQ range. More rigorous studies with larger samples and longer follow-up time are needed before firm

conclusions can be drawn.

Limited or unknown generalizability of study results is far from unique for ER training. For instance, a

recent review suggests that the accumulated research on social skills group training in children with ASD

suffer from similar shortcomings [31]. Poor external validity is also evident for research on child mental

health treatments in general. A comparison of the characteristics of the participants in 437 RCTs on

evidence-based child mental health treatments and the characteristics of a sample of 1781 youths in the

mental health system suggest that the study participants did not represent the large majority of the clinical

sample, based on attempts to match for type of mental health difficulty, gender, age, ethnicity, and setting

[59]. This might not be surprising, giving the large variety of clinical characteristics and developmental

stages represented in this age group. Consequently, handling uncertainty related to heterogeneity within

the client groups is a major challenge for child and adolescent mental health care.

Clinical implications

There is a great demand for effective interventions for children with ASD, and given the heterogeneity of

ASD it is unlikely that one single format meets the needs and preferences of all children with ASD [60].

The clear structure and presumed (since data are rarely reported) low economic cost of many ER training

programs makes them suitable for implementation. As in the case of internet-delivered cognitive

behavioural therapy [61], the structure of ER training programs favours adherence to the protocol and

makes dissemination more straightforward. This is a major advantage compared to many other

psychological interventions that are often suboptimally delivered in clinical practice due to deviations

from the treatment manual [62]. Nevertheless, the threats to external validity observed here make clinical

decisions challenging. The relatively large number of other interventions that exist for children and

adolescents with ASD, with varying levels of evidence [63], adds further complexity to treatment

decisions. This should not be interpreted as an argument for withholding treatment or allowing all kinds of

treatment regardless of scientific evidence [64]. Rather, it is advised to maximize the involvement of

young patients and their caregivers in treatment decisions in order to facilitate informed choice. Shared

decision making (SDM) could be a viable approach for this purpose, in which the clinician and client

make decisions together using the best available evidence [65]. According to this approach, children and

adolescents with ASD and their parents would be encouraged to communicate their preferences and

expectations, and thereby increase their autonomy and engagement. The level of involvement is flexible

and depends on the client’s preferences. Emerging data indicates that SDM in the treatment of children

with ASD is associated with higher parent satisfaction and improved guidance regarding treatments [66],

although more research is needed to clarify the potential benefits of the approach. It should also be noted

that substantial barriers to SDM have been reported for children with ASD [67] and a particularly low

SMD receipt has been found for this group [68]. Thus, there seems to be plenty of room for improvement

in how treatment decisions are reached.

Future directions

The feasibility and efficacy of various interventions in this domain have now been tested in several initial

studies. It might be time to move on to larger effectiveness studies of both high internal and external

validity, and to comparisons of different ER training systems. In order to inform clinical practice, robust

randomized studies with clinical samples and moderator analyses are needed. In addition, it would be

worthwhile to explore how the interventions can be tailored and optimized for specific sub-groups. For

this purpose, systematically replicated single-subject studies [69] on individuals with varying clinical

(e.g., symptom severity, comorbidity, and IQ) and sociodemographic characteristics (e.g., gender and age)

could be an important complement to randomized studies.

Another major issue that remains to be settled is to what extent treatment gains may be generalized to real-

life situations. For this purpose, development of sensitive and psychometrically sound measures of the

quality of social interaction and its response to training are needed. We suggest that experience sampling

methodology (ESM) might be valuable as a complement to blinded observations. ESM requires that

the participants report their experiences at random points throughout the day,

which is likely to result in higher ecological validity than standardized interviews or questionnaires

that often seek to determine behavior over a longer period (weeks or months). The feasibility of ESM has

recently been demonstrated in children with ASD [70], and it has been reported to be helpful in

identifying the nature and quality of social experiences in this population [71]. However, it is important to

note that this methodology sometimes could require some level of assistance, and may not be a viable

option for all children and adolescents with ASD. Again, single-subject designs could be a valuable

method to explore both how measurement should be adapted to different subgroups and how training

effects are generalized to daily social skills.

We would also like to emphasize the usefulness of systematically collected data from clinical settings.

Large and representative clinical samples of the heterogeneous group of children and adolescents with

ASD have the potential to reveal predictors of treatment response, which in turn eventually can lead to a

better decision basis. This type of data can also be used to catch important signals indicating occurrences

of untoward events during treatment. Available studies give hardly any information about adverse events

or harms, which is a general shortcoming of studies of psychological treatment [72].

Limitations

Some limitations should be noted. First, the present review was restricted to RCTs. It is likely that also

other study designs can give valuable insight into how specific subgroups will respond to the treatment

[73,74]. Single-subject designs in particular have traditionally played an important role in autism research.

ER has, to the best of our knowledge, only been evaluated in a small number of non-randomized studies.

A few single-subject studies have been published [75-77], but must be replicated systematically in order to

contribute to our understanding of generalizability.

Second, our aim was to assess generalizability from the information available from the published reports.

Consequently, we did not contact the authors for missing information, nor did we review all possibly

available literature and information outside of scientific journals. We also did not investigate a number of

factors necessary for successful implementation, including availability and costs of the evaluated

programs, educational material, and training courses.

Finally, we focused exclusively on external validity. While assessment of risk of bias in the individual

studies was outside the scope of this review, we noted that only few studies used blinded assessments of

relevant outcomes. In addition, there was a substantial difference in key variables between the intervention

group and control group pre-treatment in several studies. The latter is presumably an unfortunate

consequence of small samples randomized, further underscoring the need for larger and more rigorous

studies. Proper assessment of risk of bias can be found in a recent systematic review by Fletcher-Watson

and colleagues [27]. Although several of the studies included in the present review were published after

the search for the review by Fletcher-Watson et al. was conducted, we did not find evidence that

methodological quality has improved dramatically in the interim.

Conclusion

Despite an increasing number of promising treatment studies on ER training, the clinical generalizability

is still largely unknown. In clinical practice, this uncertainty should be taken into consideration by

involving children and adolescent with ASD and their caregivers in SDM and carefully monitoring their

progress. High priorities for future research are to increase the knowledge about how improved ER

influences everyday social interaction, and to investigate for which subsets of individuals with ASD this

intervention might be beneficial or less beneficial/ineffective, respectively.

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Appendix 1: Documentation of literature search

Table of ContentsGeneral information......................................................................................................................... 1

Search strategy for Medline (Ovid)............................................................................................... 2

General information

Date: November 2015 (update of the search made in May 2015)

Search specialists: Klas Moberg, University Library, Karolinska Institutet

Total number of hits:

Before deduplication: 4884 After deduplication: 201 (Deduplication has been made against the prior search and among new articles)

Databases used:

Cochrane (Wiley) Medline (Ovid) Embase (Elsevier) Cinahl (Ebsco) Psychinfo (Ovid) Social Services Abstracts (Proquest) ERIC (Ovid)

o

Search strategy for Medline (Ovid)

Date: 2015-11-20

Number of hits: 1327 (1214 in 2015-05-05)

1. exp child development disorders, pervasive/

2. autis$.tw.

3. pervasive developmental disorder$.tw.

4. pdd.tw.

5. (language adj3 delay$).tw.

6. (speech adj3 disorder$).tw.

7. childhood schizophrenia.tw.

8. kanner$.tw.

9. asperger$.tw.

10. or/1-9

11. randomized controlled trial.pt.

12. controlled clinical trial.pt.

13. randomi#ed.ab.

14. placebo$.ab.

15. drug therapy.fs.

16. randomly.ab.

17. trial.ab.

18. groups.ab.

19. or/11-18

20. exp animals/ not humans.sh.

21. 19 not 20

22. 10 and 21

23. exp Nonverbal Communication/

24. exp Emotions/

25. "Recognition (Psychology)"/

26. Speech Perception/

27. Auditory Perception/

28. Social Perception/

29. Pattern Recognition, Visual/

30. Visual Perception/

31. Theory of Mind/

32. Voice/

33. Face/

34. ((Emotion* or face or facial or body or voice or gesture*) adj3 (recognition or expression or processing)).tw.

35. theory of mind.tw.

36. or/23-35

37. 22 and 36

Appendix 2: Excluded publications (with reasons)

Population1. Bölte S, Feineis-Matthews S, Leber S, Dierks T, Hubl D, Poustka F. The development and evaluation of a

computer-based program to test and to teach the recognition of facial affect. International Journal of Circumpolar Health 2002;61 Suppl 2:61-68.

2. Bölte S, Feineis-Matthews S, Poustka F. Brief report: Emotional processing in high-functioning autism--physiological reactivity and affective report. Journal of Autism and Developmental Disorders 2008;38:776-781.

3. Bölte S, Hubl D, Feineis-Matthews S, Prvulovic D, Dierks T, Poustka F. Facial affect recognition training in autism: can we animate the fusiform gyrus? Behavioral Neuroscience 2006;120:211-216.

4. Bölte S, Poustka F. The recognition of facial affect in autistic and schizophrenic subjects and their first-degree relatives. Psychological Medicine 2003;33:907-915.

5. Golan O, Baron-Cohen S. Systemizing empathy: teaching adults with Asperger syndrome or high-functioning autism to recognize complex emotions using interactive multimedia. Developmental Psychopathology 2006;18:591-617.

6. Golan O, Baron-Cohen S, Hill J. The Cambridge Mindreading (CAM) Face-Voice Battery: Testing complex emotion recognition in adults with and without Asperger syndrome. Journal of Autism and Developmental Disorders 2006;36:169-183.

7. Golan O, Baron-Cohen S, Hill JJ, Rutherford MD. The 'Reading the Mind in the Voice' test-revised: a study of complex emotion recognition in adults with and without autism spectrum conditions. Journal of Autism and Developmental Disorders 2007;37:1096-1106.

8. Philip RC, Whalley HC, Stanfield AC, Sprengelmeyer R, Santos IM, Young AW, Atkinson AP, Calder AJ, Johnstone EC, Lawrie SM, Hall J. Deficits in facial, body movement and vocal emotional processing in autism spectrum disorders. Psychological Medicine 2010;40:1919-1929.

Intervention1. Fortin M, Dionne J, Pinho G, Gignac J, Almirall J, Lapointe L. Randomized controlled trials: do they have

external validity for patients with multiple comorbidities? Annals Family Medicine 2006;4:104-108.

2. Tanaka JW, Wolf JM, Klaiman C, Koenig K, Cockburn J, Herlihy L, Brown C, Stahl SS, South M, McPartland JC, Kaiser MD, Schultz RT. The perception and identification of facial emotions in individuals with autism

spectrum disorders using the Let's Face It! Emotion Skills Battery. Journal of Child Psychology and Psychiatry 2012:53:1259-1267.

Design1. Ratcliffe B, Wong M, Dossetor D, Hayes S. The association between social skills and mental health in school-

aged children with autism apectrum disorder, with and without intellectual disability. Journal of Autism and Developmental Disorders 2015;45:2487-2496.

Publication type1. Fletcher-Watson S, McConnell F, Manola E, McConachie H. Interventions based on the Theory of Mind

cognitive model for autism spectrum disorder (ASD). Cochrane Database of Systematic Reviews 2014:CD008785.

2. Rodgers JD, Thomeer ML, Lopata C, Volker MA, Lee GK, McDonald CA, Smith RA, Biscotto AA. RCT of a psychosocial treatment for children with high-functioning asd: Supplemental analyses of treatment effects on facial emotion encoding. Journal of Developmental and Physical Disabilities 2015;27:207-221.

3. Wass SV, Porayska-Pomsta K (2014) The uses of cognitive training technologies in the treatment of autism spectrum disorders. Autism 2014;18:851-871.