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Background: Research trials of early intervention (EI) programs for children with autism spectrum disorder (ASD)
generally demonstrate medium-to-large gains, on average, compared with “treatment as usual,” in different
developmental domains. Almost all children with ASD receive their treatment through community-based services,
however, and studies suggest that evidence-based interventions rarely make their way into community practice.
Understanding the effectiveness of community-based EI and factors associated with these effects is the first step in
developing strategies for wide-scale implementation of effective EI. Methods: Studies of community-based EI for
children with ASD were identified through a systematic search. Changes in cognitive, communication, social, and
adaptive functioning from pre-treatment to post-treatment were assessed using standardized mean gain scores.
Effect sizes were estimated using random effects models. Moderators of interest included type of community EI
program, year of publication, intervention duration, and sample selection. Moderator effects were assessed using
analysis of variance of mixed-effects models and meta-regression analyses. Results: Forty-six groups from 33
studies met inclusion criteria (1,713 participants, mean age 37.4 months, 81.1% male). There were small but
statistically significant gains in each of the four domains. Hedges’s g ranged from 0.21 for adaptive behavior to 0.32
for communication outcomes, after removing outliers and correcting for publication bias. EI programs associated
with universities and hospitals were superior, on average, to other community EI programs for cognitive and adaptive
behavior outcomes. Intervention duration was negatively associated with effect sizes for communication and adaptive
behavior outcomes. Conclusions: These results indicate that there remains a large gap between outcomes observed
in community settings and those reported in efficacy trials. Keywords: Autism spectrum disorders; meta-analysis;
early intervention; community programmes.
2017), school-based providers do not typically incor- 7. Outcome measures included at least one of the following,
porate evidence-based practices with fidelity over the reported as standard scores or developmental quotients:
long-term (Cook, Landrum, Tankersley, & Kauffman,
a Cognitive: Early Learning Composite from the Mullen
2003; Mandell et al., 2013; Suhrheinrich et al.,
Scales of Early Learning (MSEL, Mullen (1995)) or Full
2013). In addition to regularly altering and modifying Scale IQ from a standardized cognitive assessment that
evidence-based practices to fit child needs and included both verbal and non-verbal IQ (e.g., Differential
resource constraints, providers also report frequently Abilities Scale (Elliott, 1990). Studies that only included
using practices that do not align with the evidence non-verbal IQ or a cognitive measure that only assessed
non-verbal IQ (e.g., Merrill-Palmer (Roid & Sampers,
base (Hess, Morrier, Heflin, & Ivey, 2008; Stahmer,
2004)) were excluded to minimize measurement differ-
Collings, & Palinkas, 2005). ences;
The gap between research and practice is well- b Communication: Vineland Adaptive Behavior Scales
documented in psychology, medicine, and special (VABS) Communication domain (Sparrow, Balla, & Cic-
education (Balas & Boren, 2000; Greenwood & chetti, 1985; Sparrow, Cicchetti, & Balla, 2005);
c Social: VABS Socialization domain (Sparrow et al., 1985,
Abbott, 2001; Williams & Beidas, 2018). To begin
2005);
to bridge this gap it is important to first understand d Adaptive Behavior: VABS Composite (Sparrow et al.,
the current state of the effectiveness of routine 1985, 2005); and
clinical care for young children with ASD. Many
8. Reported unadjusted pre- and post- intervention means
factors (e.g., clinical expertise, use of evidence-based and standard deviations for outcome measures (based on
practices, geographic location, intervention dose, recommendation from the What Works Clearinghouse
child characteristics) may be associated with com- (2014)).
munity outcomes. If research-based evidence is Studies only reporting follow-up data were excluded. For
making its way into routine practice for children studies with overlapping (or potentially overlapping) samples,
with ASD, improvements in community outcomes the study with the largest sample for each outcome was used.
over time would be expected. However, to date, no A systematic search of research databases was conducted
through January 2018 to identify relevant studies. PsycINFO
systematic review or meta-analysis has examined
and Medline databases were searched for terms related to
community outcomes for young children with ASD in autism and intervention (see Appendix S1 for a sample search
public programs and routine clinical care. The strategy). The reference list of retrieved articles, existing
present study leverages studies of community EI reviews, and meta-analyses were also examined for eligible
for ASD and control groups from trials of early studies.
Studies were first screened for eligibility based on title and
intervention, combining community groups across
abstract using these exclusion criteria: (a) did not include
studies to provide a rigorous assessment of out- children with ASD, (b) n ≤ 10 children with ASD, (c) not written
comes from community-based interventions and in English, (d) participants outside the age range, and (e) did
explore patterns in the results. not include a behavioral intervention. The first author and two
In the present study, we: (a) determine the overall reliable coders (85% agreement between coders and first
author) conducted screening and then full-text review for
effect sizes for community-based EI for ASD in four
eligibility. The first author completed final review based on
outcome domains, and (b) assess moderators of inclusion criteria (described above). See Figure 1 for the
these effects. PRISMA flow diagram (Liberati et al., 2009).
Study coding
Methods The first author extracted and coded data regarding partic-
Search procedures and selection of studies ipant, intervention, and study characteristics presented in
Tables S1 and S2. Interobserver agreement between the first
Studies were included that met these criteria: author and a reliable coder was calculated for 18 groups
1. Published, written in English; from nine studies. Percent agreement ranged from 83% to
2. Prospective, pre-test, post-test group design; 100% for all extracted and coded moderators, except for total
3. Presented outcomes for children with ASD separately; hours of intervention, which was 78%. EI programs were
4. Included more than 10 children with ASD receiving com- categorized as follows to reflect the range of services that
munity-based intervention; children experience in the community: (a) “Model” programs
5. Child age at study intake was <73 months, which corre- (MO): intervention in the community associated with univer-
sponds with the typical age of early intervention in the sities and/or hospitals (e.g., university laboratory schools)
United States; that offered routinely available clinical care, (b) “Specified
6. Provided information on outcomes of educational or treatment as usual” (SPC): treatment from a local school/
behavioral services available in the community or treat- agency or standard educational provisions that had defined
ment as usual (e.g., publicly funded services, routine procedures described in the article (including programs that
clinical care) for a defined group or subgroup of study reported using eclectic interventions), and (c) “Unspecified
participants. This included school, clinic, hospital, and treatment as usual” (USP): a wait-list group or services as
home-based interventions that were routinely available. usual in the community where participants received an
Groups that received intervention provided by researchers undefined variety of services. Study design was coded based
or groups where research funds were used to pay for on the following criteria: (a) “Single” (SI): reported outcomes
training to community providers were excluded as they from one community program, (b) “Comparison to other
did not reflect standard community practice and routinely community groups” (CC): compared community programs, (c)
available services; “Comparison to experimental group” (CE): the community
Identification
Records identified through Additional records identified
database searching through other sources
(n = 6194) (n = 25)
Studies included in
Included
quantitative synthesis
(meta-analysis)
(n = 33)
group was the control group for a researcher-implemented Potential outliers were detected using the sample-adjusted
treatment, and (d) “Other:” did not fit the other categories. meta-analytic deviancy (SAMD) statistic; including extreme
Treatment group allocation was coded based on the following studies may overestimate true variability (Huffcutt & Arthur,
criteria: (a) “Random assignment” (RA): participants assigned 1995). A conservative cutoff of |2.58| was used to exclude
to groups utilizing random assignment, (b) “Standard assign- groups from analyses (Beal, Corey, & Dunlap, 2002). SAMDs
ment” (SA): participants assigned to groups based on stan- were rank-ordered and scree plots were examined to confirm
dard community practice, (c) “Not applicable” (NA): included outlier status.
a single treatment group, and (d) “Other:” did not fit the Weighted mean effect sizes, heterogeneity, moderators, and
other categories. publication bias statistics were calculated using Comprehen-
sive Meta-Analysis Version 3.3.070 (Borenstein, Hedges, Hig-
gins, & Rothstein, 2014). Separate random effects models were
Analyses conducted for cognitive, communication, social, and adaptive
behavior outcomes, as recommended by Borenstein, Hedges,
All outcome data were continuous. Changes between baseline Higgins, and Rothstein (2009) and Lipsey and Wilson (2001).
and posttreatment assessments were evaluated using stan- Effect sizes were weighted to account for its relative precision
dardized mean gain scores. As shown in the equation below,
based on the standard error of the effect size and tau-squared
effect sizes were calculated by dividing the mean change from using the reciprocal of the squared standard error plus tau-
baseline to post-treatment by the pooled standard deviation of squared. Study quality was not used to weight effect sizes due
the difference score and transformed to Hedges’s g estimates
to inconsistent reporting.
(Hedges, 1981) to correct potential bias from small sample Heterogeneity of effect sizes was examined using the Q and I2
sizes: statistics. Exploratory moderator analyses were conducted for
models with a significant Q statistic or an I2 ≥ 50, indicating at
3
g ¼ 1 least moderate heterogeneity (Higgins, Thompson, Deeks, &
4ðn 1Þ
2 3 Altman, 2003). Categorical moderators were examined using
MPost MPre
6qffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffi 7 an analysis-of-variance (ANOVA) of mixed-effect models for
4 ffi pffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffiffi5 each variable hypothesized to moderate the overall effect size.
SDPre 2
þ SDPost 2
2rSDPre SDpost = ð2ð1 r ÞÞ Meta-regression analyses were used to examine continuous
moderators: age of the sample at intake, intervention duration,
No study reported the data needed to calculate pretest- approximate hours of intervention, and year of publication.
posttest correlations among outcome measures (r in the Year of publication was used as a proxy for recency of
equation above), therefore test-retest reliabilities from test intervention, as <50% of studies reported when the interven-
manuals and published papers were used as proxies (Lipsey tion occurred. Adjusted meta-regression was used to examine
and Wilson (2001). Because test-retest reliability may over- differences by EI category controlling for other covariates. Due
estimate pretest-posttest correlations, sensitivity analyses to the small number of studies, only statistically significant
with r values of 0.3 (low), 0.5 (medium), and 0.8 (high) were moderators were included in adjusted analysis.
conducted. The calculated effect sizes were similar, so the To assess publication bias, funnel plots and Duval and
test-retest reliabilities were used. Tweedie’s (2000) trim-and-fill procedure were calculated.
Model estimates were calculated using the trim-and-fill cor- groups (13.0%) from four studies reported track-
rection when this test indicated significant asymmetry in the ing fidelity to specific intervention practices.
funnel plot.
Categorical moderator
EI Category 13.94 (2) 2.98 (2) 2.48 (2) 13.48 (2)
MO 6 0.54 0.10 10 0.39 0.13 9 0.34 0.17 11 0.37 0.12
SPC 13 0.25 0.12 12 0.30 0.14 12 0.22 0.17 12 0.08 0.15
Allison S. Nahmias et al.
Continuous moderator
Age 0.00 0.01 0.51 (1) 0.00 0.01 0.63 (1) 0.00 0.01 0.05 (1) 0.00 0.01 0.13 (1)
Year 0.00 0.03 0.01 (1) 0.03 0.03 4.30 (1) 0.01 0.03 0.50 (1) 0.00 0.02 0.00 (1)
Duration 0.00 0.01 0.11 (1) 0.01 0.01 8.23 (1) 0.01 0.01 2.60 (1) 0.02 0.01 11.72 (1)
Hours 0.00 0.00 1.03 (1) 0.00 0.00 3.64 (1) 0.00 0.00 0.94 (1) 0.00 0.00 4.61 (1)
AU = Australia; CA = Canada; CC = group design comparing community EI programs; CE = group design comparing a community EI program to an experimental EI program; CI = 95%
Confidence Interval; CO = Country; Coeff = Coefficient; ES = Hedges’s g; IT = Italy; IS = Israel; MO = “model” EI program; NA = Not applicable (single treatment group); NO = Norway;
RA = random assignment to treatment group; SA = assignment to treatment group using standard community practice; SI = study of one community program; SPC = specified treatment
as usual program; SW = Sweden; TA = Taiwan; UK = United Kingdom; US = United States; USP = unspecified treatment as usual program.
Italic = significant at Bonferroni-corrected p < .05; Bold = significant at Bonferroni-corrected p < .01; Bold & Italic = significant at Bonferroni-corrected p < .001.
2014; Iverson et al., 2018; Lord, Risi, & Pickles, adaptive behavior, although not cognitive or social
2004), therefore the longer the duration of inter- outcomes.
vention, the greater the observed gap will be Several study limitations should be noted. We
between the scores of children with autism and conducted multiple comparisons to examine poten-
age-adjusted norms. It is also possible that children tial moderators and did not pre-register a protocol
may not make progress when receiving a long for the study, which can inflate the risk of type I
duration of the same ineffective intervention or the error. We have attempted to mitigate this risk by
parent-report instruments used to measure com- using Bonferroni-corrected p-values, but results
munication and adaptive behavior are not sensitive should be interpreted with caution. We were limited
to gains or to clinically important gains made in in the characteristics of the intervention models and
other domains more broadly. Regardless of the the participants that we could include in our anal-
reason for the negative associations, the results ysis. Communication, social, and adaptive behavior
highlight the importance of monitoring treatment outcomes were all measured using the same parent
progress using systematic assessment with mea- reported outcome measure (i.e., VABS) and parent
sures sensitive to intervention targets, so clinicians reported outcomes may be biased towards programs
can make appropriate clinical decision regarding that include parent training/model programs. We
changing treatment strategies or treatments if ben- also required standardized scores, which may have
efit is not observed (Kazdin, 2016; National resulted in important studies being excluded. The
Research Council, 2001). As one possibility, cur- restrictions on inclusion criteria may have con-
riculum-based assessments have been used to tributed to type II error. Additionally, we included
measure progress on proximal intervention goals only study design and group allocation methods as
and assist with treatment individualization and indicators of study quality. Other methodological
planning for toddlers with ASD (Bacon et al., 2014). factors (e.g., diagnostic processes; assessment pro-
Improved results were not observed in any out- cedures) may contribute to variable quality in these
come domain in more recent studies relative to older studies.
studies. Evidence-based practices may not be mak- Despite these limitations, these findings have
ing their way into standard community care or are important implications, especially if the small effects
not being implemented well in community settings. of community-based interventions resulted from
This finding also may result from restricted range; poor translation of research into practice. For exam-
most studies published prior to 2004 did not meet ple, community providers may lack high quality
our inclusion criteria. Year of publication may also training and supervision. Differences also could
have been a poor proxy for the year that data were result from fewer resources to implement complex,
collected (e.g., conducting secondary data analysis, resource intensive programs. Differences in charac-
publishing many years after the completion of treat- teristics of children and families between community
ment, see Table S2). settings and research trials also may lead to different
Programs associated with universities and hospi- outcomes. Lord et al. (2005) point out that in treat-
tals had significantly better cognitive and adaptive ment studies that report demographic characteris-
behavior outcomes than other community programs, tics of participants, the overwhelming majority are
suggesting that academic involvement may bolster white and of high socio-economic status. Families
effectiveness in these domains. We were limited in enrolling in studies may have more resources, fewer
our ability to investigate this question further, as obstacles, and more motivation/skill. Unlike
limited details regarding treatment strategies, fide- research trials, community sites often must accept
lity, or other potential metrics of quality were not all children without exclusion. Thus, community
consistently provided. However, these types of stud- programs have children that differ from those in
ies reflect an important step in studying treatments clinical trials in functioning, at-home support, family
in “progressively more genuine circumstances” resources, native language, and complex comorbidi-
(Chorpita, 2004; Southam-Gerow, Silverman, & ties. We largely did not see differences in the results
Kendall, 2006; Weisz, 2004). Public-academic col- based on study design and group allocation method,
laborations may offer an important step towards which suggests that the small effect sizes for com-
improving community practice. Academic involve- munity programs are more likely due to intervention
ment may lead to greater access to experts in ASD as opposed to participant characteristics.
treatment (Stahmer, Carter, Baker, & Miwa, 2003) Any of these reasons for the observed
which could increase use of evidence-based prac- differences point to the need for closer and
tices and access to supervision. This is a rich area for bidirectional partnerships between academic
further exploration. Other explanations are that and community sites, and the urgency of moving
these programs are more likely to measure an from tests of efficacy, or even effectiveness, to
outcome more proximal to their intervention target testing strategies that increase the successful
or to publish results because they have positive implementation of demonstrated-effective inter-
findings. In support of the latter hypothesis, publi- ventions for young children with autism in
cation bias was identified for communication and community settings.
Key points
Children with Autism Spectrum Disorder can make moderate to large gains in cognitive, communication,
social, and adaptive behavior domains when receiving effective early intervention (EI).
Less is known about the overall effectiveness of EI delivered in community settings.
Findings show that children with ASD make small gains when receiving community-based EI.
Model programs, associated with universities, are particularly promising for cognitive and adaptive behavior
outcomes.
Additional research is warranted to bring effective EI into standard community practice.
Higgins, J.P.T., Thompson, S.G., Deeks, J.J., & Altman, D.G. Reichow, B., Barton, E. E., Boyd, B. A., & Hume, K. (2012).
(2003). Measuring inconsistency in meta-analyses. BMJ, Early intensive behavioral intervention (EIBI) for young
327, 557–560. children with autism spectrum disorders (ASD). Cochrane
Huffcutt, A.I., & Arthur, W. (1995). Development of a new Database of Systematic Reviews https://doi.org/10.1002/
outlier statistic for meta-analytic data. Journal of Applied 14651858.cd009260.pub2
Psychology, 80, 327–334. Roid, G.H., & Sampers, J.L. (2004). Merrill-palmer-revised:
Iverson, J.M., Northrup, J.B., Leezenbaum, N.B., Parlad e, Scales of development. Wood Dale, IL: Stoelting.
M.V., Koterba, E.A., & West, K.L. (2018). Early gesture and Shire, Stephanie Y., Chang, Y.-C., Shih, W., Bracaglia, S.,
vocabulary development in infant siblings of children with Kodjoe, M., & Kasari, C. (2017). Hybrid implementation
autism spectrum disorder. Journal of Autism and Develop- model of community-partnered early intervention for tod-
mental Disorders, 48, 55–71. dlers with autism: A randomized trial. Journal of Child
Kasari, C., & Smith, T. (2016). Forest for the trees: Evidence- Psychology and Psychiatry, 58, 612–622.
based practices in ASD. Clinical Psychology: Science and Shire, Stephanie Yoshiko, & Kasari, C. (2014). Train the trainer
Practice, 23, 260–264. effectiveness trials of behavioral intervention for individuals
Kazdin, A.E. (2016). Closing the research–practice gap: How, with autism: A systematic review. American Journal on
why, and whether. Clinical Psychology: Science and Practice, Intellectual and Developmental Disabilities, 119, 436–451.
23, 201–206. Southam-Gerow, M.A., Silverman, W.K., & Kendall, P.C.
Liberati, A., Altman, D.G., Tetzlaff, J., Mulrow, C., Gøtzsche, (2006). Client similarities and differences in two childhood
P.C., Ioannidis, J.P.A., . . . & Moher, D. (2009). The PRISMA anxiety disorders research clinics. Journal of Clinical Child &
statement for reporting systematic reviews and meta-anal- Adolescent Psychology, 35, 528–538.
yses of studies that evaluate health care interventions: Sparrow, S.S., Balla, D.A., & Cicchetti, D.V. (1985). Vineland:
Explanation and elaboration. PLoS Medicine, 6, e1000100. Adaptive behavior scales: Interview edition: Survey form
Lipsey, M.W., & Wilson, D.B. (2001). Practical meta-analysis. manual. Circle Pines, MN: American Guidance Service.
In L. Bickman & D.J. Rog (Eds), Applied social research Sparrow, S.S., Cicchetti, D.V., & Balla, D.A. (2005). Vineland-
methods series: Vol. 49 (pp. 247). Thousand Oaks, CA: Sage II: Vineland adaptive behavior scales, survey forms manual.
Publications. Circle Pines, MN: American Guidance Service.
Lord, C., Risi, S., & Pickles, A. (2004). Trajectory of language Stahmer, A.C., & Aarons, G.A. (2009). Attitudes toward adop-
development in autistic spectrum disorders. In M.L. Rice & tion of evidence-based practices: A comparison of autism
S.F. Warren (Eds), Developmental language disorders: From early intervention providers and children’s mental health
phenotypes to etiologies (pp. 7–29). Mahwah, NJ: Lawrence providers. Psychological Services, 6, 223–234.
Erlbaum Associates Publishers. Stahmer, A., Carter, C., Baker, M., & Miwa, K. (2003). Parent
Lord, C., Wagner, A., Rogers, S., Szatmari, P., Aman, M., perspectives on their toddlers’ development: comparison of
Charman, T., . . . & Yoder, P. (2005). Challenges in evaluating regular and inclusion childcare. Early Child Development
psychosocial interventions for autistic spectrum disorders. and Care, 173, 477–488.
Journal of Autism and Developmental Disorders, 35, 695– Stahmer, A.C., Collings, N.M., & Palinkas, L.A. (2005). Early
708. intervention practices for children with autism: Descriptions
Magiati, I., Charman, T., & Howlin, P. (2007). A two-year from community providers. Focus on Autism and Other
prospective follow-up study of community-based early Developmental Disabilities, 20, 66–79.
intensive behavioural intervention and specialist nursery Suhrheinrich, J., Stahmer, A.C., Reed, S., Schreibman, L.,
provision for children with autism spectrum disorders. Reisinger, E., & Mandell, D. (2013). Implementation chal-
Journal of Child Psychology and Psychiatry, 48, 803–812. lenges in translating pivotal response training into commu-
Mandell, D. S., Stahmer, A. C., Shin, S., Xie, M., Reisinger, E., nity settings. Journal of Autism and Developmental
& Marcus, S. C. (2013). The role of treatment fidelity on Disorders, 43, 2970–2976.
outcomes during a randomized field trial of an autism Weisz, J.R. (2004). Treatment dissemination and evidence-
intervention. Autism: The International Journal of Research based practice: Strengthening intervention through clini-
and Practice, 17, 281–295. cian-researcher collaboration. Clinical Psychology: Science
Mullen, E.M. (1995). Mullen scales of early learning manual. and Practice, 11, 300–307.
Circle Pines, MN: American Guidance Service. What Works Clearinghouse. (2014). Procedures and standards
Murza, K.A., Schwartz, J.B., Hahs-Vaughn, D.L., & Nye, C. handbook version 3.0. Available from: https://ies.ed.gov/
(2016). Joint attention interventions for children with ncee/wwc/Handbooks [last accessed 19 November 2018].
autism spectrum disorder: A systematic review and meta- Whitehouse, A.J.O., Granich, J., Alvares, G., Busacca, M.,
analysis: Joint attention meta-analysis. International Jour- Cooper, M.N., Dass, A., . . . & Anderson, A. (2017). A
nal of Language & Communication Disorders, 51, 236–251. randomised controlled trial of an iPad-based application to
Nevill, R.E., Lecavalier, L., & Stratis, E.A. (2018). Meta- complement early behavioural intervention in Autism Spec-
analysis of parent-mediated interventions for young children trum Disorder. Journal of Child Psychology and Psychiatry,
with autism spectrum disorder. Autism, 22, 84–98. 58, 1042–1052.
Ospina, M.B., Krebs Seida, J., Clark, B., Karkhaneh, M., Williams, N.J., & Beidas, R.S. (2018). Annual Research Review:
Hartling, L., Tjosvold, L., . . . & Smith, V. (2008). Behavioural The state of implementation science in child psychology and
and Developmental Interventions for Autism Spectrum psychiatry: A review and suggestions to advance the field.
Disorder: A Clinical Systematic Review. PLoS ONE, 3, e3755. Journal of Child Psychology and Psychiatry, 60, 430–450.
Reichow, B. (2012). Overview of meta-analyses on early inten-
sive behavioral intervention for young children with autism Accepted for publication: 15 April 2019
spectrum disorders. Journal of Autism and Developmental
Disorders, 42, 512–520.