Research in Developmental Disabilities 43–44 (2015) 21–31

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Research in Developmental Disabilities

A controlled trial of the SibworkS group program for siblings of children with special needs Rachel M. Roberts a,*, Anastasia Ejova a,b, Rebecca Giallo c, Kate Strohm d, Meredith Lillie a, Belinda Fuss a a

School of Psychology, University of Adelaide, Australia Institute for Research of Children, Youth and Family, Faculty of Social Studies, Masaryk University, Brno, Czech Republic Murdoch Childrens Research Institute, Parkville, Victoria, Australia d Siblings Australia, Australia b c

A R T I C L E I N F O

A B S T R A C T

Article history: Received 5 November 2014 Received in revised form 3 June 2015 Accepted 4 June 2015 Available online 4 July 2015

Siblings of children with a disability are an at risk group for emotional and behavioral problems. This study evaluated an intervention to promote the emotional and behavioral functioning of siblings of children with disabilities and chronic health conditions. SibworkS is a six-week manual-based, cognitive–behavioral group support program focussed on strengthening siblings’ perceived social support, self-esteem, problem-solving skills, adaptive coping behaviors and positive sibling relationships. Fifty-six children aged 7–12 were allocated to either the SibworkS program (n = 30) or waitlist control (n = 26) in alternating sequence. The primary outcome was siblings’ emotional and behavioral functioning. Additional outcomes were self-esteem, perceived social support, the sibling relationship and coping behaviors. Siblings were followed-up immediately after the intervention and at 3-months. Siblings participating in the SibworkS intervention were reported to have fewer emotional and behavioral difficulties than siblings in the control group immediately following the intervention and at the 3-month follow-up. Participation in SibworkS was associated with fewer emotional and behavioral difficulties for siblings. Implications for practice and future research include recommendations for improving program participation. Crown Copyright ß 2015 Published by Elsevier Ltd. All rights reserved.

Keywords: Siblings Intervention Group Special needs Chronic condition Disability

1. Introduction Studies investigating the impact on children of having a sibling with a disability have reported an increased risk of poorer outcomes for this group (Fisman et al., 1996; Giallo & Gavidia-Payne, 2006). Fisman et al. (1996) found that parents reported significantly more behavior problems in siblings of children with pervasive developmental disorder compared to siblings of typically developing children. Similarly, Giallo and Gavidia-Payne (2006) found that parents of these children gave significantly higher ratings of overall behavioral difficulties, in particular emotional and social difficulties, and significantly lower ratings of prosocial strengths, in comparison to normative data. Additionally, a meta-analysis study reported a statistically significant small negative effect on measures of psychological functioning and peer activities for having a sibling

* Corresponding author. Tel.: +61 883135228; fax: +61 088313 3770. E-mail address: [email protected] (R.M. Roberts). http://dx.doi.org/10.1016/j.ridd.2015.06.002 0891-4222/Crown Copyright ß 2015 Published by Elsevier Ltd. All rights reserved.

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with a disability (Rossiter & Sharpe, 2001). This effect was stronger for children compared with adolescents. Overall, these findings suggest that the siblings of children with a disability constitute an at risk group that may benefit from interventions to reduce the likelihood of experiencing these poorer outcomes. Moreover, a recent longitudinal study found that siblings of children with disabilities exhibited the same level of behavioral problems between the ages of 4 and 11, while their peers exhibited a decreasing trend (Giallo et al., 2014). Another longitudinal study conducted over 3 years found that the siblings of children with Pervasive Developmental Disorder demonstrated more adjustment difficulties over time than control children (Fisman, Wolf, Ellison, & Freeman, 2000). These findings further suggest that childhood is a vulnerable period for these siblings and therefore this may be a time during which an intervention might be particularly beneficial. In designing interventions for the siblings of children with disabilities, clinicians have made use of research examining additional risk factors associated with poorer outcomes. Many identified risk-factors are unalterable, such as diagnosis of disability (Fisman et al., 1996), severity of disability symptoms (Meyer, Ingersoll, & Hambrick, 2011; Petalas et al., 2012) and sibling birth order (Hastings, 2003). Interventions have instead focused on addressing alterable risk-factors, such as perceived lack of social support (Benson, Gross, & Kellum, 1999), lack of a warm, non-conflictual sibling relationship (Fisman et al., 1996), and use of maladaptive coping strategies rather than adaptive strategies such as problem-solving (Gamble & McHale, 1989; Giallo & Gavidia-Payne, 2006; Van Riper, 2000). Usually taking the form of group support sessions, informed by cognitive–behavioral therapies and held over a number of weeks, interventions seek to enhance perceived social support by teaching children appropriate strategies for seeking help from a variety of sources. Children are also taught to express both positive and negative emotional states (e.g., Lobato & Kao, 2002). In seeking to strengthen the sibling relationship, interventions provide information about relevant disabilities and teach mutually enjoyable ways of interacting with their sibling (Roeyers & Mycke, 1995). Activities that teach strategies for coping (e.g., relaxation) and problem-solving are incorporated to prevent the use of maladaptive coping strategies (e.g., Strohm & Nesa, 2010). Although several interventions to promote sibling adjustment have been developed and are widely available (e.g., Sibshops; Meyer & Vadasy, 2008), studies evaluating the effectiveness of these interventions are scarce. In a systematic review of interventions to support siblings of children with a disability or chronic illness only 16 studies (5 controlled, 9 uncontrolled) were identified (Hartling et al., 2014). Although study differences made it difficult to conduct the analysis, it was concluded that the higher-quality trials demonstrated benefits such as improved mood and behavioral functioning, and lower anxiety. However, there is a range of limitations of the interventions reported to date. Some interventions have addressed only certain risk factors discussed above, omitting key components such as addressing the sibling relationship or strategies for coping and problems solving. Another issue is that several interventions have not been described in manuals. This not only restricts the extent to which evaluation results can be replicated, but also the extent to which the programs in question could be administered (Chinitz, 1981; Dyson, 1998; Granat, Nordgren, Rein, & Sonnander, 2012; McLinden, Miller, & Deprey, 1991; Phillips, 1999; Smith & Perry, 2005; Williams et al., 2003). Developed to overcome these limitations, the most commonly run intervention for the siblings of children with special needs in Australia is SibworkS (Paior & Strohm, 2009). SibworkS is a manual-based program that is designed to address all of the risk factors discussed above (Strohm & Nesa, 2010). It is delivered to children aged 8–12, who attend six weekly 2-h group sessions. Additionally, parents receive an information sheet and a set of ‘parent tips’ following each session. The aim of this study was to evaluate the efficacy of SibworkS in promoting sibling wellbeing. A review of previous intervention evaluations suggests the need for a control group, as only five studies to date have used a randomised controlled design (Feinberg et al., 2013; Giallo & Gavidia-Payne, 2008; McLinden et al., 1991; Phillips, 1999; Williams et al., 2003). Another desirable design feature is the inclusion of a longer-term follow-up. Only four studies so far have conducted such follow-ups (Granat et al., 2012; Lobato & Kao, 2002; Lobato & Kao, 2005; Williams et al., 2003). Therefore, the present study evaluated the SibworkS program using an alternating allocation controlled design, where children participating in the program were compared to a waitlist control group both immediately post-treatment and 3 months post-treatment. The 3 months follow-up was included to assess maintenance of any improvements over time, as well as to take into account the trajectory of typically developing children declining in behavior problems over childhood, while siblings of children with disabilities do not (Giallo et al., 2014). It was anticipated that, compared to the control group at both evaluation points, the treatment group would show decreased symptoms of internalising (anxiety, depression) and externalising (aggression) problems. Additional outcomes were assessed and it was expected that the treatment group would demonstrate increased self-esteem, increased perceived social support, increased use of adaptive coping behaviors and reduced use of avoidance coping, and improved relationship with their sibling with special needs (increased warmth, reduced conflict). Child and parent satisfaction with the intervention was also examined. 2. Method 2.1. Study design This study was approved by the University of Adelaide Human Research Ethics Committee and the Women’s and Children’s Health Network Human Research Ethics Committee. An alternating allocation controlled trial with two conditions was conducted, where participants were allocated to either the SibworkS program or the waitlist control group in alternating

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order. Evaluation measures were completed by participants and their parents at three time points: (1) prior to the treatment group’s first session (baseline), (2) immediately after the treatment group’s last session (post-treatment), and (3) three months after the treatment group’s last session (follow-up). Once the follow-up questionnaires were received, siblings in the control group were then offered the SibworkS program. 2.2. SibworkS intervention SibworkS is a group-based support program for children aged 8–12 years. It is available from Siblings Australia (http:// siblingsaustralia.org.au/resources.php). Sessions run weekly after school for 2 h, with the program manual also describing an alternative schedule of activities for practitioners wishing to run the program over two full days (Strohm & Nesa, 2010). The activities described in the manual are largely based on cognitive–behavioral therapy principles and relate to (1) getting to know each other and establishing group goals, (2) noting similarities and differences between oneself and one’s sibling, as well as sharing knowledge about various disabilities, (3) expressing feelings and seeking social support, (4) using a six-step problem-solving approach, (5) developing strategies for coping with stresses, including those relating to one’s sibling, and (6) recognising one’s individuality, even in terms of what one has taken away from the program. The parent component of SibworkS consists of Parent Information Sheets provided in the manual. These summarise the main theme of each session (1–6 above) and the rationale for exploring it. They also offer ‘Parent Tips’, which are a mixture of practical tips and advice for encouraging further discussion about the session theme. In this study, four six-week SibworkS groups were run for the treatment group. Each group was attended by 6–8 children. SibworkS is recommended to be facilitated by a clinician with expertise in working with children. The groups were facilitated by a paid group leader, who was a postgraduate clinical psychology student and a Provisionally Registered Psychologist supervised by a Clinical Psychologist. The group leader was assisted by a volunteer who had experience working with children, and, in most cases, experience in supervising outings for the siblings of children with special needs. A research assistant was also present in an administrative capacity. Fidelity to the manual was assessed by the group facilitators and the research assistant following each session. All completed a checklist of the activities prescribed by the manual for that week’s session. 2.3. Participants Forty-two children (25 girls, 17 boys) aged between 7.5 and 12.5 years (M = 9.3, SD = 1.38) completed the baseline evaluation and at least one other assessment point. The majority (N = 36) had a brother or sister with an Autism Spectrum Disorder. Other conditions of the brothers and sisters with a disability or chronic health condition were Angelman’s Syndrome, Down Syndrome, Phelan-McDermid Syndrome, Global Developmental Delay, Pervasive Developmental Disorder, intellectual disability, and Optic Nerve Hypoplasia. The age range of siblings with disabilities was between 4 and 17 years (M = 8.86, SD = 3.45). The difference in age between participants and their siblings ranged from 0 to 7 years, (M = 2.86, SD = 1.42), where 21 participants (48.84%) were older than their relevant sibling with a disability. Participants were recruited through South Australian disability services. A service for people with Autism Spectrum Disorder did not have information on whether their clients had siblings and so notified all parents of clients aged between 5 and 15 of the study (n = 2, 461). A service for children with physical disabilities (e.g., cerebral palsy), mailed study information to 40 families who had expressed interest in sibling programs. Additionally, study information was made available through both organisations’ social media. Information about the study was also made available on the social media pages of Siblings Australia, an organisation providing support and advocacy for siblings. Parents and siblings who showed interest in the program were given an information pack which included separate information sheets and consent forms for parents and children. Families were allocated to the intervention or waitlist control group by a research assistant (alternating allocation) according to the order in which consent forms were received. In two instances parents wished for two of their children to participate, and in these cases both children were allocated to the same group. Parents were then contacted and informed about their allocation. There were three inclusion criteria for participation in the study; (a) the sibling had to be aged between 8 and 12 years with 6 months allowance either side, (b) both sibling and parent consent had to be obtained, and (c) siblings themselves could not have a significant disability or chronic health condition. At all three evaluation time points (baseline, post-treatment and follow-up) measures were sent to families to be completed at home and mailed back. A parent cover letter advised that children should complete their questionnaires independently, and children were provided with their own cover letter and reply-paid envelope. Parents were invited to contact the research assistant for assistance and/or request a house visit from the research assistant, but only two families made the request. See Fig. 1 for the CONSORT diagram describing the recruitment procedure and drop-out of participants at different stages (Schulz, Altman, Moher, & CONSORT Group, 2010). 2.4. Outcome measures Parents completed the following outcome measure on the behalf of participants.

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Fig. 1. Recruitment and drop-out of participants.

2.4.1. Emotional and behavioral functioning The Strengths and Difficulties Questionnaire – Parent Version (SDQ; Goodman, Meltzer, & Bailey, 1998), is a widely used 25item measure of emotional and behavioral functioning. The questionnaire has five subscales: emotional symptoms (e.g., ‘‘Many worries or often seems worried’’), conduct problems, hyperactivity/inattention, peer relationship problems, and prosocial behavior. The Total Difficulties Score is calculated by adding all items (except the prosocial items). Items were scored on a 3-point rating scale ranging from 1 (’not true’) to 3 (’certainly true’). The SDQ has adequate retest stability (mean .62 after 4–6 months) and internal consistency for the Total Difficulties Score (.82) (Goodman, 2001). Total Difficulties Scores can be described as normal (0–13), borderline (14–16) and abnormal (17–40) (Goodman, 2001). For the current study, the Cronbach’s alpha was .87 (baseline alphas are reported in this section). Participants completed the remaining four outcome measures. Parents were instructed not to provide them with assistance during completion. 2.4.2. Perceived social support The Social Support Scale for Children (SSSC; Harter, 1985) is a measure of the social support and positive regard the respondent receives from significant others in their life. The scale consists of 24 items – six for each of the following groups: parents, teachers, classmates and friends. The respondent decides which of two alternative statements best describes their circumstances (e.g., ‘‘Some kids have parents who don’t really understand them’’ vs. ‘‘Other kids have parents who really do

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understand them’’) and then indicates whether the chosen statement is ‘‘really true’’ or ‘‘sort of true’’ for them, yielding a score on a 4-point scale. Total scores for ‘Parent’, ‘Teacher’, ‘Classmates’ and ‘Friends’ subscales are calculated by averaging the relevant items. The total score is the sum of the four mean scores. The internal consistency reliabilities for the subscales range from .72 to .88 (Harter, 1985). For the current study, the Cronbach’s alpha for the subscales were as follows: Parent .60, Teacher .74, Classmates .80 and Friends .69. 2.4.3. Sibling relationship: warmth and conflict The Sibling Relationship Questionnaire (SRQ; Buhrmester & Furman, 1990) is a measure of the quality of the relationship shared between the respondent and their sibling. Two subscales were used for the study. The Warmth/Closeness subscale consists of 15 items (e.g., ‘‘How much do you and this sibling love each other?’’). The other subscale, Conflict, consists of 6 items (e.g., ‘‘How much do you and this sibling insult and call each other names?’’). All items are scored on a scale ranging from 1 (‘hardly at all’) to 5 (‘extremely much’), with averages being calculated to create subscale scores. The SRQ has been used in Australian research (Fox, Barrett, & Shortt, 2002) and has good psychometric properties. Buhrmester and Furman (1985) found it to have good subscale internal consistency (M = .80), fair to good test–retest reliability (.58–.86), and a low correlation (r = .14) with a social desirability measure. For the current study, the Cronbach’s alpha was .89 for the Warmth/ Closeness subscale and .87 for the Conflict subscale. 2.4.4. Coping The Self-Report Coping Scale (SRCS; Causey & Dubow, 1992) is a 34-item measure assessing use of avoidant coping responses and approach-based responses. Avoidant responses include Distancing (e.g., ‘‘Make believe nothing happened’’), Internalising (e.g., ‘‘Go off by myself’’) and Externalising (e.g., ‘‘Yell to let off steam’’). Approach responses include Problem Solving (e.g., ‘‘Try to think of different ways to solve it’’) and Seeking Social Support (e.g., ‘‘Get help from a friend’’). Responses are made on a scale ranging from 1 (never) to 5 (always). Subscale scores are derived from the totals for each coping strategy. Causey and Dubow (1992) found the SRCS to have moderate internal consistency, with alpha coefficients ranging from .68 to .84 for the subscales. For the current study, the Cronbach’s alpha for the subscales were Distancing .75, Internalising .78, Externalising .68, Problem Solving .69 and Seeking Social Support .73. 2.4.5. Self-esteem The Rosenberg Self-Esteem Scale (RSES; Rosenberg, 1989) is a 10-item measure of global self-esteem. Respondents indicate the extent to which they agree with statements such as ‘‘I am able to do things as well as most other people’’, on a scale from 1 (‘strongly agree’) to 4 (‘strongly disagree’). Scores on each item are summed to provide a total score, with negatively worded items (e.g., ‘‘I feel I do not have much to be proud of’’) reverse-scored. The RSES has been found to have good test–retest reliability, ranging from .85 to .88 in a study by Silbert and Tippett (1965). For the current study, the Cronbach’s alpha was .83. 2.5. Additional measures 2.5.1. Sibling characteristics At baseline, parents described the age, gender and diagnosis of their child with a disability. They also completed The Brief Developmental Behaviour Checklist – P24 (DBC-P24; Taffe et al., 2007), on the behalf of this child. This is a 24-item measure of behavioral and emotional problems. The DBC-B24 has excellent specificity and sensitivity if values above a cutoff score (.48) are taken as indicating significant levels of psychopathology (Taffe et al., 2007). For the current study, the Cronbach’s alpha was .79. If parents reported having more than one child with a disability, they were asked to respond in relation to the child with the most severe disabilities. 2.5.2. Participant characteristics and co-interventions At all data collection points, parents were asked whether participants had attended any other support program in the preceding six weeks. Parents answering in the affirmative provided further details. The Australian Bureau of Statistics, Socioeconomic Indexes for Area (SEIFA; Australian Bureau of Statistics, 2006) was used to indicate family socioeconomic status based on their postcode. This index is based on income, unemployment and educational achievement by area. Higher scores reflect higher socioeconomic status (Australian mean = 1000, SD = 100). 2.5.3. Qualitative measure: ‘‘What I’ve learned’’ One of the last activities of the program’s final session (Week 6) involved listing three things learned during SibworkS. Responses were recorded by the research assistant. 2.5.4. Satisfaction with SibworkS Following their last SibworkS session, participants and their parents completed a brief 6-item satisfaction survey adapted from those used by Marsland et al. (2013) e.g., ‘‘To what extent has participation in the program helped you [your child]?’’ Responses were given on a 4-point scale, where 1 indicated high satisfaction and 4 indicated low satisfaction. For the current study, the Cronbach’s alpha was .77 for the child version and .87 for the parent version.

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2.6. Data analysis Independent sample t-tests and Chi-square analyses were used to test for differences between the treatment and control groups on demographic variables and evaluation measures. A series of single-factor between-subjects analysis of covariance (ANCOVA) were conducted to compare the intervention and control groups on each of the evaluation measures at postintervention and the 3-month follow-up, controlling for the baseline scores. Effect sizes in the form of partial h2 were reported, with effect sizes reported as small (.01), medium (.06) and large (.14), respectively (Cohen, 1969 as cited in Algina & Olejnik, 2003; Haase, Waechter, & Solomon, 1982; Richardson, 2011). 3. Results Table A1 (see appendices) illustrates the patterns of missing data among all participants (N = 50) who returned at least one set of questionnaires. Data for these participants was not missing completely at random, as indicated by Little’s MCAR Test on all outcome measures and participant and family characteristics (x2(551) = 4,079, p < .001). However, margin plots showing the distributions of present and missing values when two variables are plotted against each other (van Buuren & Groothuis-Oudshoorn, 2011) indicated that socioeconomic status and the behavioral and emotional problems of the sibling with a disability (DBC scores) did not predict missingness in any of the outcome measures over time. Analyses of baseline characteristics and evaluation measures across groups included only those participants who returned their baseline questionnaire and one other (N = 42). For each baseline characteristic and evaluation measure, only complete cases were included. 3.1. Sample characteristics Baseline characteristics of the sample are presented in Table 1. The child with a disability was rated by parents as having significant levels of psychopathology with the mean DBC-P24 score for both groups well above the clinical cut off of .48. Notably, 60 percent of participants were ‘borderline’ or ‘abnormal’ with respect to emotional and behavioral functioning as measured by the SDQ. In terms of demographics, the treatment group had more males (x2(1) = 3.80, p = .05) and had more members who had taken part in some other support program in the preceding 6 weeks (x2(1) = 4.55, p = .03). The sample was of average socio-economic status, compared with the Australian population. Descriptive statistics for the treatment and control groups on the evaluation measures at baseline, post-intervention, and follow-up are presented in Table 2. At baseline, the treatment group reported higher levels of coping through problemsolving (t(39) = 2.12, p = .04, d = .68), perceived social support from parents (t(39) = 2.39, p = .02, d = .77), perceived social support from teachers (t(38) = 2.65, p = .01, d = .86), and perceived social support from friends (t(39) = 2.13, p = .04, d = .68). The ANCOVAs used in assessing intervention outcomes took these baseline differences on evaluation measures into account (Van Breukelen, 2006). 3.2. Attendance and fidelity Participants attended an average of 5.4 out of six sessions (SD = .73). Fidelity ratings from the group facilitators and research assistant indicated that on average 94% of activities were completed across the six sessions with 86% of incomplete ratings indicating a single activity was omitted. Of the remaining 13.64%, two ratings omitted two activities and one rating omitted three activities, although two of these were at least partially completed. Most comments concerning incompletion related to late arrival of participants, late set up, and participant preference for a different activity to the one scheduled by the manual (e.g., outdoor ball game instead of poster work). Other comments related to participant over-engagement in one activity to the neglect of another, participant disinterest in the closing activity due to parents waiting inside the program venue, and participant behavioral issues.

Table 1 Participant characteristics at baseline. Demographics, sibling characteristics and co-interventions

Treatment group N = 22

Waitlist control group N = 20

Gender (number of boys:girls) Attended other support programs in the preceding 6 months SESa Have one or more other siblings (without a disability) Have additional siblings with a disability Older than sibling with a disability Developmental Behaviour Checklist total for sibling with a disability Mean score on all items (clinical cutoff = .48)

12:10 41% Median: 1039.5, IQR: 946–1067 27% 0% 50% M (SD): 20.23(8.28) .84

5:15 15% Median: 1007, IQR: 918–1057 55% 10% 50% M (SD): 18.45(5.93) .77

a

Australian Bureau of Statistics Index of Socioeconomic Disadvantage by Suburb (Australian Bureau of Statistics, 2006). IQR, interquartile range.

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Table 2 Descriptive statistics for three evaluation points for all participants and participants who provided data at follow-up (N in brackets). Measure

Participants contributing to data

Treatment group

Baseline

Emotional and behavioural functioning-SDQ

Social support

All participants Participants who provided data at follow-up All participants Participants who provided data at follow-up

Sibling relationship: warmth

All participants Participants who provided data at follow-up

Sibling relationship: conflict

All participants Participants who provided data at follow-up

Coping: Social support

All participants Participants who provided data at follow-up

Coping: Problem solving

All participants Participants who provided data at follow-up

Coping: Distancing

All participants Participants who provided data at follow-up

Coping: Internalising

All participants Participants who provided data at follow-up

Coping: Externalising

All participants Participants who provided data at follow-up

Self-esteem

All participants Participants who provided data at follow-up

Control group

Post-

M

SD

M

SD

15.4 (20) 15.1 (14)

8.21

12.5 (20)

6.74

13.6 (21) 14.1 (15)

3.2 (22) 3.3 (15)

2.9 (22) 3.1 (15)

24.1 (22) 25.1 (14)

25.5 (22) 26.7 (14)

17.3 (22) 17.1 (14)

19.6 (22) 18.7 (14)

8.8 (22) 7.7 (14)

20.7 (22) 21.4 (15)

7.02

1.72

13.4 (21)

3.2 (22)

3.3 (15)

2.7 (22)

26.7 (14)

18.3 (22)

17.6 (14)

19.5 (22)

18.4 (14)

8.2 (22)

4.88

7.7 (14)

21.6 (22)

4.46

4.38

2.84

3.77

2.73

5.56

6.76

5.84

6.14

3.83

1.11

5.24

4.38

5.56

.75

6.6

5.38

4.93

1.59

6.04 25.9 (14)

24.1 (22)

4.05

1.02 3.1 (15)

24.3 (22)

SD

.81

5.43

5.59

M

13.8 (15)

1.25

6.03

M

1.58

.79

1.16

Baseline

10.71 (14)

1.47

.80

Follow-up

2.52

5.39 22.3 (15)

3.64

14.4 (19) 14.4 (19)

PostSD

Follow-up

M

SD

14.37 (19)

6.81

8.19

12.4 (18) 12.5 (17)

1.77

2.8 (18) 2.8 (17)

.67

2.7 (18) 2.7 (17)

1.03

21.8 (18) 21.6 (16)

6.23

21.7 (18) 22.1 (16)

4.39

16.7 (18) 16.0 (16)

5.81

18.8 (18) 18.4 (16)

5.42

6.7 (18) 7.9 (16)

3.36

18.7 (18) 19.1 (17)

4.86

11.9 (18)

4.38

2.6 (17)

1.04

21.0 (16)

7.78

22.1 (16)

7.29

15.6 (16)

4.38

19.2 (16)

3.82

8.5 (16)

4.23

19.71 (17)

5.50

5.05

3.11

18.4 (18)

.74

5.25

5.64

9.7 (18)

2.9 (17)

5.28

5.79

19.4 (18)

2.00

6.08

4.40

16.6 (18)

12.6 (17)

7.17

6.54

21.0 (18)

7.13

1.08

1.04

20.9 (18)

14.0 (19)

.71

.68

2.5 (18)

SD

2.23

1.81

2.9 (18)

M

4.77

3.3. Post-intervention The ANCOVAs revealed that, after participation in SibworkS, the treatment group displayed significantly fewer emotional and behavioral difficulties as measured by the parent-completed SDQ. The associated effect size was medium to large. The treatment group reported less coping through externalising with a medium effect size, but this was not statistically significant. Also, the treatment group reported greater self-esteem with a medium effect size, although once again this was

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Table 3 ANCOVA results: F-value, p-value and effect size. Follow-up

Post-intervention

Emotional and behavioral functioning (SDQ) Social support Sibling relationship: Warmth Sibling relationship: Conflict Coping: Social support Coping: Problem solving Coping: Distancing Coping: Internalising Coping: Externalising Self-esteem

h

F(df)

p

p

F(1,36) = 4.92

.03

F(1,36) = 1.84 F(1,37) < 1 F(1,37) < 1 F(1,37) = 1.16 F(1,37) < 1 F(1,37) < 1 F(1,37) < 1 F(1,37) = 2.88 F(1,37) = 2.23

2

h2

F(df)

p

p

.12

F(1,30) = 6.20

.02

.17

.18

.05

.29

.03

.30 .26 .31 .31

.04 .05 .04 .04

.10 .14

.07 .06

F(1,29) < 1 F(1,29) < 1 F(1,29) = 1.10 F(1,27) = 1.35 F(1,27) = 1.06 F(1,27) = 1.08 F(1,27) < 1 F(1,27) < 1 F(1,29) < 1

Table 4 Participants’ responses when asked to list three things they learned. Response

Examples

n

Social support: Group peers

‘‘Lots of other people are in my situation’’ ‘‘There are other people who have a sibling with disabilities who I can talk to’’ ‘‘I made some new friends’’ ‘‘We are all different’’ ‘‘Laughter yoga’’ ‘‘How to calm down in difficult situations’’ ‘‘How to handle problems’’ ‘‘Lots of ways to resolve things with brother and sister’’ ‘‘Problem-squashing’’ ‘‘There are other people with disabilities’’ ‘‘New words: difference between Aspergers and Autism’’ ‘‘Not all brothers and sisters [with autism] are the same. Their obsessions are different.’’ ‘‘Tolerance’’ ‘‘Differences and similarities’’ ‘‘How other people feel’’ ‘‘How to be more sociable’’ ‘‘Support networks’’

12

Coping Problem-solving

Sibling relationship: Knowledge of disability

Sibling relationship: Similarities and differences with siblings Social support: Emotional literacy Social support: Existing supports

12 12

5

2 2 1

not statistically significant. All results are presented in Table 3. It is noteworthy that an almost identical set of results was obtained following the removal of outliers.1 3.4. Follow-up The significant large effect for emotional and behavioral functioning was maintained at three-month follow-up. The ANCOVA with parent-completed SDQ as the dependent variable (with outliers removed) indicated that the treatment group continued to display fewer emotional and behavioral difficulties, the effect size being large. The medium effects observed for self-esteem and externalising at post-intervention were not maintained at follow up. 3.5. Qualitative analysis: ‘‘What I’ve learned’’ Table 4 presents participants’ responses to identifying three things they had learned from the program. Thematic analysis was conducted and responses were grouped according to themes. Three topics were mentioned most frequently – social support from peers from the group, coping and problem-solving. 3.6. Satisfaction with the intervention Both parents and children reported high levels of satisfaction with the program (Table 5).

1 This was done by calculating the Cook’s distances for each ANCOVA and removing those cases that had a Cook’s distance greater than 3 times the mean of the Cook’s distances: http://www.mathworks.com.au/help/stats/cooks-distance.html.

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Table 5 Child and parent satisfaction with SibworkS. Item

Response

1. To what extent has participation in the program helped you [your child]? 2. How would you rate the whole program [from a parent’s perspective]? 3. Would you tell a friend that they [their child] should take part in this program? 4. Have the things you have [your child has] learned in this program helped you to [him/her] deal with upsetting events (stress) in your [his/her] life? 5. Are you happy with the help you [your child has] received in the program? 6. Did you [your child seem to] enjoy the sessions?

A great deal/Somewhat Excellent/Good Yes, definitely/Yes, I think so

90 90 90

85 95 100

Yes, they helped a great deal/Yes, they helped somewhat Yes, very happy/Yes, mostly happy

90

85

100

95

95

100

Yes, definitely/Yes, I think so

Child %

Parent %

4. Discussion This randomised controlled trial of SibworkS is among the few efficacy trials of a sibling intervention conducted to date. Despite the modest sample size, this trial reported evidence for the effectiveness of this manual-based program to improve emotional and behavioral functioning of the siblings of children with disabilities. Immediately after the program, parents of children participating in SibworkS reported improved emotional and behavioral functioning with a medium to large effect size. This finding was maintained 3 months after program participation, suggesting it to be a robust intervention effect. Additionally, the program attendees themselves reported non-significant improvements of medium effect size in the form of enhanced self-esteem and less avoidant coping (externalising) at post-intervention. However, these findings were not maintained at follow-up. This study suggests that the approach used in designing the SibworkS program, namely to address a wide range of risk factors in a manualised program, with structured supporting material provided to parents, results in an effective intervention. The controlled design enabled a comparison with outcomes of children not receiving the program, and the 3 months follow-up enabled assessment of longer-term effects for children after the program had ended. These findings highlight the potential this program has to address the poorer psychosocial outcomes experienced by siblings of children with disabilities. Measurement issues are likely to have played a significant role in the pattern of findings, and these have several implications for both interpreting the finding reported here, as well as for the design of future studies. Emotional and behavioral functioning, as reported by parents, displayed treatment effects most strongly and consistently. Given the program’s multiple aims and use of cognitive–behavioral therapy principles, similar findings were anticipated for the study’s other outcomes, including social support, sibling relationship, coping, and self-esteem. However, issues related to the measurement of these outcomes may have affected results. It is possible that the children’s self-reports underestimated their difficulties at baseline. The siblings of children with disabilities have been found to report less pronounced difficulties at baseline compared to parents and direct observation by researchers (Rossiter & Sharpe, 2001). This may reflect siblings downplaying their difficulties so as not to add to parental stress, or to give the impression of being the ‘good’ child in the family. Conversely, given that none of the self-report measures produced significant findings, reports of improved emotional and behavioral functioning could merely reflect parents’ investment in the intervention to observe improved outcomes for their child. Parents were informed of the program’s aims prior to their child’s participation. Therefore, whether their child benefited from the program or not, parents may have reported observing such improvements. Both of these interpretations suggest that prospective evaluations of SibworkS would benefit from the use of outsider reporters, such as teachers or blind assessors, to assess intervention effects. The parent-report version of the SDQ used by this study can also be completed by teachers (Goodman et al., 1998). Outcomes such as the sibling relationship and coping strategies could be assessed through observer-rated videotaped interactions (Feinberg et al., 2013). Another limitation of the current study was the low response rate by families to invitations to participate. Ways to make the program more accessible to some of the 2500 families originally invited into this study need to be considered. One way to do this may be to offer SibworkS in the two-day format described in the manual. The majority of siblings who participated in this study were rated by their parents as experiencing significant emotional and behavioral difficulties at baseline. It is likely that parents who were concerned about their children consented for them to participate. Given that this program is designed as a universal prevention program, offering the program in a different format may encourage more parents to enroll their children, including those without current clinically significant difficulties. Finally, in relation to study design, the study would have been stronger had we conducted the random allocation after the families had returned the baseline questionnaires. Research with a larger sample would make it possible to test the medium-sized effects of treatment reported by children but not found to be statistically significant in the current study. A larger sample would also enable the in-depth investigation of the response patterns that emerged from the qualitative data. Comments about group peers were prominent in participants’ responses about ‘what they learned’ from the program. This pattern suggests that group cohesion contributes to the program’s effectiveness. A measure of group cohesion could be included alongside ‘program satisfaction’ measures to test this assertion (e.g., Evans & Jarvis, 1986).

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4.1. Implications and conclusions This study provides much needed evidence for the efficacy of a group-based intervention for siblings of children with disabilities. This is the most frequently used intervention in Australia with this group of children, but to date the lack of evidence to support its use has hindered the take-up of the program. Challenges ahead include the need for more research to examine the effectiveness of the program when delivered by professionals in disability agencies – the most common location for the delivery of the program. Increasing the accessibility of the program for families, given the low take up of the 6-week format also needs consideration. A two-day school holiday program is one option. Online delivery of the program could also be considered, given the positive outcomes reported in an evaluation of a siblings program delivered using a combination of workbooks and telephone support (Giallo & Gavidia-Payne, 2008). Future research to inform the wider dissemination and scalability of the program is now needed. Considering ways to improve the accessibility of interventions such as SibworkS is an important next step toward promoting and improving the emotional and behavioral functioning of siblings of children with special needs. Acknowledgement This work was supported by a Channel 7 Children’s Research Foundation Grant 13685.

Appendix A Table A1. Table A1 Missing data across all outcome measures at baseline, post-intervention and follow-up. Baseline

Post-intervention

Follow-up

Number of instances

SS SR: SR: C: C: C:D C:I C:E SE SDQ SS SR: SR: C: C: C:D C:I C:E SE SDQ SS SR: SR: C: C: C:D C:I C:E SE SDQ W C SS PS W C SS PS W C SS PS 1 1 1 1 1 1 1 1 0 0 1 0 1

1 1 1 1 1 1 1 1 0 1 1 0 1

1 1 1 1 1 1 1 1 0 1 1 0 1

1 1 1 1 1 1 1 1 0 1 1 0 1

1 1 1 1 1 1 1 1 0 1 1 0 1

1 1 1 1 1 1 1 1 0 1 1 0 1

1 1 1 1 1 1 1 1 0 1 1 0 1

1 1 1 1 1 1 1 1 0 1 1 0 1

1 1 1 1 1 1 1 1 0 1 1 0 0

1 1 1 1 1 1 1 1 0 1 1 0 1

1 1 0 1 1 1 1 0 1 0 0 0 0

1 1 0 1 1 1 1 0 1 1 0 0 0

1 1 0 1 1 1 1 0 1 1 0 0 0

1 1 0 1 1 1 1 0 1 1 0 0 0

1 1 0 1 1 1 1 0 1 1 0 0 0

1 1 0 1 1 1 1 0 1 1 0 0 0

1 1 0 1 1 1 1 0 1 1 0 0 0

1 1 0 1 1 1 1 0 1 1 0 0 0

1 1 0 1 1 1 1 0 1 1 0 0 0

1 1 0 1 0 1 1 0 1 1 1 1 0

1 0 0 0 0 1 1 1 1 0 1 1 0

1 0 0 0 0 1 1 1 1 0 1 1 0

1 0 0 0 0 1 1 1 1 0 1 1 0

1 0 0 0 0 1 0 1 1 0 0 1 0

1 0 0 0 0 1 0 1 1 0 0 1 0

1 0 0 0 0 1 0 1 1 0 0 1 0

1 0 0 0 0 1 0 1 1 0 0 1 0

1 0 0 0 0 1 0 1 1 0 0 1 0

1 0 0 0 0 1 1 1 1 0 1 1 0

1 0 0 1 0 0 1 1 1 0 1 1 0

28 5 5 2 2 1 1 1 1 1 1 1 1

Note: SS, social support; SR:W, sibling relationship: warmth; SR:C, sibling relationship: conflict; C:SS, coping: social support; C:PS, coping: problem solving; C:D, coping: distancing; C:I, coping: internalising; C:E, coping: externalising; SE, self-esteem; SDQ, Strengths and Difficulties Questionnaire.

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