OBJECTIVE: To test Stepping Stones Triple P (SSTP) and Acceptance and Commitment Therapy (ACT) in a trial targeting behavioral problems in children with cerebral palsy (CP).
METHODS: Sixty-seven parents (97.0% mothers; mean age 38.7 ± 7.1 years) of children (64.2% boys; mean age 5.3 ± 3.0 years) with CP (Gross Motor Function Classification System = 15, 22%; II = 18, 27%; III =12, 18%; IV = 18, 27%; V = 4, 6%) participated and were randomly assigned to SSTP, SSTP + ACT, or waitlist. Primary outcomes were behavioral and emotional problems (Eyberg Child Behavior Inventory [ECBI], Strengths and Difficulties Questionnaire [SDQ]) and parenting style (Parenting Scale [PS]) at postintervention and 6-month follow-up.
RESULTS: SSTP with ACT was associated with decreased behavioral problems (ECBI Intensity mean difference [MD] = 24.12, confidence interval [CI] 10.22 to 38.03, P = .003; ECBI problem MD = 8.30, CI 4.63 to 11.97, P < .0001) including hyperactivity (SDQ MD = 1.66, CI 0.55 to 2.77, P = .004), as well as decreased parental overreactivity (PS MD = 0.60, CI 0.16 to 1.04, P = .008) and verbosity (PS MD = 0.68, CI 0.17 to 1.20, P = .01). SSTP alone was associated with decreased behavioral problems (ECBI problems MD = 6.04, CI 2.20 to 9.89, P = .003) and emotional symptoms (SDQ MD = 1.33, CI 0.45 to 2.21, P = .004). Decreases in behavioral and emotional problems were maintained at follow-up.
CONCLUSIONS: SSTP is an effective intervention for behavioral problems in children with CP. ACT delivers additive benefits.
- ACT —
- Acceptance and Commitment Therapy
- ANCOVAs —
- analyses of covariance
- ASDs —
- autism spectrum disorders
- CI —
- confidence interval
- CP —
- cerebral palsy
- ECBI —
- Eyberg Child Behavior Inventory
- GMFCS —
- Gross Motor Function Classification System
- MD —
- mean difference
- PS —
- Parenting Scale
- RCT —
- randomized controlled trial
- SDQ —
- Strengths and Difficulties Questionnaire
- SSTP —
- Stepping Stones Triple P
- Triple P —
- Positive Parenting Program
- WL —
- waitlist control
What’s Known on This Subject:
One in 4 children with cerebral palsy (CP) have a behavioral disorder. Parenting interventions are an efficacious approach to treating behavioral disorders. There is a paucity of research on parenting interventions with families of children with CP.
What This Study Adds:
This is the first study to demonstrate the efficacy of a parenting intervention in targeting behavioral problems in children with CP. Further, results suggest that Acceptance and Commitment Therapy delivers additive benefits above and beyond established parenting interventions.
Cerebral palsy (CP) is a permanent disorder of the development of movement and posture that is caused by nonprogressive disturbance to the developing fetal or infant brain.1 It is the most common physical disability in childhood, occurring in 2.0 to 2.5 of every 1000 live births.2 Children with CP, akin to children with disabilities generally, are more likely to experience behavioral and emotional problems.3–5 A recent meta-analysis showed that 1 in 4 children with CP have a behavioral disorder6 compared with 1 in 10 typically developing children7; however, despite recognition of the problem, there is a paucity of research and clinical services to address this issue.
Parenting interventions target behavioral and emotional problems of childhood through enhancing parenting.8 Parenting interventions based in social learning theory and behavioral analysis have wide empirical support with typically developing children.9–11 Stepping Stones Triple P (SSTP) is a variant of the widely disseminated Positive Parenting Program (Triple P) that targets families of children with disabilities.12 A recent meta-analysis found that SSTP has a moderate effect on child behavioral outcomes (d = 0.537) and a large effect on parenting style (d= 0.725),13 consistent with meta-analyses of Triple P with typically developing children.14–17 Further, SSTP has demonstrated efficacy specifically for families of children with autism spectrum disorders (ASDs).18 Although parenting interventions, such as SSTP, are an evidence-based approach for targeting behavioral and emotional problems in childhood and are readily accessible by parents, there are no published randomized controlled trials (RCTs) of a parenting intervention for families of children with CP.19
Acceptance and Commitment Therapy (ACT) is a new cognitive behavioral therapy that emphasizes nonjudgmental psychological contact with present moment experience and engagement in meaningful, values-driven activities.20 The goal of ACT is to increase psychological flexibility, the ability to persist or change one’s behavior, with full awareness of the situational context and one’s own present-moment experience, in the service of chosen values. ACT has a growing evidence base; it is at least as effective as older cognitive behavioral therapy models with some evidence suggesting greater efficacy.21–23 ACT may enhance established behavioral parenting interventions by addressing parental cognition and emotions.24,25 This new approach is particularly promising for families of children with disabilities, with RCTs urgently needed.26
Our aim was to test the efficacy of SSTP, with and without ACT, in targeting child behavioral and emotional problems and dysfunctional parenting in families of children with CP.
The study design is detailed in full in the study protocol.27 This was a 2-phase RCT with 3 groups (SSTP, SSTP + ACT, waitlist control [WL]). The first phase, the primary focus of the study, involved a comparison among SSTP, SSTP with ACT, and WL groups at postintervention. After postintervention assessment, the WL group, for ethical reasons, was offered SSTP. If WL families completed SSTP, then they also completed additional postintervention assessment, along with 6-month follow-up assessment. The second phase of the study examined effects at follow-up and included all families who received an intervention and completed 6-month follow-up assessment. The second phase included a pre-post design component, examining the retention of intervention effect from postintervention to 6-month follow-up, as well as a comparison between families who received SSTP and families who received SSTP with ACT at 6-month follow-up.
Ethical clearance was obtained from the Children’s Health Queensland Human Research Ethics Committee, the University of Queensland Behavioral and Social Sciences Ethical Review Committee, and the Cerebral Palsy League of Queensland Research Ethics Committee; all participating parents signed a consent form before participation.
Participants were parents of children (2–12 years) with a diagnosis of CP (all gross motor functioning severity levels) who self-identified as needing a parenting intervention after discussion on what a parenting intervention could target. Participants were recruited from the databases of the Queensland Cerebral Palsy and Rehabilitation Research Centre, the Cerebral Palsy League, and the Queensland Cerebral Palsy Register, and through presentation at the Queensland Cerebral Palsy Health Service (Fig 1).
Sample Size Calculation
Sample size calculations were based on the primary outcome: child behavior. An effect size of 0.25 was assumed because it is consistent with a clinically important difference of 0.5 SD and is comparable to the effect size for SSTP obtained with families of children with ASD, η2 = 0.27.18 This leads to a total sample size of 98 (power 0.8, 2-tailed, P = .05) and 110 accounting for attrition. This was not obtained, with recruitment efforts in the available population leading to a final sample size of 67.
The randomization process was completed by computerized sequence generation with block randomization to ensure equal (or near equal) allocation of participants to groups. The group allocations were placed inside sealed, opaque, and numbered envelopes by a staff member not involved in this study. On enrolment of a family, the study coordinator opened the next envelope in sequence. Each study participant was randomized to 1 of 3 groups: SSTP, SSTP with ACT, or WL.
The interventions (SSTP and SSTP + ACT) were delivered in a combined group (3–10 families per group) and telephone format. SSTP consisted of 6 (2-hour) group sessions plus 3 (30-minute) telephone consultations and was delivered by psychologists with accreditation in SSTP. SSTP sessions included strategies for building a positive parent-child relationship, encouraging desirable behavior, teaching new skills and behaviors, managing misbehavior, and managing high-risk situations. Parents made specific goals for change and were supported in enacting plans for managing challenging parenting situations. For the SSTP with ACT group, the ACT sessions (two 2-hour group sessions) preceded SSTP. ACT sessions included identifying values, mindfulness, cognitive defusion (distancing from thoughts), acceptance of emotions, and making specific goals for acting on values. For some groups, a weekend workshop format was used to allow for intervention delivery as an outreach program in far North Queensland (Table 1).
The Family Background Questionnaire was used to gather demographic data28 and the Gross Motor Function Classification System (GMFCS) was used to classify gross motor functional ability.29 This article focuses on reporting the primary outcomes: child behavioral and emotional problems (Eyberg Child Behavior Inventory [ECBI], Strengths and Difficulties Questionnaire [SDQ]) and parenting style (Parenting Scale [PS]). All outcomes are parent-report. The ECBI30 produces 2 scales, the intensity and the problem scales, and is considered to show high reliability and validity.31,32 The SDQ33 produces 5 subscales (emotional symptoms, conduct problems, inattention/hyperactivity, peer problems, and prosocial behavior) and is considered to have high reliability and validity.34 The PS35 is a measure of 3 dysfunctional discipline styles: laxness, overreactivity, and verbosity. The PS shows strong reliability and validity. For full details, see the study protocol.27
The first phase, a comparison among SSTP, SSTP with ACT, and WL groups at postintervention, was achieved through a series of analyses of covariance (ANCOVAs), with preintervention scores as a covariate. Significant results were followed-up with linear contrasts examining group-by-group differences (ie, WL vs SSTP, WL vs SSTP + ACT, SSTP vs SSTP + ACT). A Bonferroni correction was applied to linear contrasts to correct for multiple comparisons, resulting in a P value of .0167. A sensitivity analysis was conducted with the last observation carried forward for all participants who failed to complete the postintervention assessment.
The second phase of the study examined effects at follow-up and included all families who received an intervention and completed 6-month follow-up assessment (n = 28; SSTP = 12, SSTP + ACT = 11, WL = 5). A pre-post examination of the retention of the intervention effect from postintervention to 6-month follow-up was tested with a series of t tests. A comparison between families who received SSTP (n = 16) and families who received SSTP with ACT (n = 12) at 6-month follow-up was conducted via a series of ANCOVAs with preintervention scores as a covariate. All WL families received SSTP alone except 1 that received SSTP with ACT.
A series of χ2 and analysis of variance tests identified no differences between the groups on any demographic variable at baseline. Sample characteristics are presented in Table 2.
Fewer than 10% of the data were missing and the pattern of missing data was random. In generating scale scores if <30% of items were missing for that participant on that scale, then the scale score was generated from the remaining items. If >30% of items were missing for that participant, then that participant was excluded from the analysis for that scale.
The assumption of equality of variance was violated for the PS Verbosity scale, and the assumption of homogeneity of regression slopes was violated for the PS Laxness scale. Original, untransformed data are reported.
Intervention Protocol Adherence
The SSTP and ACT content was delivered as per protocol in all scheduled group sessions with the exception that in 8.19% of sessions some aspect of the SSTP DVD was not played owing to technical difficulties or time management. In all circumstances, the content on the SSTP DVD was still delivered verbally. Protocol delivery was rated by a second therapist for 50.81% of sessions with 100% agreement with the primary therapist. Eleven families received the intervention via weekend workshop format (4 SSTP groups, 4 SSTP + ACT groups, 3 WL groups). Within the SSTP group, participants attended a mean of 5.31 (SD 0.79) of 6 group sessions and a mean of 2.87 (SD 0.34) of 3 phone consultations. Within the SSTP with ACT group, participants attended a mean of 5.25 (SD 0.97) of 6 group sessions, a mean of 2.75 (SD 0.44) of 3 phone consultations, and a mean of 1.95 ACT group sessions (SD 0.22). If a participant missed a scheduled group session, every attempt was made to arrange an individual make-up session, with SSTP participants receiving a mean of 0.44 (SD 0.40) SSTP make-up sessions and SSTP with ACT participants receiving a mean of 0.55 (SD 1.0) SSTP make-up sessions and a mean of 0.10 (SD 0.31) ACT make-up sessions.
Primary Outcomes of RCT: Comparison of Groups at Postintervention
Consistent with an intervention effect, the 3 groups showed significant differences at postintervention for parent-reported child behavioral and emotional problems, including on the ECBI Intensity scale, F2,54 = 6.15, P = .004; the ECBI Problem scale, F2,48 = 11.03, P < .0001; the SDQ Emotional symptoms scale, F2,53 = 4.88, P = .01; and the SDQ Hyperactivity scale, F2,54 = 4.55, P = .01. Significant differences were not found on the Conduct problems, Peer problems, Prosocial, or Impact scales of the SDQ. In addition, the 3 groups showed significant differences in dysfunctional parenting styles on the PS Overreactivity scale, F2,52 = 3.84, P = .03, and the PS Verbosity scale F2,53 = 3.80, P = .03. Significant differences were not found for the PS Laxness scale. The results of all ANCOVAs are presented in detail in Table 3.
SSTP with ACT participants showed decreased parent-reported child behavioral and emotional problems in comparison with the WL group on the ECBI Intensity scale (mean difference [MD] = 24.12, P = .003), the ECBI Problem scale (MD = 8.30, P < .000), and the SDQ Hyperactivity scale (MD = 1.66, P = .004), as demonstrated in the MD scores. Significant differences between SSTP with ACT and WL were not found on the SDQ Emotional symptoms scale. SSTP showed decreased parent-reported child behavioral and emotional problems in comparison with the WL group on the ECBI Problem scale (MD = 6.04, P = .003) and the SDQ Emotional symptoms scale (MD = 1.33, P = .004). Differences approached significance for the ECBI Intensity scale (MD = 15.43, P = .04). Significant differences between SSTP and WL were not found on the SDQ Hyperactivity scale. No significant differences between SSTP and SSTP with ACT were found. Differences between the SSTP and SSTP with ACT approached significance for SDQ Emotional symptoms scale only, with SSTP demonstrating lower parent-reported emotional symptoms.
SSTP with ACT showed decreased dysfunctional parenting styles in comparison with the WL group on the PS Overreactivity scale (MD = 0.60, P = .008) and the PS Verbosity scale (MD = 0.68, P = .01). No significant differences were found between SSTP and WL on dysfunctional parenting styles. No significant differences were found between SSTP and SSTP with ACT on dysfunctional parenting styles. Linear contrasts are presented in full in Table 4.
Sensitivity Analysis: Intention to Treat
A conservative sensitivity analysis, repeating ANCOVAs with the last observation carried forward for all families who failed to complete postintervention assessments, was conducted to satisfy intention to treat (n = 67). The interpretation of the results was in all cases consistent with the results reported previously.
Retention of Effect: A Pre-Post Analysis From Postintervention to Follow-Up
Families receiving SSTP showed significant improvements on the SDQ Prosocial scale, t14 = –0.26, P = .01, from postintervention to 6-month follow-up and significant increases in dysfunctional parenting on the PS Verbosity scale, t13 = –2.31, P = .04, from postintervention to 6-month follow-up. Families receiving SSTP with ACT showed significant increases in dysfunctional parenting from postintervention to 6-month follow-up on the PS Overreactivity scale, t10 = –2.49, P = .3, and the PS Verbosity scale, t10 = –3.09, P = .01. All other t tests were nonsignificant, consistent with maintenance of gains.
Comparison of Families Receiving SSTP and SSTP + ACT at Follow-Up
Families that received SSTP with ACT showed decreased child behavioral problems and dysfunctional parenting in comparison with families that received SSTP alone at 6-month follow-up on the SDQ Hyperactivity scale, F2,24 = 7.29, P = .012; the PS Laxness scale, F2,23 = 4.8, P = .038; and the PS Verbosity scale, F2,24 = 10.70, P = .003. These comparisons should be interpreted with caution owing to lower sample size (SSTP = 16; SSTP + ACT = 12). The ANCOVAs and follow-up means are presented in full in Table 5.
Children with CP are at increased risk of behavioral and emotional problems, with 1 in 4 developing a behavioral disorder.6 This study is the first to demonstrate that parenting intervention, particularly SSTP or SSTP combined with ACT, is efficacious in targeting behavioral and emotional problems in children with CP. SSTP alone was associated with reductions in parent-reported child behavioral and emotional problems consistent with previous research.13,18 Further, SSTP combined with ACT was associated with reductions in dysfunctional parenting styles. The effect sizes obtained for the primary outcome (ECBI Intensity = 0.19; ECBI Problem = 0.32) are comparable to effects obtained in families of children with ASDs (ECBI Intensity = 0.26; ECBI Problem = 0.16).18 This illustrates the urgent need for clinical services to address behavioral and emotional problems in children with CP, as well as the good fit between this clinical need and the efficacy of parenting intervention. Parenting interventions, particularly Triple P, are ideally translatable. Triple P is designed for population-level dissemination, easily implemented within health or educational services, deliverable in high- and low-resource areas, and available in 25 countries.36 Parenting interventions, such as SSTP, should therefore form part of standard care for families of children with CP.
To our knowledge, this was the first RCT to test the additive benefit of ACT, above and beyond an established behavioral parenting intervention. The results suggest that ACT provides an additional contribution, with particular benefits shown for parenting style and child hyperactivity. The combined SSTP and ACT intervention, but not SSTP alone, was associated with reductions in child hyperactivity, parental overreactivity, parental verbosity, and child behavioral problems on the ECBI Intensity scale. At 6-month follow-up, families who had received the combined SSTP with ACT intervention showed reductions in child hyperactivity, parental laxness, and parental verbosity compared with families who had received SSTP alone. The combined SSTP with ACT intervention may have enhanced parenting by increasing psychological flexibility.24
Families receiving SSTP alone and not families receiving combined SSTP with ACT, showed decreased child emotional symptoms on the SDQ compared with WL. Further, the differences between SSTP alone and combined SSTP with ACT approached significance, with SSTP showing decreased child emotional symptoms. This is an intriguing finding, as it is challenging to understand how the addition of ACT may have decreased the intervention effect of SSTP. It may be that ACT, with a focus on mindfulness, acceptance of emotions, and valued parenting acts, increased parental awareness of child affect, thus inflating child emotional symptoms scores on the parent-report measure of the SDQ. This requires further research.
A limitation of this study is that the sample size goal of 98 families was not reached, leading to reduced power. Further, primary outcomes are parent-report. Future research should explore if parenting intervention is a useful supplement to existing interventions for families of children with CP; for example, supporting an environmental enrichment intervention37 or a home therapy program.38 In addition, research should focus on confirming an additive benefit of ACT, investigating generalizibility, and testing an integrated ACT parenting intervention. The effects of parent-delivered ACT on child emotional symptoms and parental awareness of child affect warrants further research. If ACT does increase parental awareness of child emotions, it may provide a means to target emotional responsiveness and the parent-child relationship.39
This study demonstrates, via an RCT, the efficacy of a readily available parenting intervention, SSTP, in targeting behavioral and emotional problems in children with CP. Further, results suggest that ACT delivers additive benefits above and beyond established parenting interventions. It is recommended that parenting intervention be incorporated into standard care for families of children with CP.
- Accepted February 14, 2014.
- Address correspondence to Koa Whittingham, PhD, Queensland Cerebral Palsy and Rehabilitation Research Centre, Level 7, Block 6, RBWH Herston, Brisbane, Australia 4029. E-mail:
Dr Whittingham conceptualized and designed the study, with mentorship from senior authors, managed the randomized controlled trial, conducted the analysis, and drafted the initial manuscript; Drs Sanders, McKinlay, and Boyd provided mentorship in the conceptualization and design of the study, and reviewed and revised the manuscript; and all authors approved the final manuscript as submitted.
This trial has been registered with the Australian New Zealand Clinical Trials Registry (identifier 00336291).
FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose.
FUNDING: This work was supported by a National Health and Medical Research Council postdoctoral fellowship, grant 631712, to Dr Whittingham; a National Health and Medical Research Council career development fellowship, grant 1037220, to Dr Boyd; and a Smart State Fellowship to Dr Boyd.
POTENTIAL CONFLICT OF INTEREST: Stepping Stones Triple P is owned by the University of Queensland and sublicensed to Uniquest, the University of Queensland’s Technology Transfer Company. Dr Sanders is a coauthor of the Stepping Stones Triple P program and receives royalty payments from the publisher, Triple P International, in accordance with the University of Queensland Intellectual Property Policy. The other authors have no conflicts of interest to disclose.
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- Copyright © 2014 by the American Academy of Pediatrics