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Weitlauf AS, McPheeters ML, Peters B, et al. Therapies for Children With Autism Spectrum Disorder: Behavioral Interventions Update [Internet]. Rockville (MD): Agency for Healthcare Research and Quality (US); 2014 Aug. (Comparative Effectiveness Review, No. 137.)

  • This publication is provided for historical reference only and the information may be out of date.

This publication is provided for historical reference only and the information may be out of date.

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Therapies for Children With Autism Spectrum Disorder: Behavioral Interventions Update [Internet].

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Results

Results of Literature Searches and Description of Included Studies

Article Selection

We identified 2,639 citations and abstracts (Figure 2). We excluded 2,012 studies at abstract review and assessed the full text of 627 studies. Among these, 79 publications, comprising 65 unique studies, met our criteria. Eight of these studies report followup data to papers included in the 2011 review of therapies for children with autism spectrum disorder (ASD). The 65 included studies comprise 48 randomized controlled trials (RCTs) and 17 nonrandomized trials or cohort studies. Table 5 outlines study characteristics. Appendix E includes a list of all studies excluded at the abstract and full-text review stages.

This figure illustrates the disposition of studies identified for the review. We identified a total of 2639 citations and abstracts from database searches and other sources. Of these, we excluded 2012 at abstract review and reviewed the full text of 627 studies. Of these 627, we excluded 548 for reasons including lack of relevance to the key questions and ineligible study size or type. We included a total of 79 publications comprising 65 unique studies.

Figure 2

Disposition of studies identified for this review. KQ = Key Question; n = number a Numbers do not tally as studies could be excluded for multiple reasons.

Table 5. Overview of included studies.

Table 5

Overview of included studies.

KQ1. Effects of Behavioral Interventions on Core and Commonly Associated Symptoms in Children With ASD

A wide range of interventions can be classified as behavioral. For the 2011 review and this update, we included studies of early intensive behavioral and developmental interventions, which comprised University of California, Los Angeles (UCLA)/Lovaas-based approaches, the Early Start Denver Model (ESDM), and parent training approaches incorporating principles of Applied Behavior Analysis (ABA) to improve outcomes among young children with ASD; social skills interventions; focal play-based /interaction-based interventions; behavioral interventions focused on commonly associated behaviors; and a small group of other behavioral interventions assessing other interventions in core/associated areas (e.g., sleep workshops).

Early Intensive Behavioral and Developmental Interventions

Key Points

  • Of the 25 new studies addressing early intensive behavioral and developmental interventions, eight were good, 13 were fair, and four were poor quality.
  • Many studies used parent-report measures of adaptive and ASD symptom-related outcomes, which may be confounded by parental stress, parental involvement in treatment protocols, and nonrandom assignment based upon parental treatment preferences.
  • All studies of ABA-based interventions compared a minimum of two treatment groups. No study included a control group that was not receiving some type of intervention (including school enrollment or eclectic community-based therapies, such as medication or occupational therapy), although some limited the number of behaviorally based treatment hours that control participants could receive.
  • Studies with parent training components reported improvements in language with inconsistent results for other outcomes.
  • No studies reported harms related to children.

Overview of the Literature

In the 2011 review, we identified 17 comparative studies57-75 (described in 19 papers), of which six were RCTs (two good quality,73, 75four fair57, 69, 71, 72), five were nonrandomized trials (four fair quality,64-68, 74 one poor70), four were prospective cohort studies (three fair60, 61, 63 and one poor quality62), and two were poor quality retrospective cohort studies.58, 59 For the current review we identified 25 comparative studies (reported in 37 publications) meeting our inclusion criteria and evaluating either ABA-based early intervention approaches73, 76-91 or approaches integrating parent training components).72, 92-109 Four of these studies (published in multiple papers) report followup data for studies reported in the 2011 review.72, 73, 79, 80, 85-90, 105, 110 Additionally, one study in the current report95 may include some participants reported in studies in the 2011 review.111, 112

ABA-based approaches. Ten studies (reported in 18 publications) assessed ABA-based early intensive behavioral and developmental intervention (Table 6).73, 76-88, 90, 91, 110, 113 Studies included two RCTs conducted in the United States;73, 84, 85 two non-randomized controlled trials conducted in Europe;76,91 three European,77-80 one U.S.-based,83 and one Israeli81 prospective cohort study; and one Canadian retrospective cohort study that reported on segments of the same population in multiple publications.86-90, 110 Five studies compared ABA-based approaches to care-as-usual community therapies73, 78, 79, 81, 85, 86 and five to preschool-based programs.76, 77, 83, 84, 91 Mean participant age ranged from 15–72 months. Treatment duration ranged from 6 to 24 months. We rated two studies as good, seven studies as fair, and one study as poor quality.

Table 6. Key outcomes of ABA-based early intervention studies.

Table 6

Key outcomes of ABA-based early intervention studies.

Parent training. We identified 15 studies (reported in 19 publications) of early intervention with parent training components (Table 7).72, 92-109 Studies included five European95, 97, 101, 102, 108 and one Australian100 prospective cohort studies; four RCTs conducted in the United States or Canada,93, 98, 103, 104, 107, 109 two (including one crossover) in Asia,92, 99 one in Australia,96 and two (one with suboptimal randomization) in Europe.72, 94, 105 Seven studies compared parent training to treatment as usual (community-based intervention).72, 92-,94, 98, 101, 102, 105- 107 Five compared ABA-based parent training to other parent-training paradigms97, 100, 103, 104, 108, 109 or multiple other interventions,95, 96 and the comparison arm in one study received no specific intervention.99 Mean participant age ranged from 14 to 81 months. Treatment duration ranged from 12 weeks to 2 years. We rated six studies as good, six studies as fair, and three studies as poor quality.

Table 7. Key outcomes of early intervention studies with parent training components.

Table 7

Key outcomes of early intervention studies with parent training components.

Detailed Analysis

ABA-Based Approaches

One fair quality RCT examined the use of the Learning Experiences and Alternative Program for Preschoolers and Their Parents (LEAP) protocol in preschool classrooms in the United States.84 The study compared 27 classrooms (n children=177; mean age: 50.1 months±4.6 months) with teachers trained in the full LEAP curriculum (including peer mediated social skills, incidental teaching, pivotal response training, the Picture Exchange Communication System (PECS), and positive behavior support) to 23 classrooms (n children=117; mean age: 50.7 months±4.2 months) where teachers received the LEAP manual but no formal training. Both groups received an average of 17 hours per week of intervention over two years. Relative to the manuals-only group, children in the full LEAP training classrooms showed significant (p< .05) improvement on investigator-rated Childhood Autism Rating Scale (CARS) scores, language, cognitive, and social skills measures. The students of teachers rated as having better intervention fidelity showed better outcomes on all measures.

Five additional studies (reported in multiple publications) examined the use of school-based ABA programs (one fair quality nonrandomized controlled trial and four fair quality prospective cohort studies. 76-77, 78, 81-83 All five compared standard special education preschool curriculums to special education preschools with some sort of enhanced intervention modality, including general ABA,81, 82 individual UCLA/Lovaas-based behavioral intervention,76, 78 Treatment and Education of Autistic and Communication related handicapped Children (TEACCH)- or LEAP- programs,83 and a mix of behaviorally-based operant conditioning techniques.77 Mean treatment intensity ranged from 13.8–28.38 hours per week, with length of enrollment varying from 8–24 months. Mean child ages ranged from 25.1–53.5 months.

The effects of enhanced school-based interventions relative to standard special education preschool curricula were mixed. Some studies76-78 found that the enhanced intervention groups showed greater gains in cognitive outcomes and parent-reported adaptive skills. Other studies found that children in all groups improved on cognitive, adaptive, and ASD symptom measures76, 81-83 regardless of intervention type, although in some cases treatment groups showed greater improvements.76 Others found declines in both groups on standardized scores of motor skills.81, 82 Intervention efficacy was associated with baseline cognitive scores in one study of TEACCH classrooms,83 with lower baseline cognitive scores associated with more improvement. Lower baseline ASD severity was associated with parent-reported cognitive and adaptive growth for children who received eclectic vs. ABA intervention, but not with standardized test scores.81, 82 Additional UCLA/Lovaas-style intervention over-and-above classroom involvement was associated with reduced ASD symptoms as rated by clinicians on the CARS78 but not as rated by parents using the Scale of Pervasive Developmental Disorder in Mentally Retarded Persons.76 Where examined, total hours of intervention per week were not associated with cognitive or adaptive outcomes, although hours were similar across intervention groups within each study (e.g., comparing half-day programs to other half-day programs).

Four studies (reported in multiple publications) compared ABA-based early intervention to eclectic treatment as usual.73, 79, 80, 85-91, 110 One good quality RCT compared ESDM to community-based interventions.73, 85 It randomized children into two groups based on gender and IQ. For two years, 24 children in the ESDM arm (mean age: 23.9±4.0 months at study entry, mean IQ: 61.0±9.2) received 1:1 therapist-delivered manualized intervention (mean of 15.2±1.4 hours/week) as well as parent-delivered treatment (mean 16.3±6.2 hours/week). The comparison group of 21 children (mean age: 23.1±3.9 months, mean IQ: 59.4±8.6) received individual (mean 9.1 hours/week) and group (mean 9.3 hours/week) therapies, including speech-language therapy, occupational therapy, and developmental preschool enrollment. The ESDM intervention targets social communication and engagement as well as general child development. After one year of treatment, The ESDM group showed significantly greater improvement in IQ but not adaptive behavior. After two years of treatment, the ESDM group continued to show significantly more IQ improvement as well as receptive and expressive language. Both groups improved in all domains of adaptive behavior but socialization, with greater improvements in the ESDM group. Neither group showed significant differences in Autism Diagnostic Observation Schedule (ADOS) severity scores or repetitive behavior, although the ESDM group demonstrated a diagnostic shift toward a milder diagnosis (PDD-NOS) at followup. Electroencephalography (EEG) measures of engagement and cognitive processing for children in the ESDM group with usable data were comparable to typically developing children.

A good quality non-randomized trial from Europe91 compared children (mean age=62.52±16.96 months) with ASD and co-occurring intellectual disability receiving school-based ABA therapy (n=20) to a matched control group of children receiving care-as-usual (e.g., enrollment in TEACCH classrooms, PECS; n=20). The intervention group received one-to-one treatment (mean=4.98 hours, SD=1.45; range: 1.32–7.11) from master's level interventionists. Eleven participants received 2 years of treatment and 9 participants received only one year due to funding loss and school noncompliance. Independent ratings indicated high treatment fidelity (90.3%). Monthly meetings between therapists and parents and teachers provided them with strategies on skill instruction and maintenance.

Cognitive, adaptive, and language skills, and ASD symptoms, were assessed at 12- and 24-months after starting treatment. Both the treatment and control groups showed significant improvement from baseline to 12-month and from 12-month to 24-month followups in developmental age, adaptive skills, and receptive language, with the treatment group showing significantly more improvement than the control (p values<.04, effect sizes ranging from 1.09–2.61). The treatment group showed significant improvements in IQ (p<.001, effect size=.40) between baseline and the 12-month assessment but not the 24-month assessment, whereas the control group did not show significant IQ gains at either time point. Fifty-five percent of treatment group participants showed reduced levels of intellectual disability post-treatment versus 5 percent in the control group. The treatment group (but not the control group) also had reduced ASD symptoms as measured by the CARS and ADOS (p values<.01, effect sizes 1.50–1.51). Neither group significantly improved in expressive language. Multiple baseline child factors were significantly correlated with progress over time, including developmental age, hours of treatment per week, IQ, adaptive and play skills, and receptive language.

A fair quality Canadian retrospective cohort study86-90, 110 matched children receiving a large-scale, publicly funded, community-based early intensive intervention program that incorporated ABA, discrete trial training, and naturalistic approaches (n=61, mean age=42.93±11.53 months) to waitlisted children receiving care-as-usual (n=61, mean age=42.79±10.51 months). The intervention group received treatment (mean 25.81±3.44 hours/week) conducted by trained instructor therapists in specialized centers, preschools, and the home environment. The control group received a mean of 17.9±12.3 hours/week of school-based services and <10 hours/week of behavioral intervention conducted by community-based interventionists in community settings. The approaches included low-intensity ABA, speech and occupational therapy, and behavioral consultation. Children in the treatment arm were enrolled in treatment longer (mean=27.84±8.11 months) than children in the waitlist group (mean=17.01±2.81months), and analyses controlled for this difference.

ASD severity improved for the treatment group compared with control, as did Vineland composite standard and ratio scores and IQ estimates (p values≤.033, effect sizes ranging from 0.53 to 0.83). Although treatment group participants had cognitive scores an average of 19 points higher than controls at followup, this should be interpreted with caution due to a lack of baseline cognitive data. Outcomes were related to age at enrollment, treatment duration, and higher baseline adaptive scores, with duration becoming nonsignificant after accounting for group membership (correlation of duration, group=.57, p<.01). A significant interaction emerged between age at enrollment and group membership, with younger starting age influencing outcomes for the treatment group but not control. Analyses including participants in the cohort study and additional participants found that younger age at intake, higher initial developmental levels90 and treatment intensity88, 90 were related to treatment outcomes.

Additional analyses of some children in these earlier Canadian studies (overlap not clear) assessed the effects of baseline age and IQ on cognitive and adaptive outcomes in 207 children, and, in a separate analysis of matched older and younger children, effects of baseline age on the same outcomes.110 In the initial retrospective analysis of 207 children, participant ages at intake ranged from 2 to 14.5 years, IQ from 10 to 104, and mental age from 3 months to roughly 7.5 years. Higher baseline IQ and younger age were significantly associated with greater cognitive rate of change (pre-post change in mental age/time in intervention) and with higher IQ at followup (all p<.001), but change in IQ was not significantly associated with higher initial IQ. Higher baseline IQ was also associated with higher adaptive behavior scores at followup (p<.001), but age was not a significant predictor. Longer duration of intervention was associated with slower rate of IQ and adaptive behavior development (p values ≤.01); however, as this analysis was not prospective, the children who received more intervention could have been making slower progress. In the analysis of older (n=60, age 6–13.58 years at baseline) and younger (n=60, age 2.08–5.92 at baseline) children matched on developmental trajectory (i.e., number of intervention hours, baseline IQ and adaptive behavior), younger children had significantly better followup IQ outcomes compared with the older group. Younger children gained an average of roughly 17 IQ points (effect size=0.80) while older children gained an average of 2 points. Cognitive rate improved significantly for younger (effect size=3.19) but not older children. Both groups improved over time in adaptive behavior, but differences between groups were not significant (improvement of 4 points in younger children and 5 in older).

Finally, a poor quality UK study79, 80 compared the long-term effects (2 years post-treatment) of 1:1 home-based early intervention (both university-provided and privately-provided) to community-based treatment-as-usual, including PECS, TEACCH, and medication. The early intervention group included 23 children (mean age=35.7±4 months; mean IQ=61.43±16.43 months), and the community-based group included 18 (mean age=38.4±4.4 months; mean IQ=62.33±16.64) at the two-year followup, with children in the community-based group significantly older at the start of treatment (p< .05). For 24 months, children in the early intervention group received an average of 25.6 hours/week of ABA-based intervention using discrete trial training in the home environment, whereas children in the community-based arm received an unspecified amount of eclectic treatment. After 24 months of intervention, IQ, mental age, and language comprehension/expression improved significantly for the ABA group versus community-based (p ≤.05; effect size for IQ change=0.77). At the two year followup, IQ gains were only maintained for children who received privately-provided ABA-based intervention. IQ remained stable for children in the community-based group and significantly declined for children who received university-provided intervention (effect size=.49). This result is confounded by nonrandom assignment and the fact that at baseline, the university-based group had higher levels of ASD symptoms, lower levels of adaptive behavior, and fewer total intervention hours.

Parent Training Approaches

One good quality RCT examining parent training96 was conducted in Australia and compared two variations of the Building Blocks® program—home or center-based-- to waitlisted controls. The program targeted social and communication skill development. Mean child ages at enrollment ranged from 41.5 to 43.7 months. Mean IQs ranged from 57–66. Treatment duration was 40 weeks. Not all enrolled children had autism spectrum diagnoses; the breakdown was 100 percent of the home-based group, 82.8 percent of the center-based group, and 78.6 percent of the control group. To be enrolled in the center-based group, children had to have a baseline level of “social maturity,” a lack of “high levels” of problem behavior, and parents willing to attend sessions. The home-based group (n at followup=27) received individualized 2-hour visits every 2 weeks in the home environment. Center-based children (n=29) received weekly manualized, 2-hour, center-based sessions in small groups of 4 to 6 children, as well as parent training and a parent support group. The control group (n=28) comprised a non-randomized treatment comparison waitlist. All groups received concomitant additional interventions classified as educational (home-based: 2.37 interventions±1.28; center-based: 2.41±1.50; control: 3.11±1.64) or ASD-specific (home-based: .22±.42; center-based: .14±.35; control: .54±.79). Providers were multidisciplinary teams of teachers, speech-language pathologists, occupational therapists and psychologists.

Children in all three groups showed significant improvements in Vineland Communication scores. Compared with the home-based group, children receiving center-based intervention had significantly greater improvement in language comprehension and expression as measured by the Reynell Developmental Language Scales. Waitlisted children had significantly greater improvements in followup Vineland Socialization scores than children in either treatment group. No other significant differences emerged among the three groups on other child outcomes. When analyses were limited only to children with autism spectrum diagnoses, the magnitude of the effects increased, but the presence of statistical significance did not change.96

Another good quality RCT from the United States 109 compared the language development of two groups of children with ASD diagnoses, one whose parents received training in a component of ABA, Pivotal Response Training (n=20, mean age=29.5 months, SD=6.9), and another whose parents received training in PECS (n=19, mean age=28.9, SD=4.2). Exclusion criteria included having more than nine intelligible words and having primary diagnoses of intellectual disability, neurological pathology or major sensory impairment. Participants were matched on word use, age, and cognitive functioning. Over the course of 23 weeks, parents completed weekly or biweekly 2-hour parent training sessions from doctoral students. Participants received one-on-one treatment in the home (mean=247 hours, range=181–263) from undergraduate student therapists. Therapist and parent-educator fidelity was maintained at 80 percent. Participants continued to receive outside interventions (e.g., speech therapy) and this was monitored via weekly parent report, with no significant between-group differences emerging.

Outcome variables were assessed at intake, immediately post-treatment, and three months post-treatment. Not all post-treatment coders were blind to participant condition, but no differences were found across blinded vs. not blinded sites. Data were not available on all participants at followup (n=38 for Mullen Scales of Early Learning, 35 for MacArthur Communicative Development Inventories, 35 for Vineland). No differences emerged between Pivotal Response Training and PECS groups. Both improved similarly on all variables over time, with effect sizes across collapsed groups of .216 for expressive communication (Mullen Scales of Early Learning; p<.001), .486 for words produced (MacArthur Communicative Development Inventories; p<.001), and .110 for adaptive communication skills (Vineland; p=.037). The authors reported significant variability in participants' responses to treatment. Parent satisfaction post-treatment was similar across groups, with the only significant difference being the parent-reported difficulty of PECS (p=.005).

Two prospective cohort studies also received good quality ratings. The first was conducted in Australia100 and compared professional-led parent training (n=17; mean child age, 36.38 months±7.54; 88.2% male) to a self-directed video-based parent intervention (n=22; mean=35.71 months±6.92; 72.7% male). Nearly 80 percent (77%) of participants were diagnosed with autism and the rest with an ASD. Mean IQ was 53.06±9.06 for the professionally led group and 52.86±6.53 for the video-based group. Exclusion criteria included being enrolled in early intervention, passing the Modified Checklist for Autism in Toddlers (M-CHAT), or receiving more than 20 hours/week of services. No information was provided about manualization.

In the professionally led group, parents attended a two-day group workshop and completed a series of 10 hour-long home visits, which occurred two times a week for 5 to 6 weeks. These visits focused on parental stress and child communication. In the video group, parents received an interactive instructional DVD called “Being Responsive: You and Your Child with Autism.” They independently completed video lessons and accompanying worksheets. Followup assessments were conducted 3 months after treatment finished. All outcomes were based on parent report. Children in the professionally led arm showed significantly greater improvement in social communication than children in the video-based arm, regardless of baseline scores. Parents in the professionally led group also reported reduced child-related stress relative to parents in the video group, with fathers reporting more stress than mothers in both groups. Parents in the professionally led group with low baseline self-efficacy reported higher followup self-efficacy levels than parents in the video arm.100

The second good quality prospective cohort study was conducted in Italy and reported in two papers.101, 102 It compared staff- and parent-led ABA-based intervention ( n=24, 92% male; mean age=55.67±17.63 months) to eclectic community-based therapy (n=20, 95% male; mean age=41.94±13.07 months). Group assignments were not random and were based upon parental preference. Children were excluded based on the presence of major medical issues. In the parent training group, children alternated between one week (average of 25 hours) of therapist-led center-based intervention (discrete trial training, incidental teaching, natural environment teaching) and 3 weeks (average of 14 hours/week) of parent-led home intervention. Treatment focused on individual skills, problem behaviors, and facilitated play and social interactions. In the eclectic group, children received in-home developmental and cognitive behavioral treatments (approximately 12 hours/week) with minimal parent involvement. Treatment goals were based upon staff expertise and preferences.

Compared with the eclectic group, children in the parent training arm showed a significant decrease in ASD symptom severity and increases in language production and mental development. The parents of children in the eclectic group reported that their children showed improved socialization and motor skills, but this was not confirmed by behavioral observation. In the parent training group, older children achieved better adaptive behavior outcomes; younger children made more gains in early language comprehension and production. Children who gained more language comprehension had higher adaptive behavior scores pre-treatment. Pre-treatment language comprehension predicted post-treatment language production. In the eclectic group, higher pre-treatment mental development state and early language skills predicted better outcome on parent-reported adaptive behaviors. Initial higher adaptive behaviors predicted better post-treatment early language comprehension. In both groups, child outcomes on early language skills, mental developmental state, and adaptive behaviors were significantly influenced by self-reported parental stress, children's ability to respond correctly to prompts, the number and difficulty of treatment targets, and children's problem behaviors in sessions. Children who were perceived by their parents as more difficult had less improvement in ASD severity.101, 102

Two studies compared interventions focused on increasing parental responsivity. A good quality RCT from Europe (reported in two papers)72, 105 compared treatment-as-usual + a manualized, communication-focused parent training (n=14, median age 48 months) to treatment-as-usual alone (n=14, median age=51 months) over 12 months. The intervention focused exclusively on parents and targeted increased parental response to child communication. The additional targeted treatment consisted of a recommended 30 minutes/day of parent-led intervention. Parents received monthly training for 6 months followed by training every 2 months for another 6 months. The intensity of treatment as usual alone was not reported but approaches consisted of speech pathology and ABA-based community treatments. The additional treatment group showed improvements in ASD symptoms, expressive language, and number of communicative acts during interactions with parents. Parents in the additional treatment group showed increased responsiveness to their children during videotaped interactions, which was correlated with reduced ASD symptom severity. No between-group differences were found in adaptive behavior or parenting stress. Greater language gains were seen in children who were younger with lower functioning levels at baseline.

A second fair quality RCT conducted in the United States also focused on enhancing parental responsivity and child communication.93 It compared Hanen's More Than Words intervention to treatment-as-usual. The More Than Words group ( n=29, mean age=21.11±2.71 months) received eight manualized group sessions with parents only and three in-home individualized parent-child sessions over a span of 3.5 months, whereas the control group ( n=26, mean age=21.61±2.82 months) received no treatment or treatment as usual. There was no treatment effect on parental responsivity. The More Than Words group showed differential effects on child communication depending on children's baseline object interest; children with lower levels of baseline object interest had greater growth in communication skills, whereas children with higher levels of object interest showed attenuated growth.

A good quality RCT conducted in the United States compared the manualized Assessment Evaluation and Programming System for Infants (AEPS) with and without additional joint attention and social interaction opportunities.103, 104 Both the AEPS group (n=24, mean age=28.6±2.6 months; mean intervention hours=205.66±18.63) and the control (n=24; mean age=28.8±2.8 months; mean intervention hours=196±21) received identical amounts of classroom-based treatment (10 hours/week), home-based parent training (1.5 hours/month), parent education (38 hours), and intervention methods. However, AEPS children received extra training in “interpersonal synchrony,” targeting the three outcome variables of socially engaged imitation, initiation of joint attention, and shared positive affect. No significant (p<.05) differences emerged post-treatment on variables of interest. At the 6-month followup, the AEPS treatment group engaged in significantly more socially engaged interaction than controls (p<.05), with most of the growth in this skill occurring during the treatment period (p<.05) but not during followup (p=.24). No between-group differences were found for initiations of joint attention, shared positive affect, expressive language, or nonverbal problem solving. The AEPS group showed significant growth over time for all variables (p values<.01), but the control group only showed significant growth for expressive language (p=.01). Combined group analyses including 34 children from both the AEPS and control groups examined long-term outcomes an average of 37.6 months after the end of treatment (mean participant age=72.6±months). In this sample, cognitive skills and Vineland-II communication standard scores increased significantly from baseline (mean change 21.4±22.9, effect size=1.02, p<.001 and 12.7±19.4, effect size=0.81, p<.001, respectively), but there was no significant change in ASD symptom severity based on the ADOS.

A fair quality prospective cohort study95 compared outcomes for four different types of intervention after 9 months of treatment: 1:1 home-based, manualized ABA (n=14, mean age=39±6.9 months); special education classroom enrollment (n=21, mean age=41.5±4.0 months); comparatively low-intensity, home-based, manualized behavioral intervention (“portage;” n=18, mean age 39.5±6.3 months), and 1:1 behavioral intervention (“local authority”) that included an intensive introductory 5-day parent training component (n=13, mean age=40.2±6.3 months). The home-based ABA group received an average of 30.4 hours/week of intervention, 28.3 of which were 1:1. The special education group received an average of 12.7 hours/week (3.1 hours 1:1). The portage group received an average of 8.5 hours/week (6.5 1:1), and the local authority group received an average of 12.6 hours/week (12.2 1:1). Participants were not receiving any other teaching interventions during the study.

Post-treatment, mean cognitive and adaptive scores were not significantly different across groups. Children in the home-based ABA group showed significant improvements in educational outcomes as measured by the British Abilities Scale relative to other groups (p<.05). The authors created composite scores based on cognitive, adaptive, and educational functioning, but between-group comparisons only approached significance (p<.06). Baseline ASD severity and total intervention hours did modify effects of treatment significantly. First, baseline ASD severity was inversely related to composite change scores for all but the home-based ABA group and was positively related that group. That is, children with more severe ASD symptoms made more progress in ABA and less in the other intervention groups. Second, more intervention time was negatively related to composite change scores for children in ABA but not in the other groups. More hours of ABA were associated with less progress relative to school enrollment or other home-based interventions.95

One fair quality U.S. RCT (reported in two papers) compared parent-delivered ESDM to community-based treatment-as-usual.98, 106 The ESDM group included 49 children (mean age 21.02±3.51 months, mean developmental quotient [DQ]=64.88±17.22); their parents completed 12 1-hour sessions that included manualized parent-training and coaching. Both the ESDM group and the community group (N=49; mean age=20.94±3.42 months, mean DQ=63.08±15.93) continued receiving community-based treatment-as-usual services as well, including the Developmental, Individual Difference, Relationship-based (DIR) model, TEACCH, ABA, and occupational and speech therapies (range of hours: 0–15.9), with the community group receiving significantly more intervention hours at the second time point (mean 3.68 vs. 1.48; p<.05). Compared with the ESDM group, children in the community-based arm had more severe social affect deficits, poorer imitation skills, and higher nonsocial orienting scores at baseline (p<.05). After treatment, both groups showed improvement in DQ and ADOS Social Affect scores with no main effects of group assignment. Both groups of parents showed significant increases in parent-child interaction behaviors, with greater increases in the ESDM group (effect size=.57) than the community-based group (.37). ). Parents in the ESDM group reported significantly less parenting stress at followup (p=.04) but did not report more parenting competence. When examining combined groups, two key effects emerged. First, total intervention hours were associated with reduced restrictive and repetitive behavior and nonsocial orienting and improved DQ and vocabulary comprehension. Second, children younger than 24 months showed greater increases in DQ scores (effect size=-1.20, p=.002).

A fair quality RCT from Canada compared a DIR-based model, MEHRIT (Milton and Ethel Harris Research Initiative Treatment Program), (n=25; mean age=42.52 months, SD=8.76) to community care-as-usual (n=26; mean age=46.38 months, SD=8.29).107 Data were collected 12 months into an ongoing 24-month treatment course. MEHRIT was administered by trained occupational therapists and speech-language pathologists who worked with participants' parents for two hours per week. Community intervention included no more than 15 hours per week of ABA, speech and occupational therapy, social skills groups, and alternative treatments (mean intervention hours: 3.9 per week). Post-treatment, the MEHRIT group showed significantly more initiation of joint attention (p<.001), involvement in activities (p<.01), and attention to activities (p<.05). They also showed more enjoyment in interaction, but this group difference was also present at baseline (p<.05). Both groups showed significantly improved language skills adjusted for developmental quotients, with no significant between-groups effects (effect sizes of .451 for MEHRIT and .915 for community treatment).

Another fair quality RCT from Asia examined DIR/Floortime (n=15) compared with center-based ABA (n=16).92 Groups were stratified based on age (24–47 months, 28–72 months) and ASD severity, based upon CARS scores. Both groups continued to receive treatment-as-usual, including enrollment in preschool programs and community-based services (such as speech or behavioral therapies.) Relative to the center-based group, the DIR/Floortime group showed significant improvement on the Functional Emotional Assessment Scale (p<.05) and ASD symptoms as rated by the CARS (2.9 vs. .08, p<.01). Parents in the DIR/Floortime group also rated their children as showing significant improvements in emotional development (p<.01). A fourth fair quality RCT comparing parent training plus special education preschool to special education preschool alone reported no between-group differences on language development after 12 months of intervention, though language skills within both groups improved over time.94

Three poor quality studies, two European prospective cohort studies 97, 108 and a crossover RCT from China,99 compared parent training to lower intensity supportive interventions. Mean ages ranged from 25.33–33.6 months. Both involved home visits and working with children and parents. A lower intensity treatment model, Autism-1–2–3, compared two groups that received the same series of 10 half-hour child- and parent-training sessions, with one group having a lagged start date and serving as a control. It did not yield group differences on ASD symptoms, language skills, or parent stress scores.99 Another lower intensity model, the Barnet Early Autism Model (BEAM), incorporated aspects of ABA, TEACCH, PECS, and other occupational and speech-language interventions. It provided an average of 6.4 hours of home-based intervention per week over ten months. Participants were compared with a care-as-usual group and were not randomly assigned. Relative to the control group, BEAM recipients improved significantly more in adaptive behavior (p<.001) and receptive language (p<.05) but not IQ, with baseline levels of parenting stress negatively related to language and adaptive outcomes. 108 The Keyhole model incorporated elements of Hanen's More Than Words and the TEACCH programs. It compared 15 to 18 home visits over a 9 month period (n=35) targeting adaptive skills, ASD symptoms, and parent stress to a lower-intensity intervention model (n=26; 5 home visits, no additional services or supports). Compared with the lower-intensity group, children in the Keyhole intervention showed improved adaptive, imitation, and communication skills, based upon parent report. Mothers in in the Keyhole group also reported improved health but not stress.97

Social Skills Interventions

Key Points

  • Thirteen behavioral studies examined different social skill interventions and included children and adolescents with ASD. Overall, the quality of the studies improved in comparison to the 2011 review. Two studies were rated as good quality, while 10 studies were fair quality, and one was poor.
  • Most studies included school-aged children, without concomitant intellectual disability or language deficits. Most children had average cognitive skills (IQ>70).
  • Most studies reported short-term gains in social skills and emotion recognition as reported by parents or within study measures. Maintenance and generalization of skills beyond the treatment context was addressed within the majority of the studies, but with variable results.
  • The diversity of the intervention protocols and assessments utilized to measure outcomes continues to be a limiting factor for determining effectiveness of social skills interventions.

Overview of the Literature

In addition to the nine comparative studies assessing social skills included in the 2011 review, eight RCTs of fair114-116 and poor117-121 quality and one poor quality retrospective cohort122), 13 studies of good,123, 124 fair,117, 125-134and poor135 quality addressed interventions targeting social skills. Followup data for one study reported in the original review is included in this update.117, 134 Studies addressed in the current review included a total of roughly 462 participants (mean/study=36). Seven RCTs were conducted in the United States,117, 124-127, 129, 134, 136 one in Europe,137 one in Japan,135 and two in Australia.123, 132 Two nonrandomized studies were also conducted in Australia.131, 133 Participant ages across studies ranged from 4 to 13 years, and participants typically had high functioning ASD (IQ>70). Studies assessed group-based approaches including replications of studies evaluating the Skillstreaming model;126, 127, 129, 136 the Children's Friendship Training model; 117, 134 a Japanese pilot RCT of the Treatment and Education of Autistic and Related Communication Handicapped Children (TEACCH) model;135 incorporated peer-mediated components;124, 125, 138 and targeted emotion recognition in children with ASD.123, 128, 132, 133

Detailed Analysis

Four fair quality RCTs conducted in the United States addressed group-based social skills approaches.126, 127, 129, 136 Among these, three studies evaluated the manualized Skillstreaming model (Table 8).127, 129, 136 The studies included between 13 and 52 total participants, all of whom were considered to be high-functioning, and most of whom were male. One RCT compared a manualized performance-based approach, Sociodramatic Affective Relational Intervention, versus the knowledge-based Skillstreaming social skills intervention, which emphasizes social skills, face-emotion recognition, interest expansion, and interpretation of non-literal language. The study included 13 boys with ASD between the ages of 9 and 12. Weekly 90-minute sessions treatment sessions were held over 4 weeks after school. Treatment sessions, regardless of the intervention, included content covering considering others, emotions, consolidating, and generalization of skills. Participants in both groups increased in reciprocal friendship nominations (p=.027) and staff-reported social skills (effect size=0.59, p=.002). Participants in the sociodramatic group interacted more with each and rated one another more favorably after one session, which slightly decreased over time (effect size=0.70, p=.001). Skillstreaming participants also demonstrated gains in interactions and more favorable ratings over the course of the intervention, but at a slower rate as compared with the sociodramatic group (effect size=0.37, p=.001). No significant differences in parent report of social functioning were demonstrated for either group.129

Table 8. Summary of outcomes of social skills studies.

Table 8

Summary of outcomes of social skills studies.

A second RCT127 examined the short-term outcome of a 5-week trial of the Skillstreaming approach and replicates the intervention reported in a study115 described in our 2011 review. The study included 36 children (mean age=9.47), primarily male (94% of the total sample) with high functioning ASD (mean IQ=103) randomized either to Skillstreaming or a wait-list control group. Participants in the treatment group showed significant improvements in most parent-rated measures of social skills compared with the control group (Social Responsiveness Scale: effect size=0.625, p=.003; Adapted Skillstreaming Checklist: effect size=0.584, p=.006; Behavioral Assessment System for Children (BASC)-Withdrawal scale: effect size=1.055, p<.001); however, group differences on the BASC-Social Skills scale were not significant. Staff-report measures found similar outcomes, with significant improvements in ASD symptomology and program-targeted social skills, as well as a decrease in withdrawn behaviors in the treatment group compared with the control arm (effect sizes ranging from 0.69 to 1.4, p values ≤.007). Child-rated measures similarly improved in the Skillstreaming group compared with control (Skillstreaming Knowledge Assessment: effect size=1.272, p<.001; understanding of idioms: effect size=0.390, p<.001).127

Another RCT replicating the Skillstreaming model reported by Lopata et al.127 included 35 children with high functioning ASD between the ages of 7 and 12.136 Skillstreaming involved five 70-minute sessions treatment sessions per weekday over 5 weeks. Treatment sessions involved skill instruction (nonliteral language and face-emotion recognition) and practice as well as a behavioral system to encourage participation and decrease problem behaviors. Weekly 90-minute parent trainings were also conducted, which involved education on ASD as well as training on the treatment program. Scores on the parent-rated Skillstreaming Checklist, Social Responsiveness Scale, and Behavior Assessment System for Children-2 Withdrawal scales improved for the Skillstreaming group compared with the control (effect sizes 0.85, 0.67, 0.70 respectively, all p<.01). Child-rated measures also improved for the treatment group compared with control (Skillstreaming Knowledge Assessment effect size 1.15; language assessment=0.34, p<.001). No group differences were found in face-emotion recognition. Maintenance of effects on the Skillstreaming Knowledge Assessment and BASC Social Skills scale for the treatment group was demonstrated 2 to 3 months post-treatment (effect sizes 0.47 to 0.68).136

Another RCT examined the short-term outcome of a trial of a manualized outpatient 15-week social skills program, the Social Skills Group Intervention – High Functioning Autism (SS GRIN-HFA).126 The study included 55 children, primarily male (98% of the total sample) with IQ>85 randomized either to SS GRIN-HFA group (mean age 10.2 years) or to a traditional SS GRIN group (mean age 9.9). Participants in the SS GRIN-HFA group showed significant improvement in social skills, with significantly better scores than the control arm on all Social Responsiveness Scale domains except social cognition (effects sizes ranging from -0.67 to -0.94). In addition, parents of children in the treatment group reported significant improvement in the areas of their child's social awareness, motivation for social interaction, social communication skills, and unusual mannerisms associated with ASD. No significant difference was found between the treatment group and control group regarding child self-report of self-efficacy or loneliness.

A final RCT examined followup of the Children's Friendship Training (CFT) manualized program.117, 134 In the initial report included in the 2011 review, 117 76 children with ASD enrolled in second to fifth grades were randomly assigned to the treatment group (n= 40) or the delayed treatment group (n=36). Weekly 60-minute treatment sessions were held over 12-weeks, with parent and child training occurring concurrently in separate locations. Skills targeted as part of the treatment included conversational skills, peer entry skills, developing friendship networks, good sportsmanship, host behavior during play dates, and handling teasing. Participants in the treatment group demonstrated modest gains in the number of hosted play dates (p<.001) as well as a decrease in electronics-use during play dates (p<.001). Participants in the treatment group also demonstrated less disengaged behavior (p <. 001), internalizing behavior (p <.001), and less conflict during play dates (p =.069). In a followup analysis,134 24 participants from the initial study were followed to examine maintenance of skills. At long-term follow-up 1 to 5 years post-participation in the training, participants continued to demonstrate increased social opportunities through invited play dates, maintenance of friendships, and decreases in loneliness from baseline (p<.05). Participants also demonstrated maintenance of gains in overall social skills along with reduction of problem behaviors (p<.05).

One Japanese pilot RCT135 examined the Treatment and Education of Autistic and Related Communication Handicapped Children (TEACCH)-based manualized group social skills training. The study included 11 children (mean age=64 months) with High Functioning Autism (HFA) (IQ >75) and their mothers, who were randomly assigned to the TEACCH program (n=5) or a wait-list control group (n=6). The treatment group consisted of weekly 2-hour sessions, with 20 sessions over 6 months. The control group consisted of group meetings with the mothers on a bi-monthly basis, consisting of 30–60 minute meetings with two social workers. Participants in the treatment group showed moderate improvement in adaptive behaviors and social reciprocity of the children, parenting stress, and parent–child interactions compared with the control group.

Two RCTs124, 125 and one nonrandomized controlled trial138 assessed interventions targeting social skills and incorporating typically developing peers or siblings. Studies included 21 to 60 participants, generally with high functioning ASD. One RCT examined short-term outcomes of a trial of an outpatient peer tutor social skills training program.124 The study included 44 children (mean age=9 years, IQ>70) who met criteria for a pervasive developmental disorder. Sixteen out of 23 participants in the treatment group were considered treatment responders as rated by their parents, compared with 0/18 in the control arm (p≤.001). Children with Asperger syndrome were more likely to be responders compared with children with PDD-NOS (p=.03); IQ was not associated with response status. No significant differences were found between the treatment group and wait list group on social competence measures.

A second fair quality RCT evaluated child-directed social skills training (CHILD) compared with peer-mediated social skills training (PEER) applied to children with high-functioning autism attending regular education classrooms.125 The study included 60 children (mean age=8.14, mean IQ=90.7) randomized to one of four treatment groups (n=15/group): 1) CHILD group 2) PEER group 3) CHILD+PEER and 4) a control group. Treatment occurred over 6 weeks. In the CHILD condition, it included 1:1 training and practice in social skills targeting deficits identified for each child. In the PEER arm, it included peer interaction focused on positive social modeling. Participants who received PEER interventions (PEER alone or PEER+CHILD) showed significant improvements in social network salience (prominence of a child within the classroom social network) compared with the other groups (p≤.006). At the final followup 12 weeks after the end of the intervention, salience remained higher for the CHILD+PEER arm compared with CHILD alone and the control group but not compared with PEER alone. Teacher ratings of social skills also improved from baseline to final followup for the peer-mediated group as did measures of solitary engagement and joint attention.

One fair quality, non-randomized trial examined the effectiveness of including siblings in social skills training groups for boys with Asperger's Syndrome.131 The study included 21 children with Asperger syndrome between the ages of 8 and 12. Investigators partially randomized participants to one of three treatment groups (first 15 randomly assigned to one of three groups; later recruits assigned based on whether they had an older sibling; if no sibling, participants were randomly assigned to “no sibling” training or wait-list control group). Eight weekly 2-hour sessions treatment sessions were held in a clinical setting. Treatment sessions included content covering nonverbal social cues, such as eye contact, body language, tone of voice, and facial expression. Techniques included extended time, repeated practice, conceptual explanations, role play, and use of social dilemmas. Participants were also assigned a different partner each week to encourage social interaction and cooperation. Sibling participants were not given any specific training or instruction other than what was provided as part of the treatment sessions. Homework tasks were given to facilitate generalization. Participants in the active treatment groups demonstrated significant improvement in identification of nonverbal cues to identify emotions compared with the waitlist control group (effect size=0.47, p<.001). While the ability to identify social cues was maintained by the participants in the active treatment groups, no increase in skills was demonstrated at 3-months post-intervention. Parents in all groups rated socials skills for both children with ASD and siblings as improved over time (effect size=0.55, p<.001). No difference in teacher report of social skills for target participants or siblings was demonstrated.

Three RCTs, one of good and two of fair quality, addressed interventions targeting emotion recognition in children with ASD.123, 128, 132 Two studies used specialized DVDs to demonstrate emotions and one used a manualized, group-based intervention focused on Theory of Mind training, which includes recognizing emotions, understanding differences between fantasy and reality, perspective taking, and reasoning about other people's mental states. Two RCTs conducted in Australia (one good quality123 and one fair132) assessed the outcome of The Transporters DVD series as an intervention for emotion recognition. The first RCT examined changes in emotion recognition and generalization of newly acquired skills to improvements in social perception skills over a 3-month period. The study included 55 children with ASD between the ages of 4 and 7 randomly assigned to view either the Transporters DVD series or the control DVD series (Thomas the Tank Engine) for four weeks (15 minutes per day in their home setting). Parents were also provided with a diary to record the number of hours watched per day. Compared with control participants, participants in the treatment group improved in emotion identification and matching of emotions (anger only) immediately following the intervention, with improvements maintained 3-months post-intervention. Gains were also seen in the treatment group 3-months post-intervention for identification of happiness and emotion recognition within situations. In both groups, no difference was found in affect recognition, theory of mind, or social skills immediately following the intervention or at the maintenance phase. Long-term improvements in identification of happiness expressions were associated with greater ADOS severity, as was matching of emotions overall and of sadness specifically. Age was correlated with identification of fear expressions, affect recognition, and the mind reading desire-based task. Verbal IQ was also associated with some short term improvements.123

A second, 3-week RCT comparing The Transporters DVD with the control series included 25 children with PDD between the ages of 4 and 8. Parents were also provided with a user guide to facilitate their child's participation in watching the episodes as well as logbook to record the number of sessions watched per day. Participants in the treatment group improved on standardized measures of emotion and facial recognition (effect sizes range 0.48–0.92, p<.001), while both groups improved on social peer interest (effect size=0.24, p=.01) and eye contact (effect size=0.44, p<.001). In both groups, no difference was found in gaze aversion or stereotyped behavior. This study provided little information on the demographics of the participants. This study also did not provide information on the user guide, which may be a confounding variable to the obtained findings. The authors also refer to Nonverbal IQ in one of their tables, but only administered the Block Design subtest, which does not fully measure all aspects of nonverbal IQ.132

A fair-quality study examined the short-term outcome of a trial of a manualized Theory of Mind training program.128 The study included 40 children (mean age=10 years) with a diagnosis of high functioning ASD and cognitive abilities within the average range (mean IQ=100.1 in the treatment group and 103.3 in the control group). The participants were randomized either to a 16-week Theory of Mind training group or a wait list control group. Participants in the treatment group improved on their conceptual theory of mind skills compared with the control group (awareness of multiple emotions, effect size=0.84, p<.05; complex emotions, effect size=1.19, p<.01), but no significant differences were found between groups on elementary theory of mind understanding, self-reported emphatic skills, or parent-reported social behavior.

An additional study examined the short-term outcome of a trial of a pictorial system called thought bubble training on Theory of Mind tasks, including difficulty with false belief tests.133 The study included 24 children (mean age=7 years) with a diagnosis of ASD. The participants were not randomized into the thought bubble intervention group (n=17) or control group (n=7) based on standardized means, but rather on preference by school staff. The two groups were comparable in terms of chronological age, verbal intelligence, semantic language skill, syntactic language ability, and nonverbal intelligence. In addition, a within-group rather than between-group statistical analysis was utilized. Within-group analyses indicated that the children in the thought bubble intervention group showed significantly higher post-training test scores on all Theory of Mind variables. These post-test gains were also maintained at three-week followup. In contrast, the children in the control group did not show any significant improvements in their pre- and post-test scores on Theory of Mind variables, nor did they show any improvements at followup. Seven children in the thought bubble intervention group and one child in the control group were not available at followup.

Play/Interaction-Based Interventions

Key Points

  • Twelve studies addressed interaction-based approaches: three good and eight fair quality RCTs and one poor quality prospective cohort.
  • Studies of interventions targeting joint attention and delivered by teachers, parents, and interventionists reported gains in joint attention skills in treatment groups compared with controls typically over a short duration (8 to 16 weeks). Children in both treatment and comparison groups, typically received early intervention in addition to the targeted intervention.
  • One small, poor quality study of an intervention targeting pretend play showed an increase in play dialog in both groups, with a greater increase in the intervention group.
  • Studies targeting parental responsiveness to child communication reported increases in responsive parent behaviors in the treatment arms and limited increases in child communication.

Overview of the Literature

In addition to seven (reported in nine publications) comparative studies (two RCTs of fair139-141 and five of poor142-147 quality) addressing play- or interaction-based approaches described fully in the 2011 review, we identified 12 studies (reported in 16 papers) evaluating such interventions for the current review (Table 9). Among these 12 studies, one includes a population addressed in the 2011 review.140, 141, 148, 149 We considered three studies in the current review to be of good quality,150-153 eight of fair quality,140, 141, 148, 149, 154-160 and one of poor quality.161 Studies were conducted in the United States140, 141, 148-154, 156-158, 160, 161 and Europe155, 159 and included a total of 383 participants between the ages of 21 and 82 months. Intervention duration ranged from 6 to 16 weeks; three studies reported long term (≥12 months post-intervention) followup of participants.140, 141, 148-150, 156 While all studies used approaches incorporating focused interactions directed by teachers or interventionists140, 141, 148, 149, 151-153, 155, 157-159, 161 or parents/caregivers,150, 154, 160, 162 studies typically addressed outcomes related to joint attention, pretend play, imitation, or child/parent communication.

Table 9. Summary of outcomes of studies of play/interaction-based interventions.

Table 9

Summary of outcomes of studies of play/interaction-based interventions.

Detailed Analysis

Studies Addressing Joint Attention Outcomes

A fair quality pilot RCT evaluating a teacher-implemented joint attention intervention randomized child-teacher dyads in public preschools to either intervention (n=9 children, mean child age=46 ±5 months, mean mental age=30.3 ±5.01 months) or wait list control (n=7, mean age=43.01±6 months, mean mental age=33.8±8.74 months).157 The manualized JASP/ER (Joint Attention and Symbolic Play/Engagement and Regulation) intervention trained teachers in eleven key strategies including setting up the environment, following the child's lead, imitating the child's play action, contingent language, and modeling joint attention. Teachers received a 1-hour training workshop and 1-hour of individual training with the child from a JASP/ER interventionist per week. Interventionists also instructed teachers to use JASP/ER strategies daily. At the 5-week followup, children in the intervention group improved in total initiations of joint attention and in pointing compared with the control arm (p<.005) and in showing (p<.01) in classroom observations (large effect sizes for each measure, 1.85 to 2.02). Groups did not differ on measures of looking or giving. Most scores on the Early Social Communication Scales joint attention measures and frequency of joint attention initiations in videotaped interactions did not differ significantly between groups. Object engagement declined and supported engagement improved in the treatment group compared with control (large effect sizes, d=1.24 to 1.41, p≤.05). Observations of teachers also demonstrated increased use of JASP/ER strategies in the treatment arm.

In another good quality pilot RCT of JASP/ER, investigators randomized minimally verbal (<10 spontaneous functional communicative words) preschoolers enrolled in intensive ABA-based interventions for at least 12 months to either JASP/ER or control (standard intensive preschool, n=8, mean age=54.68±10.25, mean mental age=13.91±3.85).151 Treatment group participants (n=7, mean age=48.73±11.68 months, mean mental age=17.21±3.91 months) received 1-hour of JASP/ER training per week in addition to the intensive preschool. At the 3-month followup, the JASP/ER group increased in play types and decreased time unengaged significantly from baseline (p=.04). Changes were not significant for the control group. The JASP/ER group also spent less time disengaged during class observations compared with the control group (effect size=1.63, p=.05), initiated more requesting gestures (effect size=1.51, p=.01) and evidenced more diversity of spontaneous play (effect size=0.81, p=.04). Groups did not differ on Early Social Communication Scales variables related to joint attention.

Another fair quality RCT155 conducted in 59 Norwegian preschools over 8 weeks evaluated a manualized adaptation of a joint attention intervention reported below.140, 141, 148, 149 Children in the intervention group (n=34, mean age=47.6±8.30 months, DQ=53.3±19.2) attended regular or specialized ASD preschools and also received up to 80 sessions (20 minutes twice daily, 5 days/week) of intervention focused on promoting joint attention and engagement within play activities. Children in the control group (n=27, age=50.3±8.3 months, DQ=59.9±19.7) also attended regular or specialized preschools. Groups did not differ in number of preschool hours or 1:1 training or support. The control group had greater expressive language age at baseline compared with the treatment group (mean 24.9±12.8 vs. 18.8±10.5, p=.047). At the 8-week followup, frequency of joint attention skills during teacher-child play were significantly better in the treatment group compared with control (effect size=0.44) but the duration of joint engagement did not differ between groups. Duration of joint engagement was greater in mother-child play in the treatment group vs. control (mean 12.2% longer duration of joint engagement, effect size=0.67). Although initiation of joint attention skills increased in the treatment group, group differences were not significant, thus effects on joint attention seen with teachers did not generalize. Frequency of joint attention initiation as measured on the Early Social Communication Scales did not differ between groups. Adjusting analyses to account for expressive language differences did not change results. Further, investigators found no putative moderators (age, DQ, language age, program philosophy) to be significant, suggesting that the intervention may be applicable across developmental levels.155

Another fair quality RCT comparing joint attention and symbolic play interventions delivered via an interventionist included 58 children with ASD between 3 and 4 years of age. Investigators assessed language development, joint attention and play skills, and mother-child interactions at pre- and post-intervention and 6 and 12 months after the end of the 5 to 6 week intervention.140, 141, 148, 149 Children in both groups showed significantly greater growth in expressive language, initiation of joint attention, and duration of child-initiated joint attention over time than did participants in the control group (p<.01 to <.05, moderate to large effect sizes). Growth in receptive language was not significantly affected by the intervention from pre-intervention to 12 months post-intervention. Children in the symbolic play group also showed significantly more growth in play level than did children in either the joint attention (p<.01) or control (p<.001) groups.

In a subsequent report on 52 of the 58 RCT participants assessing joint attention quality, both the joint attention and symbolic play groups improved in shared positive affect during joint attention and in shared positive affect with utterances during joint attention at 6 and 12 months post-intervention (p<.05) but not at intervention exit.149 Differences between groups at the 6 and 12 month time points were not significant. The control group generally declined in instances of shared affect over the followup time points. Forty of the 58 participants in the RCT also participated in followup 5 years post-intervention.148 Fifteen of 20 children in the joint attention group, 14 of 21 in the symbolic play group, and 11 of 17 in the control returned at 5 years; mean age across groups was 8 years and 8 months. Of the 40 participants, five were enrolled in regular education, 17 in regular education with some special education support, and 18 were in special education classrooms; placement did not differ among groups. At followup, 5/15 participants in the joint attention group, 1/14 in the symbolic play group, and 2/11 in the control arm were considered non-spectrum. Thirty-two of the 40 participants achieved valid scores on language assessments at followup. Ability to use spoken language at followup (“passing” the language assessments) was predicted by children's average play level at baseline (p<.01). Number of functional play types at baseline predicted greater cognitive skills. Age at baseline, initiation of joint attention, play level and treatment group assignment predicted subsequent vocabulary ability (all p<.03); these factors together explained 64 percent of spoken language variability.

In a fair quality RCT of a joint attention intervention adapted from this study140, 141, 148, 149investigators randomized 38 caregiver/child dyads to either immediate, parent-mediated treatment (n=19) or a wait list control group (n=19).150 The 8-week treatment included individualized, developmentally appropriate play routines to promote parents' following of their children's interests and activities. Children in both groups ranged in age from 21 to 36 months (mean=30.82 months, mean mental age=19.2 months). At the end of intervention, children in the treatment group demonstrated less object-focused play, more responsiveness to joint attention, more functional play acts, and more joint engagement than children in the control group (p<.05). Groups did not differ in initiations of joint attention, diversity of symbolic play, or unengaged actions. At followup of the treatment group 12 months after the end of intervention, results suggested maintenance of gains in joint engagement, response to joint attention, and reduction of object engagement, but changes in scores were not significant. Types of functional play acts improved in the treatment group at the 12-month followup (p<.01). In analyses of potential predictors of outcome, greater caregiver quality of involvement (rated by investigators) predicted increased joint engagement (p<.05) but not other play skills or engagement outcomes. Parent-rated adherence or competence did not predict changes in any outcome. Number of hours of other intervention similarly did not predict any outcomes.

A fair quality RCT of a classroom-based joint attention or symbolic play intervention based on the manualized approaches in other studies reported above140, 141 randomized 14 special education teachers to either a symbolic play followed by a joint attention intervention (n=10 children, mean age=54.50±5.06 months, mean mental age=25.29±15.77 months), the joint attention intervention followed by symbolic play (n children=14, mean age=56.21±10.42, mean mental age=36.25±11 months), or a waitlist control (n children=9, mean age=59.67±10.61, mean mental age=30.38±13.19).158 Treatment occurred in eight weekly sessions over 8 weeks (4 weeks on either joint attention or symbolic play followed by 4 weeks on the other approach), groups did not differ on play or joint attention behaviors in classroom observations at followup. Children randomized to either treatment arm spent more time in a joint engagement state compared with the control arm (effect size=0.63). In analyses combining the treatment groups, joint engagement time, joint attention responses/minute, joint attention initiations/minute, symbolic play acts/minute, all assessed via classroom observations, increased significantly from baseline to post-intervention (effect sizes of 0.41, 0.43, 0.21, and 0.51 respectively). In investigator-mediated rating of early social communication, the number of joint attention responses increased from baseline (effect size=0.23); children were able to generalize increases in responding to joint attention to a novel individual. Initiation of joint attention did not increase significantly nor did functional play or level of structured play. No potential modifiers (age, ASD severity, mental age) were significantly associated with treatment outcomes.

Another fair quality RCT conducted in Belgium included 36 children (18 in each arm) receiving either standard care in low-intensity rehabilitation centers for children with ASD (focus on communication, social skills, play, and motor skills for 3 to 5 hours/week) or standard care + a joint attention- and imitation-focused intervention delivered for 1 hour/week (two 30-minute sessions for a total of 24 sessions).159 The joint attention/imitation intervention included games and activities to promote following and initiating requests; gaze following; pointing; initiating joint attention; and gestural, vocal, symbolic, or social imitation. Children ranged in age from 4.07 to 6.92 years, with IQs in the average to mild intellectual disability range (50–105 full scale IQ). After 12 weeks of intervention, the joint attention/imitation group had higher total joint attention scores, improved gaze following, and greater request initiations than the treatment as usual group (effect sizes 0.11 to 0.22, all p values ≤.05). The number of elicited joint attention acts increased from 6.53 to 8.41 and the number of spontaneous declarative joint attention acts increased from .89 to 1.72 for the treatment group from baseline to followup; correct imitations increased from 34.11 to 41.12. Initiating declarative joint attention decreased significantly for both groups from baseline to followup (p<.05). Scores on measures of imitation did not differ between groups, though both groups improved over time. Higher baseline verbal IQ was associated with gains in imitation in the treatment group (p<.05), but no other variables tested (age, mental age, full scale IQ, performance IQ, baseline imitation and joint attention skills) were statistically significant. Children in the treatment group improved equally regardless of age or IQ level.

Finally, a fair quality RCT of a joint attention intervention assessed the effects of a roughly 7-month home-based parent training approach targeting focusing on faces, reciprocal communication/turn-taking, and joint attention compared with community-based treatment as usual.160 The 11 participants in the experimental arm had a mean age of 24.6±4 months and mean Mullen expressive language score of 24.6±6.7 (control group: mean age=27.5±3.4, mean expressive language=24.8±6.9). Reported weekly hours, including the joint intervention sessions for experimental group participants ranged from 2.98±1.25 to 17.88±9.06. Hours/week ranged from 6.25±6.49 to 21.35±11.51 in the control arm. At followup 4 weeks post-intervention focusing on faces and responding to joint attention were significantly improved in the treatment group compared with control (p<.001); scores for the treatment arm remained significantly improved vs. the control group from the 4 week to the 8 week followup and from baseline to the 8 week followup. The effect size for between group differences at the 8 week followup on the focusing on faces outcome was 0.84 and 1.18 for responding to joint attention. Effect sizes for initiations of joint attention were not significant. Language outcomes were significantly improved for the treatment group compared with control. While both groups improved over time, Mullen receptive language and Vineland communication scores were significantly better in the treatment vs. control arm (p values <.05). Effect sizes for differences at the 4-week followup were 0.59 (Vineland) and 0.34 (Mullen); scores for the 8-week followup were not reported.

Studies Addressing Pretend Play

One poor quality nonrandomized, crossover study conducted in a private preschool included 12 high functioning children with ASD (age range 55–75 months).161 Intervention group participants received the Picture Me Playing intervention, which included scripted stories built around specific toys to model and encourage pretend play. Instances of play dialogue increased significantly following intervention for the treatment group compared with control (3.6 times more utterances over baseline vs. 1.79 times, p<.05), though frequency of play utterances in both groups improved from baseline. Gains in pretend play for both groups also generalized to a toy not used in the intervention and without scripted utterances.

Studies Addressing Imitation

A good quality pilot RCT of Reciprocal Imitation Training, which uses naturalistic approaches to promote imitation and social interaction, allocated 27 children to either Reciprocal Imitation Training (n=14, mean age=39.3±7.3 months, mental age=20.8±6.6) for 3 hours/week for 10 weeks or control/treatment as usual (n=13, mean age=36.5±8.00, mental age=17.9±7.5).152, 153 The interventionist-led imitation training included modeling of play and gestures and contingent imitation of children's responses and actions with toys. Children in both arms continued to receive between .25 and 25.5 hours of additional intervention per week. Data for 21 of the children was also reported in an earlier pilot,153 which reported gains in imitation for the treatment group compared with control (p<.05). Gains in imitation were associated with the number of spontaneous play acts at baseline. In the followup RCT,152 the intervention group made more joint attention initiations compared with control (p<.05). Intervention participants also improved on the Social-Emotional Scale compared with the control arm (p=.02). Changes in imitation were not shown to be associated with gains in social functioning.

Studies Addressing Parent/Child Communication

In a fair quality randomized trial of a focused play intervention, investigators allocated children to either the play intervention (n=36, mean age=58.3±12.7 months) or a control group (n=34, mean age=55.9±11.9 months).156 Parents of children in the treatment and the control groups could participate in a parent education program focused on advocacy for their children. Parents in the treatment group also participated in a manualized play time intervention, which used home-based sessions (90 minutes/week for 12 weeks) to promote parental engagement and encouragement of child communication. Children in both groups continued to receive a mean of 14 hours (± 5–8 hours) of school programming and individual services such as ABA-based approaches for a mean of 12±10 to 12 hours/week during the treatment phase. Children also received a mean of >12 hours of school or individual services during the 12-month followup period.

In analyses at the end of intervention, maternal synchronization (maternal direction of child attention or utterances in line with toys/actions in which child was already engaged vs. redirecting or not synchronized with child's actions) was significantly greater in the treatment group compared with control (effect size=0.08, p<.05). Maternal synchronization was moderated by baseline maternal insightfulness (p<.05) and synchronization was greater in those mothers rated as insightful compared with non-insightful (effect size=0.31, p<.05). Expressive language scores did not differ between groups at the end of intervention or at followup 12 months post-intervention (effect size for baseline to followup change=0.03, p=ns). Children with baseline expressive language abilities below 11.3 months showed greater gains in language in the intervention group vs. control (effect size=0.25 for 24 children with low language skills). The link between short-term gain in maternal synchronization and long-term language (12 months post-treatment) gains was not moderated by maternal insightfulness, nor did initial language skills moderate the link between gains in maternal synchronization after 12 weeks and long term gains in expressive language.156

Another fair quality RCT included 14 participants (age range 28 to 68 months, mean 41.14) randomized to either an adapted More Than Words curriculum focused on teaching parents to understand child communication and promote verbal responsiveness or to a waiting list.154 Treatment group parents received approximately 12 hours of training and 14 small-group parent-child coaching sessions. Overall, children had mean auditory language age of 14.79 months and expressive age of 20.21 months with greater baseline language abilities in the waitlist group compared with the treatment group. At followup, treatment group parents improved significantly compared with the control group in measures of verbal engagement with their children (p values ≤.03). Children in the treatment group increased in prompted communication acts compared with control (p<.03), but spontaneous verbal and nonverbal communication acts did not differ between groups.

Behavioral Interventions Focused on Associated Behaviors

Key Points

  • Five good quality and two fair quality studies evaluated the effects of cognitive behavioral therapy (CBT) on behaviors associated with ASD.
  • CBT improved anxiety symptoms and effects were maintained over time in six of the seven studies. The one study that did not show significant benefit compared with control group demonstrated an improvement in anxiety symptoms in the CBT group; however, it was not greater than that seen in the control group. This study was also the only study to use an active control (social recreational therapy) rather than a waitlist or treatment as usual control.
  • Two RCTs with treatment as usual control groups demonstrated significant positive effects of CBT on socialization. One study did not demonstrate significant positive effects of CBT on socialization; however, the comparison group engaged in social skills training.
  • One small RCT rated as fair demonstrated improvement in emotion regulation after treatment with CBT.
  • One good quality RCT demonstrated improvements in executive function in the CBT treatment group compared with control group receiving social skills intervention.
  • In a large fair quality RCT, augmentation of risperidone with parent training produced more significant improvement in adaptive behavior, socialization and communication than risperidone alone, but effects were not maintained after one year. This study also evaluated changes in observed appropriate behavior and did not find any between group changes.

Overview of the Literature

We identified nine comparative studies addressing interventions targeting conditions/behaviors commonly associated with ASD in the 2011 review. These studies included four RCTs163-167 and one nonrandomized trial168 of fair quality and three RCTs169-171 and one prospective cohort172 of poor quality. Studies addressed CBT for anger or anxiety or parent training approaches. In addition to these studies, we identified nine new studies (reported in 15 publications);165-167, 173-184two of these nine studies, one evaluating CBT165, 166, 178 and one assessing parent training plus risperidone,167, 179-181, 184report on populations addressed in studies in the 2011 review. As in the 2011 review, studies address either CBT or parent training modalities (Table 10).

Table 10. Summary of outcomes of studies of interventions targeting conditions commonly associated with ASD.

Table 10

Summary of outcomes of studies of interventions targeting conditions commonly associated with ASD.

Among the studies identified for the current review, eight RCTs evaluated CBT: seven conducted in the United States,173, 175-178, 182, 183 and one in Singapore.185 Three studies examined CBT compared with control groups receiving treatment as usual.173, 177, 182 Three studies examined CBT compared with wait listed controls,165, 166, 175, 176, 178 one study compared CBT with social recreational therapy,174 and another with a social skills intervention.183 Studies included two populations: five studies (reported in multiple publications) included subjects with both ASD and primary anxiety disorder diagnoses,165, 166, 173, 176-178, 182and three studies included subjects with ASD only (subjects may or may not have had a formal diagnosis of primary anxiety disorder or studies did not target anxiety).174, 175, 183 Outcomes measured included improvements in anxiety alone in five studies,173, 174, 176, 177, 182 improvements in anxiety and daily living skills in one study;165, 166, 178 improvements in executive function in one study,183 and improvements in emotion regulation in one study.175 Subjects ranged in age from 4 to 16 years. Five study interventions were conducted over 16 weeks,165, 166, 173, 174, 176-178 one study intervention was conducted over 28 weeks ,183 one over 32 weeks,182 and one over 9 weeks.175 We rated six studies as good quality165, 166, 173, 174, 176-178, 183 and two as fair.175, 182

We identified one fair quality RCT reported in multiple publications and addressing parent training approaches (also reported in the 2011 review).167, 179-181, 184The study examined the utility of augmenting risperidone with parent training vs. risperidone alone for treatment of serious behavior problems and irritability. Children had diagnoses of ASD in addition to serious behavior problems as defined by reaching specific cutoff scores on measures of irritability and problem behavior, and ages ranged from 4 to 13 years. Outcomes measured included measures of adaptive behavior in addition to measures of problem behavior and irritability and one observed measure of appropriate behavior.

Detailed Analysis

Most studies investigating CBT as the primary intervention identified anxiety as the target symptom. One good quality RCT measured changes in anxiety symptoms in addition to core ASD symptoms.177 The study included 36 children ages 7 to 11 with both ASD and primary anxiety disorder diagnoses. Subjects were randomized to an intervention group receiving 16 weekly CBT sessions or a control group receiving treatment as usual. There were no significant group differences with the exception of slightly higher proportion of subjects with Autistic Disorder compared with PDD or Asperger's in the intervention group. Primary outcome measures included the following measures of anxiety; Pediatric Anxiety Rating Scales (PARS), Anxiety Disorders Interview Schedule-IV-Child/Parent Version and Clinical Global Impressions-Severity (CGI-S). Secondary outcome measures included other measures of anxiety such as the Multidimensional Anxiety Scale for Children-Parent Version and Child Behavior Checklist, a measure of social responsiveness, the Social Responsiveness Scale, and the Columbia Impairment Scale-Parent Version, which assesses interpersonal, social and academic skill. All measures were collected at baseline, the end of the intervention and 3 months following termination of the intervention. At the end of the intervention, large treatment effects were observed in all primary outcome measures. Pediatric Anxiety Scale ratings were reduced by 21 percent in the CBT group vs. 9 percent in the control group. CGI-S scores were more improved in the CBT group than the control group (effect size 1.06, p<0.01). On the blinded, clinician-rated Anxiety Disorders Interview Schedule, 38 percent of CBT participants vs. 5 percent of control participants showed clinical remission of anxiety symptoms (effect size 1.37, p=0.01). Scores on all measures did not change significantly between the end of intervention and the 3-month followup evaluation. Among secondary outcome measures, group differences were observed with greater improvements on the Columbia Impairment Scale, internalizing symptoms on the Child Behavior Checklist, Revised Children's Manifest Anxiety Scale anxious arousal subscale, total score and social communication and social mannerisms subscales on Social Responsiveness Scale. No group differences were observed on externalizing symptoms of the Child Behavior Checklist, dysphoric mood, oversensitivity and worry subscales of the Revised Children's Manifest Anxiety Scale, or social awareness, social cognition and social motivation subscales of the Social Responsiveness Scale.

Another good quality RCT assessed a CBT-based intervention specifically developed for children with ASD (“Facing Your Fears”).173 The study included 48 children ages 7 to14 with ADOS-confirmed diagnosis of ASD randomized to either the CBT group or treatment as usual. Participants were required to be able to speak in full complex sentences and have clinically significant symptoms of anxiety measured on the Screen for Child Anxiety and Related Emotional Disorders-parent version (SCARED). No group differences were identified relative to age, IQ, sex, parents' marital status, mother's education, ethnicity, specific ASD diagnosis, or use of psychiatric medications. The intervention consisted of 12 multifamily group sessions over 4 weeks following the manualized CBT treatment. The Anxiety Disorders Interview Schedule for Children was performed at baseline and again at the end of the intervention. The CGI-S scale was obtained at the end of intervention. Independent Clinical Evaluators (ICEs) blinded to the participant's condition assigned DSM-IV diagnoses and provided summary codes of clinical severity and interferences called Clinician Severity Ratings. Group differences in severity ratings were noted for all anxiety diagnoses with medium to large effect sizes. The overall number of anxiety disorders at followup was significantly reduced in the intervention group, and there was a large effect size noted in the reduction of generalized anxiety disorder diagnoses. There were no group differences noted in diagnostic status for other anxiety diagnoses. Significant improvement was noted on the CGI-S in the intervention group as compared with the control group (effect size 1.03 and p=0.003). The SCARED was repeated at 3 and 6 months for the intervention group and indicated that reduction in anxiety symptoms had been maintained.

A third good quality RCT investigated the effects of the Coping Cat CBT program on anxiety symptoms in 22 children ages 7 to 14 with diagnosis of ASD and at least one primary anxiety disorder.176 Twelve children were assigned to the intervention group and the remaining 10 children were enrolled as waitlisted controls. There were no baseline group differences with the exception of more children in the control group receiving stimulant medications. The intervention consisted of 16 weekly 60 to 90 minute CBT sessions following the Coping Cat treatment manual. Anxiety measures were repeated just after completion of the intervention and again at 2 months after completion of treatment. At the completion of the intervention, 58 percent of the intervention group compared with 0 percent of the control group no longer met criteria for a primary anxiety disorder (p=0.003). Spence Children's Anxiety Scale ratings improved significantly in the intervention group (34.92 to 20.08) but not in the control group (32.3 to 31.7) (p=0.02). Co-morbid diagnoses decreased in the intervention group compared with control group from baseline to end of intervention (p<0.001). After 2 months, four of 11 intervention group participants continued to not meet requirements for anxiety disorder diagnosis. The authors reported a number needed to treat for the intervention of 1.72.

A good quality RCT conducted in Singapore compared the effects of CBT to an established social recreational intervention on anxiety symptoms.174 Seventy children with ASD diagnoses, verbal IQ>80, and perceptual reasoning IQ>90 were randomly assigned to the CBT group (n=36) or social recreational group (n=34). The CBT group had slightly higher verbal IQ (100.25 in CBT group compared with 93.06 in social recreational group), otherwise there were no significant differences between groups. The CBT group underwent 16 weekly 90 minute small group CBT sessions. The social recreation group underwent 16 weekly 90 minute small group sessions following a manualized treatment protocol that included activities aimed at independent living, self-engagement, motor coordination, intellectual stimulation and socialization. The Spence anxiety scale and CGI-S were repeated at the end of treatment, 3 months and 6 months after the end of treatment. Both groups demonstrated reduction in anxiety on the Spence scale between baseline and at 6-month followup; however, only the social recreational group demonstrated reduction in anxiety immediately following intervention. CGI-S scores improved over time for both groups, but group differences at final followup were not significant.

Another fair quality RCT was conducted in the United States182 evaluating the Building Confidence CBT program modified for use in children with ASD. The study included 12 children ages 7 to 11 years meeting criteria for both ASD and at least one anxiety disorder who had verbal IQs greater than 70 and no other primary psychiatric diagnosis. The intervention group underwent 32 weekly 90 minute sessions and was compared with a treatment as usual control group. There were no significant differences between groups. The outcome measured in this study was diagnosis of anxiety disorder and severity of symptoms at the end of the intervention. At the end of intervention, fewer children in the treatment group had an anxiety diagnosis (p=0.013); severity of anxiety was also more significantly reduced in the treatment group compared with the treatment as usual arm (p=.017)

One good quality RCT reported in multiple publications165, 166, 178 examined the effects of the Building Confidence CBT program adapted for children with ASD on anxiety symptoms, daily living skills, and, in a subgroup of children, socialization. Forty children ages 7 to 11 with ASD and separation anxiety, social phobia, or obsessive-compulsive disorder and IQ >70 were randomized to the CBT group or to waitlist control group. No group differences were noted with the exception of more children in the CBT vs. control group having comorbid diagnosis of major depressive disorder or dysthymia (18% vs. 0%, respectively). The intervention consisted of 16 weekly 60–90 minute CBT sessions. Assessments of anxiety included the Anxiety Diagnostic Interview Schedule, the Multidimensional Anxiety Scale for Children parent and child reports, and the Clinical Global Impressions-Improvement (CGI-I) scale. Measures of daily living skills included the Vineland and the Parent Child Interaction Questionnaire, which assesses the level of parent involvement in daily living skills. Socialization was measured with the Social Responsiveness Scale in a group of 19 children from the early stages of recruitment. Most measures were repeated at baseline, at the end of the intervention and, for 10 intervention participants who were still available, at 3 months after the end of intervention. The CGI-I was only collected at the end of intervention and at the 3-month followup. At the end of intervention, 92.2 percent of the intervention group met criteria for positive treatment response based on CGI-I and 64.3 percent no longer met criteria for any anxiety disorder on the Anxiety Disorders Interview Schedule, compared with only 9.1 percent demonstrating positive treatment response on the CGI-I and (p<0.0001) and 9.1 percent no longer meeting criteria for anxiety disorder in the control group (p<0.0001). Overall this data did not change significantly at the three-month followup period. The MASC scores were significantly lower in the intervention group vs. the control group at followup (p<0.0001) for the parental report however the child report did not demonstrate significant differences. This data also did not change significantly at the 3-month followup period. Vineland total daily living and personal daily living raw scores significantly improved for the intervention vs. the control group (p<0.05) with effect sizes of 0.45 for total daily living skills and 0.50 for personal daily living skills. Unnecessary parental involvement and parental involvement in child self-care were significantly reduced in the intervention vs. control groups (p<0.05 and p<0.01, respectively). Treatment effects on the Vineland and parental intrusiveness scales were maintained at 3 months post intervention in the 10 children for whom followup data were available. Among those participants receiving the Social Responsiveness Scale, differences favoring the intervention group were found on three of the five subscales including social communication, social motivation and social awareness (p<0.05).

A small, fair quality pilot RCT examined the utility of CBT to improve emotion regulation in a young group of 11 verbal children ages 5 to7 years.175 Children randomized to the intervention group (n=5) underwent 9 weekly 60 minute sessions of CBT focusing on skill-building, stress management and understanding expression of emotions. The remaining 6 children were randomized to a waitlist control group. This study reported demographic data for all participants but did not present data regarding potential differences between groups. Measures of the child's capacity for emotion regulation was assessed through his report of number emotion regulation strategies that might be used during the reading of a vignette, parental report on an emotion regulation scale, parent observation and notation of frequency and duration of anger/anxiety episodes, and parent report of their own self-confidence and confidence in their children's abilities to handle emotions. Measures were collected at baseline and at the end of intervention. At the end of intervention children in the CBT group reported a greater number of emotion regulation strategies in response to the vignettes (4 vs. 1.29 in control group p<0.05, effect size 0.65) and parents had greater confidence in their ability to manage child's anger and greater confidence in the child's ability to manage their own anger (p<0.05, effect sizes 0.84 to 0.89).

A good quality RCT investigated the effects of a CBT program, Unstuck and On Target compared with a social skills intervention on 57 children ages 7 to 11 with ASD.183 Children received either intervention weekly for 28 30–40 minute sessions. All children were required to meet ADOS criteria for ASD, have a full scale IQ greater than 70 and mental age greater than 8 years old. Baseline measures were obtained but not reported. The study does not report at what point post intervention measures were obtained. Both groups improved on most measures from baseline to followup. The CBT group improved significantly more on interventionist-rated measures of problem solving, flexibility, and parent and teacher-rated executive function measures when compared with the social skills group (p <0.05 with medium to large effect sizes). In classroom observations, the CBT group demonstrated greater improvement in ability to follow directions, transition smoothly and avoid “getting stuck” (p values <0.05). Higher baseline scores predicted greater improvements in flexible thinking, social tasks, parent- and teacher-rated executive function shift and planning/organization measure, parent-rated Social Responsiveness Scale total score (p values <0.05). Higher IQ predicted greater improvements in flexible thinking and the challenge task plan measure. Younger age predicted greater improvement on the challenge task and parent-rated executive function measures of shift and planning/organization (p<0.05). Female sex predicted greater improvement on the parent –rated Social Responsiveness Scale total score (p values <0.05).

One fair quality RCT (reported in multiple publications) assessed a parent training approach (treatment with risperidone alone vs. risperidone augmented with a parent-training program) to improving adaptive behavior and communication and socialization skills.167, 179-181, 184 The parent training program included 11 core sessions, one home visit and up to three optional sessions during the first 16 weeks, followed by four booster sessions over the next 8 weeks. The training focused first on antecedents, purpose, and reinforcements of problem behaviors and then on teaching parents management strategies for these behaviors. Investigators recruited 124 children ages 4 to 14 years with ASD, severe problem behaviors evidenced by positive scales on the Aberrant Behavior Checklist-Irritability subscale and CGI-S subscales, and IQ>35. Forty-nine participants were randomized to risperidone plus parent training intervention group and 75 to the risperidone alone control group. No group differences were observed with the exception of slightly higher ABC-irritability subscale scores in the intervention group.

The Aberrant Behavior Checklist, Vineland, the Home Situations Questionnaire, and the Standardized Observation Analogue Procedure were completed at baseline, at 24 weeks after completion of intervention and, for the Aberrant Behavior Checklist and Home Situations Questionnaire, one year after intervention. At 24 weeks, scores on the Home Situations Questionnaire demonstrated decreased severity in more children in the intervention group vs. control (p<0.006), and greater improvements were noted in the intervention group on the Aberrant Behavior Checklist Irritability (p=0.01), Stereotypic behavior, (p=0.04) and Hyperactivity (p=0.04) subscales compared with the control group. Also at 24 weeks post intervention, greater improvements in the intervention group were noted on Vineland socialization (p=0.01) and adaptive composite (p=0.05) standard scores and on Vineland noncompliance (p=0.03), socialization (p=0.03) and communication (p=0.05) age equivalent scores. These treatment gains were not associated with IQ or adaptive or maladaptive behaviors. Analysis indicated higher baseline Home Situations Questionnaire scores predicted greater improvement regardless of treatment (p=0.007). Authors also analyzed 21 potential moderator variables and none significantly moderated Home Situations Questionnaire or Aberrant Behavior Checklist-Hyperactivity scores, suggesting that parent training may be effective for a range of children. Scores on the standardized observation measure indicated no between group differences in child inappropriate behavior in direct observations under various conditions (free play, restrictive, etc.). In analyses combining both groups, child inappropriate behavior decreased from baseline in the demand and tangible restrictive conditions (p values<.01). Additionally, this measure reported an increase in compliance in the demand condition (p=.0004) when groups were combined.

At 1-year followup, data were available for 87 participants. Group differences at one year on the Home Situations and Aberrant Behavior Checklists were no longer significant. Data were not available for Vineland at one-year followup.167, 179-181

Other Behavioral Interventions

Key Points

  • In one study comparing CBT plus melatonin to either melatonin or CBT alone, all participants improved on measures of sleep quality, with the combination group generally improving more than the others.
  • One small, short-term study of a sleep education pamphlet for parents demonstrated little positive effect of the pamphlet; similarly, a short-term study of parent training in sleep routines reported some within-group improvements in time to fall asleep.
  • Small, short-term studies of neurofeedback reported some improvements on parent-rated measures of communication and tests of executive function

Overview of the Literature

We classified studies not cleanly fitting in any of the other categories as “other.” In addition to two poor quality RCTs targeting neurofeedback186, 187and described fully in the 2011 review, we identified six new studies (seven publications) evaluating interventions targeting sleep behaviors,188-190 feeding difficulties in ASD,:191 and neurofeedback 192-194 (Table 11).We considered one RCT comparing the effects of CBT with or without melatonin with placebo on sleep habits as fair quality,189 two RCTs evaluating the effects of sleep education as fair quality,188, 190 and two studies (reported in three publications)192-194of neurofeedback as fair192 and poor193, 194 quality, and one study targeting mealtime behaviors as poor quality.191 Studies were conducted in Europe189, 192-194 and the United States188, 190, 191 and included 303 total participants with ages ranging from 2 to 12 years. Duration of intervention ranged from 3 to 12 weeks.

Table 11. Summary of outcomes of behavioral-other studies.

Table 11

Summary of outcomes of behavioral-other studies.

Detailed Analysis

One fair quality RCT compared CBT alone, melatonin alone, CBT plus melatonin, and placebo in 160 children with ASD between the ages of 4 and 10 years.189 CBT consisted of four 50-minute sessions focused on recognizing dysfunctional attitudes about sleep, parent-management of children's sleep, and replacing poor sleep habits with appropriate behavior. Participants received 3 mg controlled release melatonin administered at the same time each day. Investigators allocated 40 participants to each group; mean age across groups ranged from 6.3 to 7.1 years, and each group lost 5 to 8 participants over the 12-week intervention due to withdrawals or missing actigraphy data. All active treatment groups improved in most measures of sleep quality compared with the control group (p<.01). In general, the combination group improved more than the others, followed by the melatonin alone and CBT alone groups. Scores for children who received melatonin alone improved on bedtime resistance, sleep onset delay, sleep duration, and night waking compared with the CBT group (p<.001). Effect sizes (exact data not reported) ranged from medium to high. Sleep onset latency (time to fall asleep) and sleep efficiency (ratio of total sleep time to total time in bed) were reduced by 50 percent (sleep latency) or 85 percent (efficiency) in 85 and 63 percent of children in the combination group and 39 and 46 percent of children in the melatonin group, respectively. In the CBT arm, 10 percent of children met each criterion, and no children in the control arm achieved these percentages of reduced latency or improved efficiency. The study reported no significant harms.

One fair quality RCT evaluated the effects of a sleep education pamphlet compared with no intervention in 36 children with ASD between the ages of 2 and 10 years.188 Parents of children in the intervention group received a four-page pamphlet with information about sleep environment, promoting bedtime routines and schedules, teaching children to fall asleep alone, avoiding naps where possible, and promoting a sleep/wake schedule; parents did not receive additional instruction. At the 2-week followup, groups did not differ significantly on sleep latency, waking after sleep onset, total sleep time, or sleep fragmentation. Sleep efficiency (total sleep time/time in bed) improved slightly in the intervention group (baseline mean 75.5%±6.1, followup 77.8%±7.0 vs. baseline mean of 76.8%±6.0, followup 75.1%±6.7 for the control group, p=.04).

A final fair quality RCT assessed short-term group or individual format sleep education for parents.190 Participants (n=80) received 1 to 4 hours of education focused on bedtime routines, sleep environment, and sleep resistance in ASD. Followup measures did not differ for any outcome at followup; however, in analyses combining data for the group and individual-education arms, sleep latency (time to fall asleep) was significantly reduced from baseline (p<.001) as was sleep efficiency (p<.001), though the improvement in efficiency (% sleep time out of total time in bed) was not clinically meaningful. Insomnia-related parameters on the parent-rated Children's Sleep Habits Questionnaire (sleep onset delay, night wakings, sleep duration, bedtime resistance, sleep anxiety) were also significantly improved from baseline to followup (all p<.001) in combined analyses.

In a nonrandomized trial including 14 high functioning children with PDD-NOS (IQ≥70) investigators assigned children to 40 sessions of neurofeedback (n participants=7, mean age=9.63±1.53 years) sessions designed to treat individuals with ADHD or to a wait-list control group (n=7, mean age=10.64±1.41 years).194, 195 Electroencephalogram data did not differ significantly between groups at followup; however, the treatment group improved on some executive function measures (auditory selective attention, inhibition of verbal responses and impulsive tendencies, all p<.05) and in nonverbal communication compared with the control group. Cognitive flexibility and goal setting improved for the treatment group vs. control but ability to recognize words did not. Parents of children in the treatment arm also rated their children's communication skills as improved following neurofeedback training. In analyses 12-months post-treatment combining data for the treatment and control group participants who went on to complete neurofeedback training (n=NR), gains in auditory selective attention, non-verbal communication, and parent measures of social behavior continued.

In an RCT evaluating neurofeedback, 10 children (mean age=9.43±1.44 years) received 40 neurofeedback sessions aimed at decreasing theta power in the frontal and central brain areas. Ten children served as controls (mean age=9.14±1.34 years); the study did not specify if control children received any type if intervention.192 In contrast to the prior neurofeedback study, children had diagnoses across the ASD spectrum, treatment occurred in school and at home, and both parents and teachers completed outcome questionnaires. Immediately after treatment, theta activity was reduced in 60 percent of the intervention group. Social behavior, especially reciprocal social interaction, as measured on the parent-rated Social Communication Questionnaire, improved for the treatment group compared with control (p<.05) as did scores on the Children's Communication Checklist and on the set-shifting domain of executive function (p<.05). Scores on other domains of executive function did not differ between group nor did scores on teacher-rated measures. At followup 6-months post-treatment, the intervention group showed continued improvement on parent-rated measures of social behavior, communication, and repetitive behavior as well as set-shifting compared with the control arm (p<.05) Parents were not blinded to treatment condition.

Finally, one poor quality RCT assessed the effectiveness of an 8-week manualized parent training program on mealtime behaviors.191 Baseline BMI among the 19 participants (age range 68–91 months) was in the normal range. Between group differences at followup were not significantly different on any mealtime behavior measures. Parenting stress was significantly reduced in the treatment group compared with the waitlist control (p=.01).

KQ2. Modifiers of Treatment Effects

Key Points

  • Not all studies were adequately designed or powered to assess modifiers of effects.
  • Associations of outcome and baseline measures of cognition, adaptive behavior, language, and ASD severity were mixed across studies.
  • In early intervention studies, younger age was associated with greater improvements, though effects were not always consistent.

Overview of the Literature

Understanding the degree to which child characteristics (i.e., specific ASD-related difficulties and skills), treatment factors (e.g., type, duration, intensity), and systems (e.g., family, community) influence response to treatments could improve targeting of treatments to the appropriate children and circumstances. Twenty papers (described in multiple publications) reported predictor, moderator, or mediator data;72, 76, 77, 79, 81-84, 86, 87, 90, 93, 95, 100, 101, 105, 123, 124, 132, 152, 153, 156, 159, 167, 179-181, 183 however, not all studies were adequately designed or powered to assess modifiers of effects.

Detailed Analysis

Child-Related Factors

Age

As in the 2011 review, several studies reported associations between age at intake and improved outcomes. In one RCT of an approach incorporating parent training, younger age was associated with greater improvements: greater language gains were seen in children who were younger with lower functioning levels at baseline.72, 105

Age effects were not consistent, however, and may reflect characteristics of subgroups and treatment characteristics that need further elucidation. For example, one study comparing preschool-delivered intensive early intervention and treatment as usual reported larger adaptive behavior gains for older children in the early intervention group.77 Another RCT compared early intensive treatment delivered by parents and by specialized center staff with eclectic treatment and identified predictors of progress: in the parent training group, older children achieved better adaptive behavior outcomes; younger children made more gains in early language comprehension and production.

In a retrospective cohort study of a community-based early intervention program, outcomes were related to age at enrollment, treatment duration, and higher baseline adaptive scores. A significant interaction emerged between age at enrollment and group membership, with younger starting age influencing outcomes for the treatment group but not the waitlist control.86 In contrast to the early intervention studies, in an RCT assessing emotion recognition, older age was correlated with improved identification of fear expressions, affect recognition, and the mind reading desire based task.123 Another RCT of a preschool-based joint attention intervention compared an 8-week treatment program focused on increasing initiating, giving, and sharing joint attention skills plus preschool to preschool alone in 61 children with ASD.155 In exploratory analyses, investigators found no putative moderators (age, developmental quotient, language age, program philosophy) to be significant, suggesting that the intervention may be applicable across developmental levels.

IQ/Cognition

Associations of outcome and IQ or measures of cognition were mixed across studies. Intervention efficacy was associated with baseline cognitive scores in one early intervention study comparing preschool models classrooms,83 with higher baseline cognitive scores associated with less improvement in children in TEACCH model classrooms. In an early intervention prospective cohort study, baseline IQ was positively correlated with socialization, communication, daily living, and composite score gains on the Vineland in the treatment group; however, baseline IQ did not correlate with IQ at followup.77 In another early intervention study, higher pre-treatment mental development state and early language skills predicted better outcome on parent-reported adaptive behaviors in the eclectic treatment group.101, 102 In a study assessing emotion recognition, higher verbal IQ was associated with some short term improvements in fear recognition and mind reading tasks,123 while in another emotion recognition RCT, IQ was not correlated with improved outcomes in either the treatment or control groups.132 In another RCT of a group-based social skills approach, IQ was not associated with response status;124 similarly, treatment gains were not associated with IQ in an RCT comparing parent training plus risperidone to risperidone alone.167, 179-181 In one study of CBT focused on executive function outcomes, higher baseline scores predicted greater improvements in flexible thinking, social tasks, parent- and teacher-rated executive function shift and planning/organization measure, parent-rated Social Responsiveness Scale total score (p values <0.05). Higher IQ predicted greater improvements in flexible thinking and the challenge task plan measure. Younger age predicted greater improvement on the challenge task and parent-rated executive function measures of shift and planning/organization (p<0.05). Female sex predicted greater improvement on the parent –rated Social Responsiveness Scale total score (p values <0.05). 183 In a play-interaction study targeting imitation and joint attention, higher baseline verbal IQ was associated with gains in imitation in the treatment group (p<.05), but no other variables tested (age, mental age, full scale IQ, performance IQ, baseline imitation and joint attention skills) were statistically significant. Children in the treatment group improved equally regardless of age or IQ level.159

ASD Severity/Symptom Severity and Diagnoses

In some studies, children with lower symptom severity or less severe diagnoses improved more than participants with greater impairments. In an RCT assessing ABA-based early intervention, lower baseline ASD severity was associated with parent-reported cognitive and adaptive growth for children who received eclectic vs. ABA intervention, but not with improvements in standardized cognitive test scores.81, 82 A prospective cohort study of preschool-based early intensive intervention reported that children in the early intervention group with PDD-NOS or Asperger diagnoses (but not autism) had greater gains in overall adaptive behavior, communication, and daily living skills.77 A prospective cohort study comparing four early intervention approaches (home-based 1:1 ABA intervention, low intensity home-based programming for children with special needs [portage], home-based, local health authority-developed intervention incorporating parent training, and special education nursery/preschool) evaluated relationships between ASD severity, time in intervention, and effectiveness of intervention.95 Hours of intervention ranged from 2 to 40 across groups, with the home-based ABA group receiving the most (mean 30.4/week) and the Portage group the least (mean 8.5/week). Baseline ASD severity and total intervention hours modified effects of treatment significantly. First, baseline ASD severity was inversely related to composite change scores for all but the home-based ABA group and was positively related in that group. That is, children with more severe ASD symptoms made more progress in ABA and less in the other intervention groups. Second, more intervention time was negatively related to composite change scores for children in ABA but not in the other groups. More hours of ABA were associated with less progress relative to more hours of school enrollment or other home-based interventions.

Two reports88, 90 including participants in a retrospective cohort study evaluating an early intervention approach86 assessed potential outcome predictors including baseline age, Vineland scores, IQ, and ASD severity (CARS). Younger age at intake, higher initial developmental levels90 and treatment intensity88, 90 were related to better treatment outcomes. Vineland standard scores and IQ and mental age were higher for the 32 children whose followup standard scores on cognitive and/or adaptive behavior were in the low average range or better (>85) and whose CARS scores were in or very close to the non-ASD range (<30), Similarly, these “average outcome” children had significantly lower intake CARS severity scores, began intervention earlier (mean 42 months vs. 55 for rest of sample), and received intervention for a longer duration. More of these children also had diagnoses of PDD-NOS. Children who had poor outcomes at followup (n=75) had statistically significantly lower baseline IQ , mental age, rate of development, and Vineland scores (except for the socialization domain), with p values ranging from .01 to <.001. Differences likely were not clinically significant, however, and diagnostic category, severity, age at entry, and duration of therapy were not significantly different in the poor outcome group compared with the rest of the sample.

In an RCT evaluating an emotion recognition intervention, long term improvements in identification of happiness expressions were associated with greater ADOS severity, as was matching of emotions overall and of sadness specifically.123 In an RCT of a theory of mind training program, children with PDD-NOS improved on most measures of emotion recognition while children with Asperger syndrome improved only in understanding of complex emotions.128 In another RCT of a group-based social skills approach, children with Asperger syndrome were more likely to be responders compared with children with PDD-NOS (p=.03).124

Finally, an RCT assessing a parent training approach targeting challenging behaviors examined 21 candidate predictors and moderators of outcome scores on the Home Situations Questionnaire (HSQ) and the Aberrant Behavior Checklist, Hyperactivity/Noncompliance (ABC-H) scale.167, 179-181 Children received either parent training plus risperidone (n=75, mean age=7.4) or risperidone alone (n=49, mean age=7.5); thus, potential moderation of effect reflects the combination of parent training and risperidone while predictors of effects reflect the impact of risperidone with or without parent training. Investigators examined variables including parent training adherence, age, IQ, family income, maternal education level, parent stress, and child baseline ratings on measures including the Vineland and ABC. Only higher baseline scores on the HSQ (greater noncompliance) predicted greater improvement in either treatment condition (p=.007), with the lower HSQ group demonstrating less mean improvement than those with higher baseline HSQ scores. Though not significant, older children had slightly more improvement than younger children. No variables predicted ABC-H outcomes, though children with higher baseline Vineland composite and communication subscale scores had greater improvement on the ABC-H. While not a significant predictor of outcomes, greater parent adherence to the training program was correlated with better HSQ outcomes (p=.006), but adherence did not correlate with ABC-H scores. No candidate variables were found to moderate the relationship between parent training and HSQ or ABC-H outcomes, which may suggest that parent training is appropriate for the broader range of children with ASD.

Adaptive Behavior

Studies reported mixed findings related to outcomes associated with baseline adaptive behavior. In one retrospective cohort, positive outcomes in both the early intervention and the waitlist control groups were related to higher baseline adaptive scores.86 In one early intervention study, initial higher adaptive behaviors predicted better post-treatment early language comprehension.101, 102 In an RCT comparing risperidone alone and risperidone plus parent training, treatment gains were not associated with adaptive or maladaptive behaviors.167, 179-181

Language/Communication

The impact of language skills and attention to objects (vs. people) were assessed in three studies. In one RCT of the More Than Words program, the treatment group showed differential effects on child communication depending on children's baseline object interest; children with lower levels of baseline object interest had greater growth in communication skills, whereas children with higher levels of object interest showed attenuated growth.93 In another study of play-focused intervention, children with baseline expressive language abilities below 11.3 months showed greater gains in language in the intervention group vs. control (effect size=0.25 for 24 children with low language skills).156 In another early intervention study, children who gained more language comprehension had higher adaptive behavior scores pre-treatment. Pre-treatment language comprehension also predicted post-treatment language production.101, 102

An RCT evaluating an imitation-based approach to affect social functioning152, 153assessed whether changes in social functioning were tied to changes in participants' imitation skills. Gains in imitation were associated with the number of spontaneous play acts at baseline; however, changes in imitation were not shown to be associated with gains in social functioning. This finding could be because the study had too few participants (n=27) to detect such an effect.

Other Factors

One RCT compared the effects of a 6-week joint attention or symbolic play intervention with a control arm in participants receiving 30 hours of early intervention; at the 5 year followup, investigators assessed diagnoses and language skills for 40 of the 58 original participants.140, 141, 148, 149Investigators also identified potential predictors of vocabulary and cognitive changes via regression analyses. Potential predictors included child age, sex, maternal education, play levels and types, and joint attention responses. Ability to use spoken language at followup (“passing” the language assessments) was predicted by children's average play level at baseline (p<.01). Number of functional play types at baseline predicted greater cognitive skills. Younger age at baseline, initiation of joint attention, play level and treatment group assignment (either joint attention or symbolic play) predicted subsequent vocabulary ability (all p<.03); these factors together explained 64 percent of spoken language variability. Importantly, this study is limited in that children were often receiving intensive levels of intervention outside of the intervention setting, making impact of prescribed intervention hard to determine.

Parent-Related Factors

Four early intervention studies assessed variables related to parents/caregivers. In one RCT incorporating parent training,72, 105 parents in the additional treatment group showed increased responsiveness to their children during videotaped interactions, which was correlated with reduced ASD symptom severity (p=.049). No between-group differences were found in adaptive behavior or parenting stress. In another parent training RCT, parents in the professionally led group with low baseline self-efficacy reported higher followup self-efficacy levels than parents in the video arm.100 In a report87 also including a population reported in a retrospective cohort86, parental stress was not associated with any outcomes. In both the early intensive intervention and eclectic treatment control group in one study, child outcomes on early language skills, mental developmental state, and adaptive behaviors were significantly influenced by self-reported parental stress, children's ability to respond correctly to prompts, the number and difficulty of treatment targets, and children's problem behaviors in sessions. Children who were perceived by their parents as more difficult demonstrated less improvementin ASD severity.101, 102

Two play/interaction-focused RCTs assessed parent responsiveness and adherence to the treatment approach on treatment effects. One study comparing an 8-week caregiver-delivered joint attention approach with a waitlist control assessed intensity of total hours of intervention (external to the study), investigator-rated quality of caregiver participation, and parent-rated adherence as predictors of outcomes at the 12-month followup.150 Greater caregiver quality of involvement predicted increased joint engagement (p<.05) but not other play skills or engagement outcomes. Parent-rated adherence or competence did not predict changes in any outcome. Number of hours of other intervention similarly did not predict any outcomes.150

Another RCT compared a 12-week intervention targeting parental responsiveness to children's playtime communication compared with a control group that received some parental education about developmental and educational needs.156 Investigators also explored relationships among maternal synchronization (responsiveness to child communications) and long-term (12 months post-intervention) child language outcomes. Maternal synchronization was moderated by baseline maternal insightfulness (p<.05) and synchronization was greater in those mothers rated as insightful compared with non-insightful (effect size=0.31, p<.05). The link between short-term gain in maternal synchronization and long-term language (12 months post-treatment) gains was not moderated by maternal insightfulness, nor did initial language skills moderate the link between gains in maternal synchronization after 12 weeks and long term gains in expressive language.156

Intervention-Related Factors

Several studies of early intensive behavioral and developmental approaches evaluated potential effects associated with characteristics of the interventions themselves. In an RCT evaluating the LEAP program (full training compared with training manuals only), the students of teachers rated as having better intervention fidelity showed better outcomes on all measures.84 In other studies assessing ABA-based early intervention, where examined, total hours of intervention per week were not associated with cognitive or adaptive outcomes, although hours were similar across intervention groups within each study (e.g., comparing half-day programs to other half-day programs).76-78, 81-83 In a retrospective cohort study,86 outcomes were related to age at enrollment, treatment duration, and higher baseline adaptive scores, with duration becoming nonsignificant after accounting for group membership (correlation of duration, group=.57, p<.01). A significant interaction emerged between age at enrollment and group membership, with younger starting age influencing outcomes for the treatment group but not control.86

In a study comparing 1:1 home-based ABA early intervention (both university-provided and privately-provided) to community-based treatment-as-usual, IQ remained stable for children in the community-based group and significantly declined for children who received university-provided ABA intervention (effect size=.49). This result is confounded by nonrandom assignment and the fact that at baseline, the university-based group had higher levels of ASD symptoms, lower levels of adaptive behavior, and fewer total intervention hours.79, 80 Finally, in a prospective cohort study, hours of intervention did not correlate with outcomes.77

KQ3. Treatment Phase Changes That Predict Outcomes

No studies were identified that provided data on changes early in treatment that predicted outcomes.

KQ4. Treatment Effects That Predict Long-Term Outcomes

Few studies assess end-of-treatment effects that may predict long-term outcomes. Several early intensive behavioral and developmental interventions change measures over the course of very lengthy treatments, but such outcomes usually have not been assessed beyond treatment windows. One family of studies140, 141, 148, 149 attempted to follow young children receiving early joint attention intervention until they were school-aged, but it failed to include adequate followup of the control group. It also involved children who were receiving many hours of uncontrolled interventions during the course of study.

KQ5. Generalization of Treatment Effects

Key Points

  • Some studies of imitation and joint attention reported generalization of skills, setting, and individual/provider from the treatment context to a novel context.

Overview of the Literature

Twelve studies (reported in multiple publications) reporting on different interventions measured generalization of effects seen in treatment. However, several studies incorporated parent- or teacher-delivered components, which may promote generalization of skills to the home and classroom.93, 101-104, 126, 129, 151-153, 155, 157, 158, 161, 166, 178, 196

Detailed Analysis

Few studies measured generalization of effects seen in treatment; however, several studies incorporated parent- or teacher-delivered components, which may promote generalization of skills to the home and classroom. Among play/interaction-focused studies, one study of imitation training reported that gains in elicited imitation skills in the treatment group were also reflected in improvements in motor imitation skills, suggesting transfer of skills learned in the intervention.152, 153 In a prospective cohort study assessing an intervention targeting pretend play, treatment group participants maintained their level of play dialog with novel toys when scripted dialog (a component of the initial intervention) was not provided.161 Four interventions targeting joint attention skills based in preschools reported generalization: in one, increases in joint attention initiations with preschool teachers generalized to longer duration of joint engagement with mothers (10% increase from baseline compared with 2% decrease for control group).155 Time jointly engaged with preschool teachers, however, did not increase. Two other studies151, 157 suggested that joint attention skills training transferred to the classroom with treatment group participants spending less unengaged time and/or initiating more gestures. In a final study, children receiving either a joint attention or symbolic play interventions were able to generalize increases in responding to joint attention to a novel individual.158

Studies of early intervention approaches reported greater socially engaged imitation that generalized across settings and context in the treatment group,103, 104 increased frequency of joint attention acts with an unfamiliar examiner,93 and maintenance of skills over time and in the home and center-based setting.101, 102 One study of a social skills intervention reported increases in participant social skills on intervention staff-rated but not parent-rated measures for either a Skillstreaming group or comparison group receiving a sociodramatic relational intervention.129 In another social skills study, parents of children in a program enhanced for children with high functioning ASD reported improvements in their children's skills in various settings while parents of children in a traditional social skills group did not.126 Finally, an analysis of Vineland and parental intrusiveness scores across income categories revealed no significant differences in one study of CBT, suggesting that the intervention is applicable across income levels.166, 178, 196

KQ6. Treatment Components That Drive Outcomes

We did not identify any studies meeting our inclusion criteria that addressed this question.

KQ7. Treatment Approaches for Children Under Age 2 at Risk for Diagnosis of ASD

Key Points

  • Mean ages in studies identified were all under three years, and all studies address interventions that can be used with children under age 2
  • Studies reported improvements in young children regardless of type of behavioral intervention

Overview of the Literature

This section presents the results of our literature search and findings regarding the use of treatment approaches in younger children who are at high risk of developing ASD based upon behavioral, medical, or genetic risk factors. In our 2011 review we identified two comparative studies (one good quality RCT73 and one fair quality nonrandomized clinical trial74) addressing interventions for very young children. For the current review, we identified three studies93, 97, 99 addressing treatment approaches for very young children. One crossover RCT was conducted in China (poor quality),99 one prospective cohort study in Europe (poor quality),97 and one RCT in the United States (fair quality).93

The mean age in most studies exceeded 24 months, although one93 included children under age two. Mean ages were all under three years, and all studies address interventions that can be used with children under age 2. The average age for diagnosis of ASD in the United States is not until at least age 3, but a reliable diagnosis can be made as early as age 2.

One fair quality RCT was completed in the clinic and home settings.93 Two poor quality studies, one crossover RCT and one prospective cohort study,97, 99 included groups receiving in-home parent training.

Detailed Analysis

A fair quality RCT focused on enhancing parental responsivity and child communication.93 It compared Hanen's More Than Words intervention to treatment-as-usual. The treatment group (n=29, mean age=21.11±2.71 months) received eight manualized group sessions with parents only and three in-home individualized parent-child sessions over a span of 3.5 months, whereas the control group (n=26, mean age=21.61±2.82 months) received no treatment or treatment as usual. There was no treatment effect on parental responsivity. The treatment group showed differential improvement on child communication depending on children's baseline object interest; children with lower levels of baseline object interest had greater growth in communication skills, whereas children with higher levels of object interest showed attenuated growth. Two poor quality studies compared parent training to lower intensity supportive interventions. Mean ages ranged from 25.33 to33.6 months. Both involved home visits and working with children and parents. The lower intensity treatment model, Autism-1–2–3, compared two groups that received the same series of ten thirty-minute child- and parent-training sessions, with one group having a lagged start date and serving as a control. It did not yield group differences on ASD symptoms, language skills, or parent stress scores.99 The higher intensity model, Keyhole, incorporated elements of Hanen's More than Words and the TEACCH programs.97 It compared 15–18 home visits over a 9 month period (n=35) targeting adaptive skills, ASD symptoms, and parent stress to a lower-intensity intervention model (n=26; 5 home visits, no additional services of supports). Compared with the control group, children in the treatment group showed improved adaptive, imitation, and communication skills, based only upon parent report. Mothers in the treatment group also reported improved health but did not report decreases in parenting stress.

In summary, young children who received behavioral interventions seemed to improve regardless of intervention type. It is important to note that none of the fair or better quality studies of young children compared children getting treatment to a no treatment control group. One poor quality study reported positive effects of treatment,97 but the level of intervention intensity varied significantly between groups, and it is unclear whether the effects were due to intensity versus the treatment type. Potential modifiers of treatment efficacy include baseline levels of object interest.93 Most outcome measures of adaptive functioning were based upon parent report, and the effect of parental perception of treatment efficacy on perception of child functioning was generally not explored.

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