Interventions to Reduce Behavioral Problems in Children With Cerebral Palsy: An RCT

Transcription

1 ARTICLE Interventions to Reduce Behavioral Problems in Children With Cerebral Palsy: An RCT AUTHORS: Koa Whittingham, PhD, a,b Matthew Sanders, PhD, b Lynne McKinlay, MD, c and Roslyn N. Boyd, PhD a a Queensland Cerebral Palsy and Rehabilitation Research Centre, School of Medicine, and b Parenting and Family Support Centre, School of Psychology, The University of Queensland, Brisbane, Australia; and c Queensland Paediatric Rehabilitation Service, Royal Children s Hospital, Brisbane, Australia KEY WORDS parenting, behavioral family intervention, cerebral palsy, acceptance and commitment therapy, mindfulness ABBREVIATIONS 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 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 ). doi: /peds Accepted for publication Feb 14, 2014 Address correspondence to Koa Whittingham, PhD, Queensland Cerebral Palsy and Rehabilitation Research Centre, Level 7, Block 6, RBWH Herston, Brisbane, Australia koawhittingham@uq.edu.au PEDIATRICS (ISSN Numbers: Print, ; Online, ). Copyright 2014 by the American Academy of Pediatrics (Continued on last page) 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. abstract 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 years) of children (64.2% boys; mean age 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] 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. Pediatrics 2014;133:1 9 PEDIATRICS Volume 133, Number 5, May

2 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 disorder 6 compared with 1 in 10 typically developing children 7 ; 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 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 Arecent 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 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 sbehavior,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 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. METHODS Design 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 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, h 2 = 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. Procedure 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 2 WHITTINGHAM et al

3 ARTICLE FIGURE 1 Study protocol and participant flow. PEDIATRICS Volume 133, Number 5, May

4 a staff member notinvolvedin 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 NorthQueensland(Table1). Assessment The Family Background Questionnaire was used to gather demographic data 28 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 ECBI 30 produces 2 scales, the intensity and the problem scales, and is considered to show high reliability and TABLE 1 Comparison of Intervention Content: SSTP and ACT Intervention Content SSTP ACT Discussion of how parents cope with stress with a focus on identifying the x workability of various coping strategies. Use of metaphor to promote psychological flexibility (eg, the quicksand metaphor x is used to explain how struggling with psychological distress often increases psychological distress). Identification of values, that is, overarching desired qualities of living (eg, being x a loving parent). Mindfulness exercises to promote psychological contact with the present x moment, including thoughts and emotions. Exercises included mindfulness of breathing, mindfulness of thoughts and mindfulness of emotions. Cognitive defusion exercises to create psychological distance from thoughts (eg, x saying the thought in the voice of a cartoon character). Setting specific goals for acting on values (eg, acting on the value being a loving x parent by responding to child s requests for physical affection). Discussion of positive parenting (eg, using assertive discipline). x Discussion of the causes of child behavior problems (eg, accidentally rewarding x child s behavior with attention). Setting specific goals for change in parent and child behavior (eg, reducing the x frequency of temper tantrums). Monitoring of child behavior (eg, recording the frequency of temper tantrums). x Reviewing parenting strategies to develop a positive parent-child relationship x (eg, quality time). Reviewing parenting strategies to encourage desirable behavior (eg, using x descriptive praise). Reviewing parenting strategies to teach new skills and behaviors (eg, using ask, x say, do to teach new skills in steps). Reviewing parenting strategies to manage misbehavior (eg, planned ignoring). x Creating planned activities routines for high-risk parenting situations (eg, x creating a plan, including various parenting strategies to improve child behavior while shopping). Reviewing implemented planned activities routines, as well as other behavioral x change goals, and considering future changes in a structured way., not included. validity. 31,32 The SDQ 33 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 PS 35 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 Statistical Analysis 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 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 4 WHITTINGHAM et al

5 ARTICLE a series of ANCOVAs with preintervention scores as a covariate. All WL families received SSTP alone except 1 that received SSTP with ACT. RESULTS Sample Characteristics A series of x 2 and analysis of variance tests identified no differences between the groups on any demographic variable at baseline. Sample characteristics are presented in Table 2. Preliminary Analysis 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 TABLE 2 Sample Characteristics of Participating Families (n = 67) Variable WL (n = 22) SSTP (n = 22) SSTP + ACT (n = 23) Demographics Child age, y, mean (SD) 4.96 (2.95) 5.45 (3.16) 5.52 (3.17) Child gender, boys, n (%) 13 (59.1) 13 (59.1) 17 (73.9) Intellectual disability, n (%) 5 (22.7) 3 (13.4) 5 (21.7) Learning disability, n (%) 6 (27.3) 7 (31.8) 6 (26.1) Autism spectrum disorder, n (%) 2 (9.1) 1 (5.9) 1 (4.3) Attention deficit hyperactivity disorder, n (%) 1 (4.5) 0 0 Vision impairment, n (%) 4 (18.2) 3 (13.6) 7 (33.4) Hearing impairment, n (%) 1 (4.5) 3 (13.6) 2 (8.7) Receiving services for emotional/behavioral 2 (9.1) 2 (9.5) 4 (17.4) problems, n (%) Classification GMFCS I 6 (27.3) 5 (22.7) 4 (17.4) GMFCS II 6 (27.3) 5 (22.7) 7 (30.4) GMFCS III 3 (13.6) 5 (22.7) 4 (17.4) GMFCS IV 6 (27.3) 5 (22.7) 7 (30.4) GMFCS V 1 (4.5) 2 (9.1) 1 (4.3) Relationship to child, mother, n (%) 20 (90.9) 22 (100) 23 (100) (if not mother, father) Parent age, y, mean (SD) (6.09) (5.55) (9.39) Parent marital status Married 18 (81.8) 19 (86.4) 14 (60.9) Defacto 0 1 (4.5) 5 (21.7) Separated 1 (4.5) 1 (4.5) 1 (4.3) Divorced 2 (9.1) 0 1 (4.3) Never married/defacto 0 1 (4.5) 2 (8.7) Family type Original family 17 (77.3) 21 (95.5) 17 (73.9) Sole parent family 4 (18.2) 1 (4.5) 3 (13.0) Step family 1 (4.5) 0 3 (13.0) Education level of participating parent, Year 10/11 1 (4.5) 2 (9.1) 3 (13.0) Year 12 4 (18.2) 1 (4.5) 1 (4.3) Trade/apprenticeship 2 (9.1) 1 (4.5) 0 TAFE/college certificate 4 (18.2) 5 (22.7) 9 (39.1) University degree 11 (50.0) 13 (59.1) 10 (43.5) Employment of participating parent Full-time 1 (4.5) 1 (4.5) 5 (21.7) Part-time 9 (40.9) 13 (59.1) 10 (43.5) Unemployed (seeking work) 1 (4.5) 1 (4.5) 0 Full-time parent/home duties 11 (50.0) 7 (31.8) 8 (34.8) Education level of partner (if applicable), (4.3) Year 10/11 1 (4.5) 4 (18.2) 3 (13.0) Year 12 2 (9.1) 1 (4.5) 2 (8.7) Trade/apprenticeship 4 (18.2) 4 (18.2) 3 (13.0) Technical and Further Education 2 (9.1) 1 (9.1) 4 (17.4) (TAFE)/college certificate University degree 9 (40.9) 9 (40.9) 6 (26.1) Employment of partner (if applicable) Full-time 16 (72.7) 18 (81.8) 13 (56.5) Part-time 1 (4.5) 1 (4.5) 3 (13.0) Unemployed (seeking work) 0 1 (4.5) 1 (4.3) Full-time parent/home duties 1 (4.5) 0 2 (8.7) Family income, (18.2) 2 (9.1) 5 (21.7) (4.5) 1 (4.5) 3 (13.0) (36.4) 2 (9.1) 4 (17.4) (36.4) 16 (72.7) 11 (47.8) Professional advice in last 6 mo from Psychologist, n (%) 5 (22.7) 6 (27.3) 5 (21.7) PEDIATRICS Volume 133, Number 5, May

6 TABLE 2 Continued Variable WL (n = 22) SSTP (n = 22) SSTP + ACT (n = 23) Psychiatrist, n (%) 1 (4.5) 1 (4.5) 3 (13.0) Counselor, n (%) 6 (27.3) 4 (18.2) 4 (17.4) Social worker, n (%) 5 (22.7) 6 (27.3) 3 (13.0) Outcome measures at baseline ECBI Intensity (36.57) (38.73) (37.36) ECBI Problem (7.88) (8.83) (8.40) SDQ Emotional symptoms 3.00 (2.72) 3.00 (2.67) 2.48 (1.53) SDQ Conduct problems 2.00 (1.90) 2.04 (1.49) 1.77 (1.48) SDQ Hyperactivity 4.82 (2.68) 4.91 (2.69) 6.22 (1.94) SDQ Peer problems 3.86 (2.34) 3.14 (1.98) 3.61 (2.54) SDQ Prosocial 5.82 (2.74) 6.14 (2.73) 5.66 (2.96) SDQ Impact 2.55 (2.78) 3.32 (3.38) 2.81 (2.66) PS Laxness 2.79 (0.89) 2.77 (0.81) 2.84 (1.05) PS Overreactivity 3.04 (0.87) 3.04 (0.77) 2.68 (0.84) PS Verbosity 3.47 (0.90) 3.62 (0.99) 3.17 (0.85) Where P values are not given, the n per cell was insufficient to test. TAFE,. 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 ontheecbi Intensityscale, F 2,54 =6.15,P =.004; the ECBI Problem scale, F 2,48 = 11.03, P,.0001; the SDQ Emotional symptoms scale, F 2,53 =4.88, P =.01; and the SDQ Hyperactivity scale, F 2,54 =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, F 2,52 = 3.84, P =.03, and the PS Verbosity scale F 2,53 = 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 comparisonwiththewlgroupontheecbi 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 betweenthesstpandsstpwithact 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 TABLE 3 Omnibus ANCOVA comparing WL, SSTP, and SSTP + ACT Groups at Postintervention with Preintervention Scores as a Covariate Variable Unadjusted Postinterention Means (SD) F Partial h 2 WL SSTP SSTP + ACT ECBI Intensity (36.27) (27.66) (30.61) P =.004* ECBI Problem (8.17) (7.03) 9.11 (5.09) P,.000* SDQ Emotional symptoms 3.24 (2.56) 1.76 (2.02) 2.48 (1.44) P =.01* SDQ Conduct problems 2.21 (2.37) 1.41 (1.54) 1.70 (1.69) P =.16 SDQ Hyperactivity 5.37 (2.24) 5.18 (2.92) 4.90 (2.09) P =.01* SDQ Peer problems 4.14 (2.22) 2.47 (1.46) 3.09 (2.26) P =.16 SDQ Prosocial 5.79 (2.64) 6.35 (3.41) 6.05 (2.69) P =.76 SDQ Impact 3.79 (3.53) 3.83 (3.95) 2.82 (2.95) P =.47 PS Laxness 2.76 (0.94) 2.34 (0.81) 2.28 (1.24) P =.20 PS Overreactivity 2.87 (0.94) 2.52 (1.02) 1.94 (0.73) P =.03* PS Verbosity 3.18 (0.77) 2.79 (1.14) 2.29 (1.11) P =.03* *, significant. 6 WHITTINGHAM et al

7 ARTICLE 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 ACTon 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, t 14 = 0.26, P =.01,from postintervention to 6-month follow-up and significant increases in dysfunctional parenting on the PS Verbosity scale, t 13 = 2.31, P =.04,frompostintervention 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, t 10 = 2.49, P =.3, and the PS Verbosity scale, t 10 = 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, F 2,24 = 7.29, P =.012; the PS Laxness scale, F 2,23 = 4.8, P =.038; and the PS Verbosity scale, F 2,24 = 10.70, P =.003. These comparisons should be interpreted TABLE 4 Linear Contrasts Identifying Group Differences at Postintervention Between WL and SSTP, WL and SSTP + ACT, and SSTP and SSTP + ACT Variable Mean Difference Between WL and SSTP Mean Difference Between WL and SSTP + ACT Mean Difference Between SSTP and SSTP + ACT ECBI Intensity (0.78 to 30.08) (10.22 to 38.03) 8.69 ( 5.65 to 23.04) P =.04 P =.003* P =.23 ECBI Problem 6.04 (2.20 to 9.89) 8.30 (4.63 to 11.97) 2.26 ( 1.61 to 6.12) P =.003* P,.0001* P =.25 SDQ Emotional symptoms 1.33 (0.45 to 2.21) 0.37 ( 0.46 to 1.21) ( 1.81 to 0.09) P =.004* P =.371 P =.03 SDQ Conduct problems 0.85 ( 0.23 to 1.72) 0.43 ( 0.41 to 1.26) ( 1.28 to 0.44) P =.056 P =.310 P =.332 SDQ Hyperactivity 0.73 ( 0.40 to 1.86) 1.66 (0.55 to 2.77) 0.93 ( 0.17 to 2.04) P =.203 P =.004* P =.097 SDQ Peer problems 0.77 ( 0.10 to 1.65) 0.64 ( 0.18 to 1.46) ( 0.98 to 0.61) P =.083 P =.122 P =.754 SDQ Prosocial ( 1.68 to 0.78) ( 1.33 to 0.78) 0.29 ( 0.91 to 1.49) P =.470 P =.784 P =.634 SDQ Impact 0.67 ( 1.14 to 2.50) 1.00 ( 0.66 to 2.67) 0.33 ( 1.42 to 2.07) P =.230 P =.230 P =.707 PS Laxness 0.39 ( 0.14 to 0.93) 0.42 ( 0.09 to 0.92) 0.02 ( 0.49 to 0.54) P =.14 P =.10 P =.14 PS Overreactivity 0.27 ( 0.18 to 0.72) 0.60 (0.16 to 1.04) 0.33 ( 0.10 to 0.77) P =.24 P =.008* P =.13 PS Verbosity 0.50 ( 0.03 to 1.04) 0.68 (0.17 to 1.20) 0.18 ( 0.36 to 0.72) P =.06 P =.01* P =.51 Values are MD (CI); *, significant. 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. DISCUSSION 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 parentreported 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 PEDIATRICS Volume 133, Number 5, May

8 TABLE 5 Omnibus ANCOVA Comparing Families Receiving SSTP and SSTP + ACT at 6- Month Follow- Up With Preintervention Scores as a Covariate Variable Unadjusted Follow-Up Means (SD) F Partial h 2 SSTP ACT intervention, but not SSTPalone, 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 SSTP + ACT ECBI Intensity (28.40) (28.83) 2.61 P = ECBI Problem 6.61 (7.51) 4.00 (5.19) 2.09 P = SDQ Emotional symptoms 1.56 (1.96) 1.64 (1.03) 0.00, P =.93 0 SDQ Conduct problems 0.87 (1.20) 1.18 (0.87) 0.00, P =.93 0 SDQ Hyperactivity 5.18 (2.97) 4.82 (1.89) 7.29, P = 012* 0.23 SDQ Peer problems 2.69 (1.49) 1.91 (2.02) 1.58, P = SDQ Prosocial 6.62 (2.70) 6.36 (2.66) 1.19, P = SDQ Impact 1.17 (1.70) 0.50 (0.97) 1.43, P = PS Laxness 2.59 (0.69) 1.94 (0.49) 4.83, P =.038* 0.2 PS Overreactivity 2.61 (0.95) 1.95 (0.60) 1.11, P = PS Verbosity 3.04 (0.71) 2.06 (0.54) 10.70, P =.003* 0.32 *, significant. WL. Further, the differences between SSTP alone and combined SSTP with ACTapproached 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 parentreport. 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 intervention 37 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 CONCLUSIONS 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. REFERENCES 1. Rosenbaum P, Paneth N, Leviton A, et al. A report: the definition and classification of cerebral palsy April Dev Med Child Neurol Suppl. 2007;109(suppl 109): Stanley F, Blair E, Alberman E. Cerebral Palsies: Epidemiology and Causal Pathways: Clinics in Developmental Medicine No London: MacKeith Press; Carlsson M, Olsson I, Hagberg G, Beckung E. Behaviour in children with cerebral palsy with and without epilepsy. Dev Med Child Neurol. 2008;50(10): Parkes J, White-Koning M, Dickinson HO, et al. Psychological problems in children with cerebral palsy: a cross-sectional European study. J Child Psychol Psychiatry. 2008;49(4): Parkes J, White-Koning M, McCullough N, Colver A. Psychological problems in children with hemiplegia: a European multicentre survey. Arch Dis Child. 2009;94(6): Novak I, Hines M, Goldsmith S, Barclay R. Clinical prognostic messages from a systematic review on cerebral palsy. Pediatrics. 2012;130(5). Available at: org/cgi/content/full/130/5/e Costello EJ, Egger H, Angold A. 10-year research update review: the epidemiology of child and adolescent psychiatric disorders: I. Methods and public health burden. JAm Acad Child Adolesc Psychiatry. 2005;44(10): Sanders MR, Dadds MR. Behavioral Family Intervention. Boston: Allyn and Bacon; Patterson GR. Coercive Family Process. Vol. 3. Eugene, Oregon: Castalia Publishing Company; Kazdin AE. Parent management training: evidence, outcomes, and issues. J Am Acad Child Adolesc Psychiatry. 1997;36(10): O Dell S. Training parents in behavior modification: a review. Psychol Bull. 1974;81 (7): Sanders MR, Mazzucchelli TG, Studman LJ. Stepping Stones Triple P: the theoretical 8 WHITTINGHAM et al

9 ARTICLE basis and development of an evidencebased positive parenting program for families with a child who has a disability. J Intellect Dev Disabil. 2004;29(3): Tellegen CL, Sanders MR. Stepping Stones Triple P-Positive Parenting Program for children with disability: a systematic review and meta-analysis. Res Dev Disabil. 2013;34(5): de Graaf I, Speetjens P, Smit F, de Wolff M, Tavecchio L. Effectiveness of the Triple P Positive Parenting Program on behavioral problems in children: a meta-analysis. Behav Modif. 2008;32(5): Nowak C, Heinrichs N. A comprehensive meta-analysis of Triple P-Positive Parenting Program using hierarchical linear modeling: effectiveness and moderating variables. Clin Child Fam Psychol Rev. 2008;11(3): de Graaf I, Speetjens P, Smit F, de Wolff M, Tavecchio L. Effectiveness of the Triple P Positive Parenting Program on parenting: a metaanalysis. Fam Relat. 2008;57(5): Thomas R, Zimmer-Gembeck MJ. 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Emotional availability: concept, research, and window on developmental psychopathology. Dev Psychopathol. 2012;24(1):1 8 (Continued from first page) 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 , to Dr Whittingham; a National Health and Medical Research Council career development fellowship, grant , 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. PEDIATRICS Volume 133, Number 5, May

10 Interventions to Reduce Behavioral Problems in Children With Cerebral Palsy: An RCT Koa Whittingham, Matthew Sanders, Lynne McKinlay and Roslyn N. Boyd Pediatrics originally published online April 7, 2014; Updated Information & Services Permissions & Licensing Reprints including high resolution figures, can be found at: Information about reproducing this article in parts (figures, tables) or in its entirety can be found online at: Information about ordering reprints can be found online:

11 Interventions to Reduce Behavioral Problems in Children With Cerebral Palsy: An RCT Koa Whittingham, Matthew Sanders, Lynne McKinlay and Roslyn N. Boyd Pediatrics originally published online April 7, 2014; The online version of this article, along with updated information and services, is located on the World Wide Web at: Pediatrics is the official journal of the American Academy of Pediatrics. A monthly publication, it has been published continuously since Pediatrics is owned, published, and trademarked by the American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk Grove Village, Illinois, Copyright 2014 by the American Academy of Pediatrics. All rights reserved. Print ISSN: