Autism spectrum disorder

Vol.:(0123456789)1 3

J Autism Dev Disord DOI 10.1007/s10803-017-3339-2

ORIGINAL PAPER

Teaching Parents Behavioral Strategies for Autism Spectrum Disorder (ASD): Effects on Stress, Strain, and Competence

Suzannah Iadarola1,7  · Lynne Levato1 · Bryan Harrison1 · Tristram Smith1 · Luc Lecavalier2 · Cynthia Johnson3 · Naomi Swiezy4 · Karen Bearss5 · Lawrence Scahill6 

© Springer Science+Business Media, LLC 2017

parental competence while reducing parental stress and parental strain.

Keywords Autism spectrum disorder · Parent training · Parental stress · Parental competence

Introduction

Parents of young children with ASD face many challenges. Children with ASD often require specialized care coordi- nation across several providers and multiple meetings on school placement. Parents may become isolated from friends and family who may not understand the child’s behavior and disability (Abbeduto et  al. 2004; Kogan et  al. 2008; Rao and Beidel 2009). These parental challenges may be influenced by the child’s age, timing of diagnosis, and level

Abstract We report on parent outcomes from a rand- omized clinical trial of parent training (PT) versus psychoe- ducation (PEP) in 180 children with autism spectrum disor- der (ASD) and disruptive behavior. We compare the impact of PT and PEP on parent outcomes: Parenting Stress Index (PSI), Parent Sense of Competence (PSOC), and Caregiver Strain Questionnaire (CGSQ). Mixed-effects linear models evaluated differences at weeks 12 and 24, controlling for baseline scores. Parents in PT reported greater improvement than PEP on the PSOC (ES = 0.34), CGSQ (ES = 0.50), and difficult child subdomain of the PSI (ES = 0.44). This is the largest trial assessing PT in ASD on parent outcomes. PT reduces disruptive behavior in children, and improves

Electronic supplementary material The online version of this article (doi:10.1007/s10803-017-3339-2) contains supplementary material, which is available to authorized users.

* Suzannah Iadarola suzannah_iadarola@urmc.rochester.edu

Lynne Levato lynne_levato@urmc.rochester.edu

Bryan Harrison bryan_harrison@urmc.rochester.edu

Tristram Smith Tristram_smith@urmc.rochester.edu

Luc Lecavalier luc.lecavalier@osumc.edu

Cynthia Johnson Cynthia.johnson@chp.edu

Naomi Swiezy nswiezy@iupui.edu

Karen Bearss kbearss@u.washington.edu

Lawrence Scahill Lawrence.scahill@emory.edu

1 University of Rochester Medical Center, Rochester, NY, USA

2 Ohio State University, Columbus, OH, USA 3 University of Florida, Gainesville, FL, USA 4 Indiana University, Indianapolis, IN, USA 5 University of Washington, Seattle, WA, USA 6 Emory University, Atlanta, GA, USA 7 Department of Pediatrics, University of Rochester Medical

Center, 601 Elmwood Avenue, Box 671, Rochester, NY 14642, USA

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of symptom severity (Greenberg et al. 2006; Hastings et al. 2005). Disruptive behaviors, including tantrums, noncompli- ance, aggression, and self-injury are common, affecting as many as 50% of children with ASD. These behaviors in the child may amplify caregiving burden (Hastings et al. 2005) and contribute to parental stress and strain (Boström et al. 2011; Hsiao 2016; Vasilopoulou and Nisbet 2016). Disrup- tive behavior in the child may also erode parental compe- tence, perceived self-efficacy, and problem-solving skills (Benson 2014; Falk et al. 2014; Pottie and Ingram 2008; Rezendes and Scarpa 2011). Interventions that reduce dis- ruptive behavior may also reduce parental stress and strain. In addition, the association between child behavior and parenting stress may be bidirectional, with stress reducing parents’ ability to address disruptive behavior (Greenberg et al. 2006).

Several parent training (PT) interventions have been developed (Smith and Iadarola 2015) to teach new skills, address skill deficits, or decrease disruptive behavior in children with ASD (Bearss et al. 2015; Strauss et al. 2012; Tonge et al. 2014). Studies on PT for children with ASD and disruptive behavior have shown decreases in child dis- ruptive behavior (Postorino et al. 2017). Few studies have reported on parental distress and related outcomes (e.g., self- efficacy). Findings on parental outcomes in parent training studies are inconclusive due to study design limitations or small sample size (Coolican et al. 2010; Tonge et al. 2006; Whittingham et al. 2009). Parent variables that moderate or mediate the effect of PT on child disruptive behavior or parental stress have not been examined (Smith and Iadarola 2015). Observational, cross-sectional studies suggest that parental cognitions such as perceived efficacy (or related cognitions such as perceived locus of control, perceived competence, engagement, and problem-solving) are associ- ated with lower parenting stress (Benson 2014; Falk et al. 2014; Pottie and Ingram 2008; Rezendes and Scarpa 2011).

Although PT is a well-established intervention for dis- ruptive behavior in non-ASD pediatric populations (Dretzke et al. 2009), findings on parental outcomes are also limited and equivocal. Common parent outcomes of interest include perceived parenting ability and satisfaction (often referred to as parental self-efficacy or parental competence) as well as stress and internalizing symptoms (i.e., anxiety, depres- sion, somatization). A meta-analysis reported medium effect sizes (0.42–0.53) of PT on measures of parental stress and competence (Lundahl et al. 2006). Associations between parenting programs for disruptive behavior and reduced stress, decreased depression, and increased locus of control (Chacko et al. 2009; Danforth et al. 2006; Moreland et al. 2016) have also been documented. However, in two large- scale studies of children with ADHD, parents reported no reduction in stress after PT (Abikoff et al. 2007; Wells et al. 2000). Thus, although PT provides specific tools to help

parents manage disruptive behavior, it is not clear that PT reduces parental stress and strain. Indeed, the task demands of applying PT could contribute to parental stress (Karst and Van Hecke 2012). It may be expected that parents who learn and practice parent training techniques will achieve a greater sense of competence. Whether increases in competence will contribute to decreased parental stress and strain is not clear.

In prior work, we built a structured PT manual that inte- grated behavior change strategies developed and tested in single subject studies for disruptive behavior in children with ASD (Johnson et al. 2007). We conducted a series of studies showing that this program is acceptable to parents, can be delivered with fidelity by trained therapists, and can augment the therapeutic effects of medication (Aman et al. 2009; Bearss et al. 2013). In a rigorous test of PT, we con- ducted a six-site, 24-week randomized controlled trial (RCT) in 180 children with ASD and disruptive behavior, aged 3–7 (Bearss et al. 2015; Lecavalier et al. 2017; Scahill et al. 2016). Children were randomized to an 11-session, struc- tured PT program or a 12-session parent education program (PEP) that controlled for time and attention. PT was superior to PEP in reducing parent-rated child disruptive behavior. The positive response rate on the Clinical Global Impres- sion Improvement Score completed by clinicians who were blind to treatment assignment was 68.5% in PT compared to 39.6% in PEP (39.6%). PT was also superior to PEP on a standardized measure of child daily living skills (Scahill et al. 2016). In a follow-up paper we identified that modera- tors of positive child outcomes in PT versus PEP included lower ADHD and anxiety symptoms and higher household income (Lecavalier et al. 2017).

The current report focuses on parent outcomes in this RCT. Our primary hypothesis was that self-reported paren- tal competence would show significant improvement and whether self-reported measures of parental stress would show significant decreases in PT compared to PEP. Based upon previous findings that parental cognitions may influ- ence parental stress (Falk et  al. 2014), we also explored whether change in parental cognitions (i.e., competence) would predict improvement in parental stress and caregiver strain, as well as child disruptive behavior. The study design allowed examination of these questions because it included multiple parent self-report measures collected at midpoint (week 12) and endpoint (week 24).

Methods

Design

The original RCT was conducted at six sites (Emory Univer- sity, Indiana University, Ohio State University, University of Pittsburgh, University of Rochester, and Yale University) from

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September, 2010 to February, 2014. The trial was approved by the institutional review boards at each site, and informed consent was obtained from a parent or legal guardian. Partici- pants who met eligibility criteria were randomly assigned to 24 weeks of PT or PEP using permutated blocks with concealed allocation to investigators. Randomization was performed within site and further stratified by educational intensity to ensure that groups contained an equal number of participants in high intensity school/therapeutic programming. High inten- sity service was defined as 15 h or more per week of 1:1 or 1:2 specialized instruction for ASD. Therapists held a minimum of a master’s degree and had been certified to deliver each study intervention after completing training and demonstrating fidel- ity (> 80% correct implementation of content in all sessions, rated by a senior investigator). Therapists received weekly local supervision and monthly during cross-site case reviews. Parent ratings on child disruptive behavior were completed every 4 weeks and every 12 weeks for parent measures. The measures included several parent-report questionnaires. The background, methods, and main child outcomes are reported in detail in Bearss et al. (2015).

Participants

One-hundred-eighty children with ASD and moderate or greater disruptive behavior between the ages of 3 and 7 years inclusive participated in the 24-week study. One parent from each household was enrolled in PT or PEP and was the informant on all outcome measures. Other parents and family members with caregiving responsibilities were invited to join therapy sessions. Eligibility required: an ASD diagnosis, a score ≥ 15 on the Irritability subscale of the Aberrant Behavior Checklist (described below), and a CGI Severity (CGI-S) score ≥ 4. Additional interventions and medications were required to be stable for 6 weeks, with no planned changes for the course of the study. Children with serious medical conditions or another psychiatric disorder in need of treatment, receptive language skills ≤ 18 months (as determined by standardized cognitive and developmen- tal assessments), or those with current or past treatment in structured PT for disruptive behavior were excluded. Clini- cal diagnosis of ASD was based on DSM-IV-TR criteria (American Psychiatric Association 2000) corroborated by the Autism Diagnostic Interview-Revised (ADI-R; Rutter et al. 2003) and the Autism Diagnostic Observation Sched- ule (ADOS; Lord et al. 2002).

Measures

Characterization Measures

The Autism Diagnostic Observation Schedule (ADOS) is an investigator-based assessment conducted in naturalistic

social situations demanding specific social, communication and restricted/repetitive responses. Behaviors are scored in the areas of social communication, social relatedness, play and imagination, and repetitive behaviors. An ADOS score above the cutoff for either autism or autism spectrum disor- der was used to support the diagnosis of ASD.

The Autism Diagnostic Interview, Revised (ADI-R), is a structured parent interview that is designed to obtain rel- evant information about a child’s early communication and language development, social development and play, and unusual interests and behaviors. The ADI-R was conducted with parents to corroborate the information collected dur- ing the ADOS and was also used to confirm diagnosis for eligibility.

Developmental/Cognitive Functioning The Stanford- Binet Fifth Edition (SB-V; Roid 2003) or the Mullen Scales of Early Learning (MSEL; Mullen 1995) were used to assess cognitive functioning. The abbreviated SB-V was attempted with all children. The Mullen was administered to children who were unable to the abbreviated SB-V. Standard scores obtained from these measures were used to determine eli- gibility and to characterize the cognitive functioning of the sample.

Outcome Measures

Parenting Stress Index-Short Form (PSI; Abidin 1995) The PSI is a 36-item measure completed by parents of children 3  months to 10  years of age designed to assess parental stress. Each item is rated on a 5-point scale (from “Strongly Disagree” = 0 to “Strongly Agree” = 5). The PSI yields a total stress score and subscale scores across three factors: parental distress, parent–child dysfunctional interaction, and difficult child characteristics. Example statements include, “I feel trapped by my responsibilities as a parent,” “Sometimes I feel my child doesn’t like me and doesn’t want to be close to me,” and “I feel that my child is very moody and easily upset.” The PSI has good test–retest reliability (ICC = 0.77) and internal consistency (IC; 0.91 (Barroso et al. 2016)). A PSI total score of ≥ 88 (85th percentile) is considered clinically significant. This measure was used to differentiate among subtypes of stress, including stress related to child behavior and interactions, as well as stress related to the parents’ internal emotional state.

Caregiver Strain Questionnaire (CGSQ; Brannan et al. 1997) The CGSQ is a 21-item, parent self-report on the burdens associated with raising a child with ASD and perceived interference with family activities. Parents rate caregiver strain on items such as “Interruption of personal time,” “Financial strain,” and “Feeling sad or unhappy” on a 1–5 scale (“Not a problem” to “Very much a problem”). This measure yields objective strain, internalized strain, and externalized strain subscales and a global score. The CGSQ

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demonstrates acceptable-to-high internal consistency for objective strain (0.91), externalized strain (0.74), internal- ized strain (0.86), and the global score (0.93). The subscales are negatively correlated with established measures of fam- ily functioning, such as the Family Assessment Device. The original sample from Brannan et al. (1997) included parents of children with unspecified emotional/behavioral disorders and reported mean scores of 2.0 (objective strain), 3.4 (inter- nalized strain) and 2.3 (externalized strain).

Parenting Sense of Competence (PSOC) The PSOC (Gibaud-Wallston and Wandersman 1978) is a 17-item, parent self-report reflecting parental satisfaction and effi- cacy. The satisfaction subscale measures parental motivation and frustration (e.g., “Even though being a parent could be rewarding, I am frustrated now while my child is at her/ her present age”). The efficacy subscale measures perceived capacity to change the child’s behavior (e.g., “I meet my own personal expectations for expertise in caring for my child”). The PSOC also yields a Total Competence score, with higher scores reflecting higher competence. In a norma- tive community sample of mothers (Gilmore and Cuskelly 2009), subscale mean scores of Satisfaction (22.72) and Effi- cacy (22.03) were reported.

Aberrant Behavior Checklist (ABC; Aman et al. 1985) is a reliable and valid 58-item parent-rated scale with dem- onstrated sensitivity to change (Kaat et al. 2014). Each item is rated from 0 (not a problem) to 3 (severe in degree). The ABC contains five subscales: Irritability (15 items), Social Withdrawal (16 items), Stereotypic Behavior (7 items), Hyperactivity/Noncompliance (16 items), and Inappropri- ate Speech (4 items). It was completed at baseline and every 4 weeks thereafter. For the current analysis, we used only the Irritability subscale, which was the primary outcome measure in the RCT.

Treatments

Parent Training (PT)

PT consisted of 11 core sessions of 60–90 min delivered individually that included direct instruction, video vignettes, practice examples, and role playing between parent and ther- apist. Weekly homework assignments gave the participat- ing parent opportunities to practice the strategies learned in session with the child in natural settings. The program included two home visits and up to two optional sessions. Sessions were conducted over 16 weeks. Therapists followed a treatment manual that included scripts and suggestions for engaging the family. Fidelity to the manual was assessed with a session-specific checklist of the required elements of the session. The PT intervention instructed parents on the application of behavioral strategies to manage behavio- ral problems in the home and community. Session content

focused on the situations and events that preceded disruptive behavior (antecedents) and the environmental responses that reinforced the behavior. Briefly, the PT program included use of visual daily schedules, positive reinforcement, planned ignoring as well as techniques to promote compli- ance and daily living skills.

Psychoeducation Program (PEP)

PEP was an active condition to control for time and therapist attention. It was also a structured intervention consisting of 12 individually-delivered sessions and one home visit. The manual covered useful topics for parents of young children with ASD, including etiology of ASD, educational plan- ning, advocacy, and information on how to select effective treatments. As with PT, PEP was delivered over 16 weeks and included regular fidelity checks. Unlike PT, PEP did not include any direct instruction in behavioral management. Each session comprised didactics, discussion, and informa- tional handouts at the end of each visit (see Bearss et al. 2015 for more detailed information about PT and PEP).

Statistical Analysis

Treatment Effects

Mixed-effects linear regression models were used to evaluate within-group changes over time from baseline to week 24 (endpoint) and between-group differences at week 12 (mid- point) and week 24 for total scores on the PSI, CGSQ, and PSOC. Exploratory analyses examined subscales on each measure. Fixed effects included treatment group, time, site, intervention intensity (i.e., whether the child was receiv- ing more or less than 15 h of direct, individual service per week), and time-by-treatment. Within-group effects were ascertained by regressing the PSI, CGSQ, and PSOC scores against time. For between-group effects, the average slopes of the regression lines were compared (PT versus PEP). Effect sizes were calculated on each measure by taking the difference in the least squares means at weeks 12 and 24 and dividing by the standard deviation at baseline for the entire sample. We assumed that missing data were missing at random (Little 1988).

Exploratory Analyses

Structural equation modeling (SEM) was employed to evalu- ate the relationships between change in PSOC in the first 12 weeks and change in the PSI, CGSQ and ABC Irritability subscale in the next 12 weeks. SEM permits examination of the model while simultaneously controlling for shared variance across measures and informants at each assessment wave. Within the SEM framework, latent difference scores

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(LDS; McArdle 2009) were calculated to model true change in predictors and outcomes over time. The LDS scores gen- erated for the PSOC (the predictor of change from baseline to week 12) and outcomes (change in PSI, CGSQ and ABC-I from week 12 to week 24). This method accounted for base- line scores on each variable.

Models (see Fig.  1) examined (a) the extent to which change in parental competence (PSOC) predicted change in outcome measures (PSI, CGSQ, and ABC-I) and (b) whether this predictive relationship significantly differed across the two treatment groups (PT and PEP). (McArdle 2009). Structural equation models were estimated using the Amos 18.0 software system (Arbuckle 2006). To maximize statistical power, we used full-information maximum likeli- hood (FIML) in AMOS and included the full sample in the analyses (Enders 2001). The model adequately represented the data, χ2 (2) = 3.53, p = .17, RMSEA = 0.07. To test for treatment-related differences, AMOS’s critical ratio (CR) of Differences was used. Pairwise parameter comparisons cal- culated the difference between the two estimates divided by the estimated standard error of the difference. The resulting difference statistic is normally distributed and tested against the z-score distribution (CR > 1.96). Therefore, the CR pro- vides an explicit test of the modifying effect of treatment group.

Results

Parent respondents were primarily female (93%), in their mid-30s (see Table 1). Children were 79% male, with a mean age of 4.2 years (SD = 1.1), and 74% had an IQ of 70 or above on the SB-V.

Treatment Effects

Table 2 presents data on parent outcome measures at base- line and week 24 within each treatment group. Baseline

scores on the PSI were elevated. On the CGSQ, scores were similar or elevated compared to the sample described in Brannan et al. (1997). Scores on the PSOC were comparable or higher than those reported in community samples. Over time, both PT and PEP showed improvements in the PSI, CGSQ, and PSOC. On the PSI total score, PT showed a 14% reduction, and PEP showed 9.3% reduction. On the CGSQ global score, PT showed 17.2% reduction, and PEP showed 7.1% reduction. For PSOC total score, PT showed 16.4% increase, and PEP showed 7.4% increase. See Supplemental Materials for figures of the total score and subscale scores for the PSI, CGSQ, and PSOC.

Table 3 shows differences in least squared means from baseline to weeks 12 and 24 between PT and PEP. PT did not show a significant advantage over PEP on the PSI total score, the parent–child interaction or the child distress scales at week 12. On the PSI difficult child factor, however, PT produced greater reductions than PEP at week 12 and week 24. The reduction in the PSI total score was greater in PT than PEP at week 24, but the difference was not significant. At week 12 and week 24, PT was superior to PEP on the CGSQ global score and Internalized subscale. There was no difference in CGSQ Externalized or objective strain sub- scales at week 12. The CGSQ Objective subscale reached significance at week 24. On the PSOC, parents in the PT group reported greater gains than parents in PEP at week 12 on the satisfaction subscale but not the efficacy subscale or total score. Improvement was significantly greater in PT compared to PEP on the PSOC total score and the efficacy subscale at week 24. The difference on the satisfaction sub- scale was no longer significant.

Exploratory Analyses

The LDS models confirmed findings from the mixed-effects linear models that parents in both treatment groups showed significant change in competence from baseline to week 12 (PT: β = .68, p < .001; PEP: β = .49, p < .001). Pairwise

Fig. 1 Model predicting change in parental stress and strain and child irritability, as predicted by change in parental competence

Change in Parent

Competence

Change in Parent

Stress/ Strain and Child Irritability

Competence at Baseline

Competence at Week 12

Treatment Group (PT

versus PEP)

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parameter comparisons indicated that parents in the PT group reported significantly greater gains on the PSOC than the PEP group during the first 12 weeks of the interven- tion (z = 2.72, p < .01). Parents in both groups also reported significant decrease in stress (PT: β = −0.38, p = .009; PEP: β = −0.39, p = .006) and strain (PT: β = −0.50, p < .001; PEP: β = −0.45, p < .001) from week 12 to week 24. However,

change in stress and strain from week 12 to 24 did not sig- nificantly differ across groups (z = 1.15 and z = 0.74, respec- tively). Similarly, parents in both groups reported significant change in child disruptive behavior on the ABC-I from week 12 to week 24 (PT: β −0.54; PEP: β −0.50, p < .001), and there was no difference across treatment groups (z = 0.983).

This exploratory analysis examined whether change in competence predicted change in stress, strain, or child irri- tability, and whether treatment groups differed. The results revealed that change in competence did not significantly pre- dict change in PSI total score, CGSQ global score, or child disruptive behavior (ABC-I). Furthermore, the magnitude of this relationship did not significantly vary across groups.

Discussion

To our knowledge, this is the largest randomized controlled study to date of PT in children with ASD and disruptive behavior. Here we examined the impact of PT on multiple parent outcomes, including parental stress, caregiver strain, and parental competence. The improvements in parent self-reports in both groups suggest non-specific treatment effects (e.g., therapist attention). However, there may also be unique effects for each intervention: increased proficiency in behavioral strategies in PT and increased knowledge about ASD in PEP. Still, the larger improvements for PT suggest that addressing child behavior was an especially effective intervention component. Although both groups improved, PT showed greater increase on perceived parental compe- tence than PEP. Compared to PEP, PT also showed greater reduction in several indices of parental strain and stress due to the challenges of raising a child with ASD and disruptive behavior. Effect sizes ranged from small to medium. Positive effects for difficult child behavior, global caregiver strain, and satisfaction with parental competence were evident at week 12, suggesting that change in parent self-reported out- comes occurred during the first half of treatment. This is consistent with the differential improvements between treat- ment groups that also emerged for child outcomes at week 12 (Bearss et al. 2015). Other dimensions, such as parental efficacy, overall parental competence, and externalized car- egiver strain, required the full 24 weeks to show differential change. These findings contribute to the growing evidence for PT in ASD and lend support to the broader finding that PT reduces disruptive behavior in many different child popu- lations (Dretzke et al. 2009; Postorino et al. 2017; Skotarc- zak and Lee 2015).

The PSI difficult child subscale, which includes disruptive behavior problems, was significantly different between treat- ment groups at week 24. Given that disruptive behavior is the target of PT, this finding is not surprising. On the PSI total score and other PSI factors (parental distress, parent–child

Table 1 Participant characteristics

No. (%)

Parent training (n = 89)

Parent education (n = 91)

Study center  Emory/Yale University 17 (19.1) 18 (19.8)  Indiana University 14 (15.7) 14 (15.4)  Ohio State University 19 (21.4) 20 (22.0)  University of Pittsburgh 19 (21.4) 18 (19.8)  University of Rochester 20 (22.5) 21 (23.1)

Parent demographics  Gender of primary informant   Female 79 (88.7) 87 (95.6)   Male 10 (11.3) 4 (4.4)

 Mother age (years) 35.4 (6.6) 35.9 (6.1)  Father age (years) 38.4 (7.7) 38.5 (7.2)  2-parent family 77 (86.5) 81 (89.0)  Education   Some high school 1 (1.1) 0   High school degree 9 (10.1) 5 (5.5)   Some college 28 (31.5) 26 (28.6)   College diploma 22 (24.7) 37 (40.7)   Advanced degree 29 (32.6) 23 (25.3)

 Family income   <$20,000 8 (9.0) 7 (7.7)   $20,001–$40,000 19 (21.3) 17 (18.7)   $40,001–$60,000 17 (19.1) 19 (20.9)   $60,001–$90,000 15 (16.9) 21 (23.1)   >$90,000 29 (32.6) 27 (29.7)

Child demographics  Gender   Female 10 (11.2) 12 (13.2)   Male 79 (88.8) 79 (86.8)

 Age (years) 4.8 (1.2) 4.7 (1.1)  IQ   < 70 13 (14.6) 16 (17.6)   ≥ 70 66 (74.2) 68 (74.7)   Missing 10 (11.2) 7 (7.7)

 Ethnicity   Black/African-American 9 (10.1) 6 (6.6)   Asian/Pacific Islander 2 (2.3) 6 (6.6)   White/Caucasian 78 (87.6) 78 (85.7)   Other 0 1 (1.10)

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dysfunctional interaction), both groups improved and there were no significant differences between groups. The greater improvements in parental competence and parental stress for PT over PEP suggest that decreases in child disruptive behavior are associated with positive parent outcomes.

Although the PEP group improved over time, PT was superior to PEP on measures of difficult child behavior, car- egiver strain, and parent perceived competence at week 24. The finding that psychoeducation programs, such as PEP, can improve child outcomes (Bearss et al. 2015; Kasari et al. 2015) suggests that a better understanding of the behavior in children with ASD may promote reductions in parental stress and strain, although this implication cannot conclusively be drawn from the data. However, our results suggest that

providing parents with specific tools to reduce disruptive behavior reduces parental stress and strain, and improves parental competence. Furthermore, the task demands of applying parent training did not appear to contribute to parental stress.

The finding that a decrease in child disruptive behavior via PT promotes improvement in parental competence, are consistent with previous research in non-ASD populations (Dretzke et al. 2009). We used structural equation models (SEM) to explore the predictive role of improved parent competence in PT on parent and child outcomes. Our pre- diction that improved parental competence would predict subsequent reduction in parental stress and strain as well as disruptive behavior in the child in PT versus PEP was

Table 2 Parenting stress, parent competence, and caregiver strain raw scores by group and timepoint

Parent training (PT) (n = 89)

Parent education (PEP) (n = 91)

Baseline Week 12 Week 24 Baseline Week 12 Week 24

Parenting Stress Index-short form total 106.2 (19.0) 96.5 (20.7) 91.4 (19.6) 103.5 (17.9) 96.9 (17.6) 93.9 (19.4)  Parental distress 33.3 (8.6) 30.7 (8.8) 28.9 (8.1) 32.7 (8.8) 29.9 (8.1) 28.7 (9.6)  Parent/child difficult interaction 29.1 (7.3) 26.8 (7.4) 25.4 (7.0) 28.6 (7.7) 26.8 (7.0) 25.7 (6.8)  Difficult child 43.6 (6.9) 38.9 (8.4) 37.0 (8.1) 42.2 (6.8) 40.1 (7.2) 39.1 (8.0)

Caregiver strain questionnaire global 2.9 (0.6) 2.5 (0.6) 2.4 (0.6) 2.8 (0.6) 2.5 (0.6) 2.6 (0.6)  Objective strain 2.9 (0.7) 2.5 (0.7) 2.4 (0.7) 2.8 (0.7) 2.5 (0.7) 2.6 (0.7)  Internalized strain 3.4 (0.7) 2.8 (0.8) 2.6 (0.8) 3.3 (0.7) 2.9 (0.74) 2.9 (0.9)  Externalized strain 2.2 (0.6) 2.0 (0.5) 1.9 (0.49) 2.0 (0.6) 2.0 (0.6) 2.0 (0.6)

Parenting sense of competence total 61.4 (11.8) 68.3 (10.4) 71.5 (9.7) 63.5 (10.7) 66.9 (11.8) 68.2 (12.2)  Satisfaction 35.2 (7.4) 38.7 (6.8) 40.1 (6.1) 36.7 (6.6) 38.1 (7.8) 39.2 (7.5)  Efficacy 26.2 (6.6) 29.6 (5.4) 31.4 (5.3) 26.8 (5.9) 28.7 (5.9) 29.0 (6.2)

Aberrant behavior checklist—irritability 23.7 (6.4) 16.1 (7.3) 11.9 (6.5) 23.9 (6.2) 18.7 (7.4) 16.6 (7.6)

Table 3 Differences between parent training and parent education program in least square means, p values, and standardized effect sizes for change from baseline at 12 and 24 weeks

Effect sizes represented as Cohen’s d LSM-Diff difference in least square means (PT-PEP) *All trends show an advantage for PT over PEP

Change from baseline to week 12*

Change from baseline to week 24*

LSM-Diff p Effect size LSM-Diff p Effect size

Parenting Stress Index-short form total −3.50 .15 0.19 − 4.51 .07 0.25  Parental distress − 0.21 .83 0.02 − 0.52 .63 0.06  Parent/child difficult Interaction − 0.41 .65 0.05 − 0.52 .56 0.07  Difficult child −2.75 .008 0.40 −3.02 .004 0.44

Caregiver strain questionnaire global − 0.16 .05 0.32 − 0.29 < 0.001 0.50  Objective strain − 0.14 .14 0.20 − 0.28 .005 0.40  Internalized strain − 0.27 .01 0.35 − 0.38 < 0.001 0.49  Externalized strain − 0.01 .89 0.90 − 0.14 .08 0.23

Parenting sense of competence total 2.41 .07 0.21 3.76 .01 0.34  Satisfaction 1.41 .04 0.20 1.32 .08 0.19  Efficacy 0.97 .18 0.15 2.40 .001 0.38

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not confirmed. We observed significant increases on the PSOC in the PT group from baseline to week 12, but this did not significantly predict more change on the PSI total score, CGSQ global score or the ABC-I from week 12 to week 24. A limitation of this analysis is that, although SEM controls for shared variance across measures, these measures are not entirely separate constructs and were highly inter-correlated in our sample, reducing our ability to separate change in competence from change in stress and strain.

The positive child and parent outcomes demonstrated in this trial add to the empirical support for PT and suggest that PT is ready for wider application for young children with ASD and disruptive behavior. Challenges ahead include identification of barriers that may hinder the broader appli- cation of PT in community settings. One obvious challenge is training a wide range of practitioners (e.g., psychologists, special educators, social workers and child psychiatric nurse practitioners), which would require institutional commitment to provide space and funding. Evaluation of ancillary effects on families (e.g., parenting styles, sibling behavior), child behavior in other settings (e.g., classrooms), and longer- term outcomes could help indicate whether PT has broader, clinically significant effects. Given the improvements in both PT and PEP, a blended intervention that includes content from both approaches may yield additive effects, but would also increase the number of treatment sessions. Alternative approaches such as group PT or PT by telehealth also war- rant further development.

The present findings should be considered in light of several limitations. Primary outcomes on child behavior and parent outcomes are based on parent report. We did not measure objective outcomes, such as physiological markers of parental stress. However, parental perception of stress and caregiver strain is testimony from parents directly facing the challenges of raising a child with ASD and disruptive behavior. We also note that parents were not blind to group assignment. Perceptions about the two treatments may have influenced their ratings. To date, few studies with other measures of parental stress and well- ness (e.g., physiological recordings) have been reported in PT studies, and this is an area in need of future devel- opment. We also note the use of multiple comparisons, which inflated the probability of Type I error. Finally, the parents who participated in this study were mostly white, middle and upper-middle class, and were well-educated. Accordingly, our findings may not necessarily extend to racially or ethnically diverse or under-resourced parents, who may face additional daily stressors not directly related to parenting. Implementation studies are needed to extend the reach of PT to more diverse and under-resourced populations. Despite these limitations, this study demon- strated the superiority of PT on reducing child disruptive

behavior, improving child adaptive behavior, decreasing parental stress and strain and improving parental sense of competence.

Acknowledgments We would like to thank our team for their con- tributions to this project: Jill Pritchett at Ohio State University; Laura Simone at Yale TrialDB; Yanhong Deng, Saankari Anusha Challa, Denis Sukhodolsky, James Dziura, and Allison Gavaletz at Yale; Car- rie McGinnis at Indiana University; Rachael Davis, David McAdam, Bridget Reynolds, Melissa Sturge-Apple, and Amit Chowdhry at Uni- versity of Rochester Medical Center. We also thank the Data and Safety Monitoring Board: Gerald Golden, M.D. (retired pediatric neurologist), Christopher Young, M.D. (Medical Director of Wellmore Behavioral Health, Waterbury, CT and Martin Schwartzman father of a child with autism).

Funding This work was funded by the National Institute of Men- tal Health by the following grants: Yale University/Emory University MH081148 (principal investigator: L. Scahill); University of Pitts- burgh/University of Florida MH080965 (principal investigator: C. Johnson); Ohio State University MH081105 (principal investigator: L. Lecavalier); Indiana University MH081221 (principal investigator: N. Swiezy); University of Rochester MH080906 (principal investigator: T. Smith). Additional support was provided by MH079130 (princi- pal investigator: D Sukhodolsky), the National Center for Advancing Translational Sciences of the National Institutes of Health under Award Numbers UL1 TR000454 (Emory University), UL1 TR000042 (Uni- versity of Rochester), UL1 RR024139 (Yale University) and the Mar- cus Foundation. We thank the families who participated in this study.

Author Contributions SI participated conceived of the current study analyses, participated in its design and coordination, and drafted the manuscript; LL conceived of the current study analyses and drafted the manuscript; BH participated in the study design, performed the statisti- cal analyses, and assisted in drafting the manuscript; TS conceived of the original study, participated in its design and coordination, assisted with data interpretation and helped to draft the manuscript; LL con- ceived of the original study, participated in its design and coordination, assisted with data interpretation and helped to draft the manuscript; CJ conceived of the original study, participated in its design and coordi- nation, assisted with data interpretation and helped to draft the manu- script; NS conceived of the original study, participated in its design and coordination, assisted with data interpretation and helped to draft the manuscript; KB conceived of the original study, participated in its design and coordination, assisted with data interpretation and helped to draft the manuscript; LS conceived of the original study, participated in its design and coordination, assisted with data interpretation and helped to draft the manuscript. All authors read and approved the final manuscript.

Compliance with Ethical Standards

Conflict of interest The authors declare that they have no conflict of interest.

Ethical Approval All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Informed Consent Informed consent was obtained from all indi- vidual participants included in the study.

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References

Abbeduto, L., Seltzer, M. M., Shattuck, P., Krauss, M. W., Orsmond, G., & Murphy, M. M. (2004). Psychological well-being and cop- ing in mothers of youths with autism, down syndrome, or Fragile X Syndrome. American Journal on Mental Retardation, 109(3), 237–254.

Abidin, R. (1995). Manual for the parenting stress index. Odessa, FL: Psychological Assessment Resources.

Abikoff, H. B., Vitiello, B., Riddle, M. A., Cunningham, C., Greenhill, L. L., Swanson, J. M., … Wigal, S. B. (2007). Methylphenidate effects on functional outcomes in the preschoolers with attention- deficit/hyperactivity disorder treatment study (PATS). Journal of Child and Adolescent Psychopharmacology, 17(5), 581–592.

Aman, M. G., McDougle, C. J., Scahill, L., Handen, B., Arnold, L. E., Johnson, C., … Wagner, A. (2009). Medication and parent training in children with pervasive developmental disorders and serious behavior problems: Results from a randomized clinical trial. Journal of the American Academy of Child & Adolescent Psychiatry, 48(12), 1143–1154.

Aman, M. G., Singh, N. N., Stewart, A. W., & Field, C. J. (1985). Psychometric characteristics of the aberrant behavior checklist. American Journal of Mental Deficiency, 89(5), 492–502.

American Psychiatric Association. (2000). Diagnostic and statistical manual of mental disorder: DSM-IV-TR. Washington, DC: Ameri- can Psychiatric Association.

Arbuckle, J. L. (2006). Amos (version 7.0) [computer program]. Chi- cago: SpSS.

Barroso, N. E., Hungerford, G. M., Garcia, D., Graziano, P. A., & Bag- ner, D. M. (2016). Psychometric properties of the Parenting Stress Index-Short Form (PSI-SF) in a high-risk sample of mothers and their infants. Psychological Assessment, 28(10), 1331–1335. doi:10.1037/pas0000257.

Bearss, K., Johnson, C., Handen, B., Smith, T., & Scahill, L. (2013). A pilot study of parent training in young children with autism spectrum disorders and disruptive behavior. Journal of Autism and Developmental Disorders, 43(4), 829–840.

Bearss, K., Johnson, C., Smith, T., Lecavalier, L., Swiezy, N., Aman, M., … Scahill, L. (2015). Effect of parent training vs parent edu- cation on behavioral problems in children with autism spectrum disorder: A randomized clinical trial. Journal of the American Medical Association, 313(15), 1524–1533.

Benson, P. R. (2014). Coping and psychological adjustment among mothers of children with ASD: An accelerated longitudinal study. Journal of Autism and Developmental Disorders, 44(8), 1793–1807.

Boström, P., Broberg, M., & Bodin, L. (2011). Child’s positive and negative impacts on parents—A person-oriented approach to understanding temperament in preschool children with intellec- tual disabilities. Research in Developmental Disabilities, 32(5), 1860–1871.

Brannan, A. M., Heflinger, C. A., & Bickman, L. (1997). The caregiver strain questionnaire measuring the impact on the family of living with a child with serious emotional disturbance. Journal of Emo- tional and Behavioral Disorders, 5(4), 212–222.

Chacko, A., Wymbs, B. T., Wymbs, F. A., Pelham, W. E., Swanger- Gagne, M. S., Girio, E., … Phillips, C. (2009). Enhancing tradi- tional behavioral parent training for single mothers of children with ADHD. Journal of Clinical Child & Adolescent Psychology, 38(2), 206–218.

Coolican, J., Smith, I. M., & Bryson, S. E. (2010). Brief parent train- ing in pivotal response treatment for preschoolers with autism. Journal of Child Psychology and Psychiatry, 51(12), 1321–1330.

Danforth, J. S., Harvey, E., Ulaszek, W. R., & McKee, T. E. (2006). The outcome of group parent training for families of children

with attention-deficit hyperactivity disorder and defiant/aggres- sive behavior. Journal of Behavior Therapy and Experimental Psychiatry, 37(3), 188–205.

Dretzke, J., Davenport, C., Frew, E., Barlow, J., Stewart-Brown, S., Bayliss, S., … Hyde, C. (2009). The clinical effectiveness of different parenting programmes for children with conduct prob- lems: A systematic review of randomised controlled trials. Child and Adolescent Psychiatry and Mental Health, 3(1), 1.

Enders, C. K. (2001). A primer on maximum likelihood algorithms available for use with missing data. Structural Equation Mod- eling, 8(1), 128–141.

Falk, N. H., Norris, K., & Quinn, M. G. (2014). The factors predict- ing stress, anxiety and depression in the parents of children with autism. Journal of Autism and Developmental Disorders, 44(12), 3185–3203.

Gibaud-Wallston, J., & Wandersman, L. P. (1978). Parenting sense of competence scale. Mahwah, NJ: Lawrence Erlbaum Associates.

Gilmore, L., & Cuskelly, M. (2009). Factor structure of the Parenting Sense of Competence scale using a normative sample. Child: Care, Health and Development, 35(1), 48–55.

Greenberg, J. S., Seltzer, M. M., Hong, J., & Orsmond, G. I. (2006). Bidirectional effects of expressed emotion and behavior prob- lems and symptoms in adolescents and adults with autism. American Journal on Mental Retardation, 111(4), 229–249.

Hastings, R. P., Kovshoff, H., Brown, T., Ward, N. J., Espinosa, Degli, F., & Remington, B. (2005). Coping strategies in mothers and fathers of preschool and school-age children with autism. Autism: The International Journal of Research and Practice, 9(4), 377–391.

Hsiao, Y.-J. (2016). Pathways to mental health-related quality of life for parents of children with autism spectrum disorder: Roles of parental stress, children’s performance, medical support, and neighbor support. Research in Autism Spectrum Disorders, 23, 122–130.

Johnson, C. R., Handen, B. L., Butter, E., Wagner, A., Mulick, J., Sukhodolsky, D. G., … Aman, M. G. (2007). Development of a parent training program for children with pervasive devel- opmental disorders. Behavioral Interventions, 22(3), 201–221.

Kaat, A. J., Lecavalier, L., & Aman, M. G. (2014). Validity of the Aberrant Behavior Checklist in children with autism spectrum disorder. Journal of Autism and Developmental Disorders, 44(5), 1103–1116.

Karst, J. S., & Van Hecke, A. V. (2012). Parent and family impact of autism spectrum disorders: A review and proposed model for intervention evaluation. Clinical Child and Family Psychology Review, 15(3), 247–277.

Kasari, C., Gulsrud, A., Paparella, T., Hellemann, G., & Berry, K. (2015). Randomized comparative efficacy study of parent-medi- ated interventions for toddlers with autism. Journal of Consult- ing and Clinical Psychology, 83(3), 554.

Kogan, M. D., Strickland, B. B., Blumberg, S. J., Singh, G. K., Per- rin, J. M., & van Dyck, P. C. (2008). A national profile of the health care experiences and family impact of autism spectrum disorder among children in the United States, 2005–2006. Pedi- atrics, 122(6), e1149-e1158.

Lecavalier, L., Smith, T., Johnson, C., Bearss, K., Swiezy, N., Aman, M. G., … Scahill, L. (2017). Moderators of parent training for disruptive behaviors in young children with autism spec- trum disorder. Journal of Abnormal Child Psychology, 45, 1235–1245.

Little, R. J. (1988). A test of missing completely at random for multi- variate data with missing values. Journal of the American Statisti- cal Association, 83(404), 1198–1202.

Lord, C., Rutter, M., DiLavore, P., & Risi, S. (2002). Autism Diagnostic Observation Schedule: ADOS. Los Angeles: Western Psychologi- cal Services.

J Autism Dev Disord

1 3

Lundahl, B., Risser, H. J., & Lovejoy, M. C. (2006). A meta-analysis of parent training: Moderators and follow-up effects. Clinical Psy- chology Review, 26(1), 86–104.

McArdle, J. J. (2009). Latent variable modeling of differences and changes with longitudinal data. Annual Review of Psychology, 60, 577–605.

Moreland, A. D., Felton, J. W., Hanson, R. F., Jackson, C., & Dumas, J. E. (2016). The relation between parenting stress, locus of control and child outcomes: Predictors of change in a parenting interven- tion. Journal of Child and Family Studies, 25(6), 2046–2054.

Mullen, E. (1995). Mullen Scales of Early Learning. Circle Pines, MN: American Guidance Service Inc.

Postorino, V., Sharp, W. G., McCracken, C. E., Bearss, K., Burrell, T. L., Evans, A. N., & Scahill, L. (2017). A systematic review and meta-analysis of parent training for disruptive behavior in chil- dren with autism spectrum disorder. Clinical Child and Family Psychology Review, 1–12.

Pottie, C. G., & Ingram, K. M. (2008). Daily stress, coping, and well- being in parents of children with autism: A multilevel modeling approach. Journal of Family Psychology, 22(6), 855–864.

Rao, P. A., & Beidel, D. C. (2009). The impact of children with high- functioning autism on parental stress, sibling adjustment, and fam- ily functioning. Behavior Modification, 33(4), 437–451.

Rezendes, D. L., & Scarpa, A. (2011). Associations between paren- tal anxiety/depression and child behavior problems related to autism spectrum disorders: The roles of parenting stress and parenting self-efficacy. Autism Research and Treatment. doi:10.1155/2011/395190.

Roid, G. H. (2003). Stanford-Binet Intelligence Scales. Itasca, IL: Riv- erside Publishing.

Rutter, M., Le Couteur, A., & Lord, C. (2003). Autism Diagnostic Interview-Revised. Los Angeles: Western Psychological Services.

Scahill, L., Bearss, K., Lecavalier, L., Smith, T., Swiezy, N., Aman, M. G., … Turner, K. (2016). Effect of parent training on adaptive behavior in children with autism spectrum disorder and disruptive behavior: Results of a randomized trial. Journal of the American Academy of Child & Adolescent Psychiatry, 55(7), 602–609.

Skotarczak, L., & Lee, G. K. (2015). Effects of parent management praining programs on disruptive behavior for children with a developmental disability: A meta-analysis. Research in Develop- mental Disabilities, 38, 272–287.

Smith, T., & Iadarola, S. (2015). Evidence base update for autism spec- trum disorder. Journal of Clinical Child & Adolescent Psychol- ogy, 44(6), 897–922.

Strauss, K., Vicari, S., Valeri, G., D’Elia, L., Arima, S., & Fava, L. (2012). Parent inclusion in early intensive behavioral interven- tion: The influence of parental stress, parent treatment fidelity and parent-mediated generalization of behavior targets on child out- comes. Research in Developmental Disabilities, 33(2), 688–703.

Tonge, B., Brereton, A., Kiomall, M., Mackinnon, A., King, N., & Rinehart, N. (2006). Effects on parental mental health of an educa- tion and skills training program for parents of young children with autism: A randomized controlled trial. Journal of the American Academy of Child & Adolescent Psychiatry, 45(5), 561–569.

Tonge, B., Brereton, A., Kiomall, M., Mackinnon, A., & Rinehart, N. (2014). A randomised group comparison controlled trial of ‘preschoolers with autism’: A parent education and skills traning intervention for young children with autistic disorder. Autism: The International Journal of Research and Practice, 18(2), 166–177.

Vasilopoulou, E., & Nisbet, J. (2016). The quality of life of parents of children with autism spectrum disorder: A systematic review. Research in Autism Spectrum Disorders, 23, 36–49.

Wells, K. C., Epstein, J. N., Hinshaw, S. P., Conners, C. K., Klaric, J., Abikoff, H. B., … Greenhill, L. L. (2000). Parenting and family stress treatment outcomes in attention deficit hyperactivity disor- der (ADHD): An empirical analysis in the MTA study. Journal of Abnormal Child Psychology, 28(6), 543–553.

Whittingham, K., Sofronoff, K., Sheffield, J., & Sanders, M. R. (2009). Stepping Stones Triple P: An RCT of a parenting program with parents of a child diagnosed with an autism spectrum disorder. Journal of Abnormal Child Psychology, 37(4), 469–480.