Abstract .uk



Title pageEmotion 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 B?lte1,2 and Ulf Jonsson1,2,51Center 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 notificationAll 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 B?lte 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 B?lte, 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, G?vlegatan 22, 11330 Stockholm, Sweden; E-mail: Steve.Berggren@ki.seEmotion recognition training in Autism Spectrum Disorder: a systematic review of challenges related to generalizability AbstractObjective: 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 wordsNeurodevelopmental disorders, autism, emotion, training, technology, intervention, Asperger syndrome, child psychiatryIntroductionAutism 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.MethodsEligibility criteriaTypes 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 parator: 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 rmation sourcesCochrane (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 selectionTwo 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 items1. 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 analysisTwo 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. ResultsStudy selectionThe 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 hereTreatment interventionNarrative 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 hereTreatment providerInformation 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. PopulationsNarrative 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 hereContext of included studiesNarrative 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. OutcomeNarrative 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 hereDiscussionThis 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 implicationsThere 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 directionsThe 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]. LimitationsSome 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.ConclusionDespite 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. References1. 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Journal of Developmental and Physical Disabilities 2015;27:207-221.57. Reynolds CR, Kamphaus RW. Behavior assessment system for children, ed 2nd. Circle Pines, MN, AGS, 2004.58. Spence SH. Social skills questionnaire; Social skills training: Enhancing social competence with children and adolescents: Photocopiable resource book. Windsor, NFER-Nelson, 199559. Chorpita BF, Bernstein A, Daleiden EL. Empirically guided coordination of multiple evidence-based treatments: An illustration of relevance mapping in children's mental health services. Journal of Consulting and Clinical Psychology 2011;79:470-480.60. Van der Paelt S, Warreyn P, Roeyers H. Effect of community interventions on social-communicative abilities of preschoolers with autism spectrum disorder. Developmental Neurorehabilitation 2016;19:162-113.61. Andersson G. The promise and pitfalls of the internet for cognitive behavioral therapy. BMC Medicine 2010;8:82.62. Waller G. Evidence-based treatment and therapist drift. 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Shared decision making in the care of children with developmental and behavioral disorders. Maternal and Child Health Journal 2016;20:665-673.69. Byiers BJ, Reichle J, Symons FJ. Single-subject experimental design for evidence-based practice. American Journal of Speech-Language Pathology. 2012;21:397-414.70. Chen Y-W, Cordier R, Brown N. A preliminary study on the reliability and validity of using experience sampling method in children with autism spectrum disorders. Developmental Neurorehabilitation 2015;18:383-389.71. Cordier R, Brown N, Chen Y-W, Wilkes-Gillan S, Falkmer T. Piloting the use of experience sampling method to investigate the everyday social experiences of children with Asperger’s syndrome/high functioning autism. Developmental Neurorehabilitation 2016;19:103-108.72. Jonsson U, Alaie I, Parling T, Arnberg F. Reporting of harms in randomized controlled trials of psychological interventions for mental and behavioral disorders: A review of current practice. Contemporary Clinical Trials 2014;38:1-8.73. Wells KB: Treatment research at the crossroads. The scientific interface of clinical trials and effectiveness research. American Journal of Psychiatry 1999;156:5-10.74. Benvenuto A, Battan B, Benassi F, Gialloreti LE, Curatolo P. Effectiveness of community-based treatment on clinical outcome in children with autism spectrum disorders: An Italian prospective study. Developmental Neurorehabilitation 2016;19:1-9.75. LaCava P, Rankin A, Mahlios E, Cook K, Simpson R. A single case design evaluation of a software and tutor intervention addressing emotion recognition and social interaction in four boys with ASD. Autism 2010;14:161–178.76. McHugh L, Bobarnac A, Reed P. Brief report: teaching situation-based emotions to children with autistic spectrum disorder. Journal of Autism and Developmental Disorder 2011; 41:1423–1428.77. Russo-Ponsaran NM, Evans-Smith B, Johnson JK, McKown C. A pilot study assessing the feasibility of a facial emotion training paradigm for school-age children with autism spectrum disorders. Journal of Mental Health Research in Intellectual Disabilities 2014;7:169–190.Appendix 1: Documentation of literature searchTable of Contents TOC \o "1-3" \h \z \u General information PAGEREF _Toc470183341 \h 1Search strategy for Medline (Ovid) PAGEREF _Toc470183342 \h 2General informationDate: November 2015 (update of the search made in May 2015)Search specialists: Klas Moberg, University Library, Karolinska InstitutetTotal number of hits:Before deduplication: 4884After 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)Search strategy for Medline (Ovid)Date: 2015-11-20Number 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/1-911. 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/11-1820. exp animals/ not humans.sh.21. 19 not 2022. 10 and 2123. 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/23-3537. 22 and 36Appendix 2: Excluded publications (with reasons)Population1. B?lte 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. B?lte 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. B?lte 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. B?lte 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. ................
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