Rehabilitation Psychology 2015, Vol. 60, No. 1, 1-16

© 2014 American Psychological Association 0090-5550/15/$ 12.00 http://dx.doi.org/10.1037/rep0000018

Emotional Health and Coping in Spina Bifida After Goal Management Training: A Randomized Controlled Trial Jan Stubberud

Donna Langenbahn

Sunnaas Rehabilitation Hospital, Nesoddtangen, Norway

New York University School of Medicine

Brian Levine

Johan Stanghelle and Anne-Kristine Schanke

Baycrest Centre, Toronto, Canada, and University of Toronto

Sunnaas Rehabilitation Hospital, Nesoddtangen, Norway, and Oslo University

Objective: Executive function impairments are common after spina bifida (SB) and potentially have a detrimental effect on the individual’s emotional health and coping. Goal management training (GMT) is a cognitive rehabilitation method for improving executive function. The purpose of this study was to determine the efficacy of GMT on aspects of perceived emotional health and coping in individuals with SB. Method: Thirty-eight adult subjects with SB were included in this randomized controlled trial. Inclusion was based upon the presence of executive functioning complaints. Experimental subjects (n = 24) received 21 hr of GMT, with efficacy of GMT being compared with results of subjects in a wait-list condition (n = 14). Four self-report questionnaires assessing emotional health and coping were utilized as outcome measures. All subjects were assessed at baseline, postintervention, and at 6-month follow-up. Results: Findings indicated positive effects of GMT relative to the control group on measures of emotional health. Of note, the GMT group showed significant improvement, compared with control subjects, on a self-report inventory of depressive and anxiety symptoms after training, lasting at least 6 months posttreatment. Furthermore, both groups showed improvements after training on mental health components of health-related quality of life. Finally, the GMT group showed a significant increase in task-focused coping and a decrease in avoidant coping after training compared with pretreatment baseline assessment scores. Conclusions: Overall, findings indicate that by us a compensatory intervention to manage executive dysfunction, effective and lasting benefits can be achieved with regard to aspects of perceived emotional health and coping. Keywords: executive functioning, cognitive rehabilitation, brain injury, goal management, evidencebased

Impact and Implications • The present article reports a subset of data from the only study that has addressed treatment of executive dysfunction in adults with spina bifida, demonstrating that a cognitive rehabilitation method originally developed

This article was published Online First December 15, 2014. Jan Stubberud, Sunnaas Rehabilitation Hospital, Nesoddtangen, Nor­ way; Donna Langenbahn, Rusk Institute of Rehabilitation Medicine, New York University School of Medicine; Brian Levine, Rotman Research Institute, Baycrest Centre, Toronto, Canada, and Departments of Psychol­ ogy and Medicine, University of Toronto; Johan Stanghelle and AnneKristine Schanke, Sunnaas Rehabilitation Hospital and Medical Faculty, Oslo University. This work was supported by the South-Eastern Norway Regional Health Authority (Grant Number 2011041). We thank all patients, caregivers, and staff, especially Astri Andersen, Lisbeth Brpndberg, Gunnar Riemer, Lena Haugen, Karen Grimsrud, Brede Dammann, Pal-Erik Plaum, Helene Barder, Per Frydenborg, and Solveig Skou, who helped to carry out the study. We also thank Marianne Lpvstad and Jan Egil Nordvik for discus­ sions concerning this study. Correspondence concerning this article should be addressed to Jan Stubberud, PhD, Sunnaas Rehabilitation Hospital-Bjoernemyrveien 11, Nesoddtangen, Aker, 1450, Norway. E-mail: [email protected]

for patients with acquired brain injury may also be used effectively to enhance emotional health for individuals with congenital brain dysfunc­ tion. • This study adds to the literature by demonstrating that aspects of perceived emotional health and coping skills could be improved by means of a cognitive rehabilitation approach for improving executive function. • Goal management training should be used with adults with spina bifida to not only manage executive dysfunction, but also to improve perceived emotional health and coping.

Introduction Spina bifida with myelomeningocele (SBM), accounting for ~90% of all spina bifida (SB) cases (Detrait et al., 2005), is a congenital neural tube defect that affects the central nervous sys­ tem (CNS). Most individuals with SBM have primary brain dysmorphology along with secondary CNS disturbances resulting from hydrocephalus (Mitchell et al., 2004). Thus, SBM has a multisystemic impact on physical, social, cognitive, behavioral, and emotional functioning (Dennis & Barnes, 2010; Fletcher & Brei, 2010). Emotional health and coping problems are more common in chronic illness populations than in the normal popu­ lation (Perry, Presley-Cantrell, & Dhingra, 2010), and this dispar-

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STUBBERUD ET AL.

ity holds true for SBM as well. SBM has been associated with an increased risk for depression, low self-worth, suicidal ideation, anxiety, and lower levels of health-related quality of life (HRQoL; Abresch, McDonald, Widman, McGinnis, & Hickey, 2007; Ammerman et al., 1998; Appleton et al., 1997; Barf, Post, Verhoef, Gooskens, & Prevo, 2010; Beilin et al., 2010; Kalfoss & Merkens, 2006; Verhoef et al., 2007). Emotional health outcomes in SBM are probably related to a number of factors, including pain, social prejudice, family satis­ faction, perceived competence (e.g., based on physical appear­ ance) and social support, inadequate coping strategies, neuroanatomical organization and development, cognitive deficits, and executive functioning (EF) impairment (Ammerman, Nortz, Ris, & Walz, 2006; Barf et al., 2010; Beilin et al., 2010; Curran, Ponsford, & Crowe, 2000; Gould, Ponsford, & Spitz, 2013; Hellemans, Sliwowska, Verma, & Weinberg, 2010; Price & Drevets, 2010). Results from a study by Kelly et al. (2012) suggest that EF accounts for the relationship between group status (i.e., having SB or not) and how youth with SB function psychologically. Specif­ ically, the results indicated that EF explained the relationship between group and depressive symptoms as reported by mothers. In 2010, Barf et al. reported on 40 youth (mean age 16.0 years) and 13 young adults (mean age 26.6 years) with SBM. They found that EF was significantly associated with quality of life (QoL). These findings suggest that implementing interventions that enhance EF can contribute to improve emotional health outcomes. Executive functioning is regularly defined as a multidimen­ sional construct including higher-order cognitive processes that control and regulate a variety of cognitive, emotional, and behav­ ioral functions (Lezak, 1995; Miyake et al., 2000; Stuss, 2011). Executive functioning in adults with SBM has been investigated in relatively few studies. However, EF impairments are common after SBM and affect the ability to function independently in daily life (Iddon, Morgan, Loveday, Sahakian, & Pickard, 2004; Kelly et al., 2012; Rose & Holmbeck, 2007; Tuminello, Holmbeck, & Olson, 2012). As the challenges of early adulthood and adoles­ cence emerge in terms of increased demand for independent prob­ lem solving, the impact of executive dysfunction increases. In fact, effective EF appears to predict successful achievement of several adult milestones in SBM such as leaving home, attending college, and engaging in social relationships (Zukerman, Devine, & Hol­ mbeck, 2011). Several autonomy-related outcomes in individuals with SBM have also been associated with EF (Heffelfinger et al., 2008; Roebroeck et al., 2006; Tarazi, Zabel, & Mahone, 2008). Furthermore, attention and EF deficits have been found to be predictive of social adjustment difficulties (Rose & Holmbeck, 2007). In summary, because skills incorporated by the construct of EF, and in particular attentional control, are implicated in a variety of day-to-day tasks, EF problems may constitute a significant hindrance to the acquisition of independent living skills, and gaps in achieving the functional expectations of early adulthood may predispose adults with SBM to poor emotional health and impaired coping capacity (Ammerman et al., 1998; Compas, Connor-Smith, Saltzman, Thomsen, & Wadsworth, 2001). Therefore, rehabilita­ tion efforts targeted at improving EF can make a substantial difference in patients’ overall emotional health and coping skills. Although problem-solving interventions are aimed primarily at cognitive deficits in individuals with acquired brain injury (ABI), these therapies often target not only cognitive components of

postinjury functioning, but also emotional factors that interfere with cognitive functioning. These so-called cognitive-behavioral therapy (CBT) techniques can improve aspects of emotional health and coping in populations with disability (e.g., facilitate emotional regulation, reduce impulsive responses, provide options to ady­ namic behavior, and increase acceptance of problems as part of life; Dams-O’Connor & Gordon, 2013). Previous research sug­ gests that providing CBT strategies addressing emotional self­ regulation (D’Zurilla & Goldfried, 1971; D’Zurilla & Nezu, 2001) before initiating compensatory strategy training for executive dys­ function results in improvements in self-ratings of problem solv­ ing, emotional self-regulation and clear thinking, and observations of role-plays of everyday problem situations, which were main­ tained 6 months posttreatment (Rath, Simon, Langenbahn, Sherr, & Diller, 2003). There is growing convergent evidence that structured groupbased compensatory interventions involving problem solving that have incorporated training in “stop-and-think” strategies can pro­ duce significant benefits for EF (e.g., Cicerone et al., 2011; Levine et al., 2000; Miotto, Evans, de Lucia, & Scaff, 2009; Rath et al., 2003; Spikman, Boelen, Lamberts, Brouwer, & Fasotti, 2010; von Cramon, Matthes-von Cramon, & Mai, 1991). The compensatory strategy training used in the present study is goal management training (GMT; Levine et al., 2000; Robertson, 1996). GMT is an EF intervention that draws upon theories concerning goal processing and sustained attention (Robertson & Garavan, 2004; Robertson & Levine, 2013). In GMT, sustained attention and alerting techniques are included within a larger metacognitive intervention to support and enhance EF (Chen et al., 2011; Fletcher et al., 1996; Levine et al., 2011; Robertson & Murre, 1999). This theory positing the importance of sustained attention then leads to interventions that attempt to raise awareness of attentional errors through use of participants’ examples of real-life attention deficits, in-session practice on laboratory and real-life tasks, and homework assignments. The primary objective of GMT is to train participants to stop ongoing behavior to define goal hierarchies (stop-andthink) and monitor performance (Levine et al., 2011). GMT has received empirical support in various adult populations (e.g., normal aging, ABI, and attention deficit hyperactivity disorder; In de Braek, Dijkstra, Ponds, & Jolles, 2012; Levine et al., 2000; Miotto et al., 2009; Novakovic-Agopian et al., 2011; van Hooren et al., 2007). To our knowledge, only one study has addressed the treatment of EF problems in adults with SBM. In a randomized trial including 38 individuals with SBM, Stubberud et al. (2013, 2014) found that compared with participants in a wait-list condition, partic­ ipants who had received GMT showed significant improvement on neuropsychological and self-report measures of EF, and in particular on measures of attentional control (Stubberud, Langenbahn, Levine, Stanghelle, & Schanke, 2013, 2014). Attentional control is a central aspect of EF and refers to a general capacity to exert effort and volition in the regulation of attention (Posner, 1995; Posner & Rothbart, 2007). The extent to which one is able to manage attentional control may have implications for aspects of emotional health (e.g., impact of symptoms of anxiety and depres­ sion, perception of one’s QoL, and capacity for emotional self­ regulation; Rueda, Posner, & Rothbart, 2004; Ruff & Rothbart, 1996) and coping capacity (Anson & Ponsford, 2006; Campbell et al., 2009; Compas & Boyer, 2001; Compas et al., 2001). In this study, coping refers to “the constantly changing cognitive and behavioral efforts to

COGNITIVE REHABILITATION IN SPINA BIFIDA

manage specific external and/or internal demands that are appraised as taxing or exceeding the resources of the person” (Lazarus & Folkman, 1984, p.141). Individuals with impaired EF and attentional control capacities are more likely to adopt avoidance-oriented as opposed to approach-oriented coping, and thus may be at risk for adjustment difficulties (Krpan, Levine, Stuss, & Dawson, 2007; Matthews & Wells, 1996; Spitz, Schonberger, & Ponsford, 2013). Clearly, effec­ tive coping styles are important for stable life management (Carver, Scheier, & Weintraub, 1989; Finset & Andersson, 2000; Lewis & Kliewer, 1996). The practical theme of sustained attention runs continuously through GMT, and is reinforced through mindfulness training (Kabat-Zinn, 1990), which is intended to enhance attentional con­ trol (Tang et al., 2007). In the GMT model, sustained attention skills amplify one’s capacity for self-regulation (Shapiro & Schwartz, 2000) by maintaining focus on the intended goal or redirecting focus back to the intended goal via the experience of the present moment (Davidson et al., 2003; Haidt & Rodin, 1999; Teasdale, Segal, & Williams, 1995). Only two GMT studies (i.e., In de Braek et al., 2012; Van Hooren et al., 2007) have used outcome measures of emotional health and coping when evaluating efficacy, with mixed results. Although it is difficult to determine the essential aspects of mul­ tifaceted interventions (e.g., Donker, Griffiths, Cuijpers, & Chris­ tensen, 2009), components of emotional health have been specif­ ically addressed in more comprehensive cognitive rehabilitation programs (e.g., Ben-Yishay & Diller, 2011; Prigatano, 1999; Prigatano & Fordyce, 1986), as well as in studies that have incorporated problem solving (Rath et al., 2003) and GMT tech­ niques (Spikman et al., 2010; Winocur et al., 2007) in ameliorating EF deficits. However, it is not possible to draw any conclusions about the specificity of these effects to the GMT portion of the rehabilitation programs as assessments were taken before and after programs that also encompassed problem-solving therapy (Spik­ man et al., 2010), and psychosocial and memory training (Winocur et al., 2007). In view of the high incidence of emotional distress and coping difficulties in SBM, the present study sought to investigate the efficacy of GMT on aspects of perceived emotional health (i.e., psychological distress, emotional regulation, and HRQoL) and coping in individuals with SBM. Considering the influence of EF in emotional health and coping, and that GMT has been demon­ strated to enhance EF, and especially attentional control, in indi­ viduals with SBM (Stubberud et al., 2013, 2014), GMT may also have positive benefits for the above-described aspects of emotional health and coping. It was anticipated that GMT techniques, with CBT elements, would provide individuals with more effective resources for dealing with disability-related challenges and, con­ sequently, improve aspects of perceived emotional health and use more effective coping strategies. In the main study (Stubberud et al., 2013), primarily cognitive outcome measures were used when investigating the efficacy of administering GMT as a group-based treatment program for per­ sons with SBM and EF deficits. However, with increased numbers of individuals with SBM surviving into adulthood, it is paramount to address and support both cognitive and emotional outcomes. As such, the present article reports a subset of data from Stubberud et al. ’s (2013) study, addressing perceived emotional health and coping. The current article addresses several areas with a lack of

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research knowledge. First, we believe that no studies have inves­ tigated the effect of cognitive rehabilitation on emotional health in adults with SBM. Second, the evidence related to long-term effects of GMT is weak, particularly in terms of effects on measures pertaining to perceived emotional health. Third, to our knowledge, no studies have published findings investigating the effect of GMT alone on coping style. When GMT is effective, it is assumed that this is as a result of underlying alterations in brain networks supporting sustained attention (Chen et al., 2011; NovakovicAgopian et al., 2011). Reliable goal-directed behavior requires the capacity to sustain attention over time. Targeting sustained atten­ tion may, therefore, lead to improvements in functioning that transfer to broader domains of goal-directed functioning. As such, we hypothesized postintervention changes in scores to reflect perceived improved emotional health as indicated by a decrease in psychological symptoms, and better emotional regulation and HRQoL. Finally, we hypothesized that participants would increase task-focused coping and decrease avoidant coping after interven­ tion.

Method The study was approved by the Regional Ethic Committee for Medical Research Ethics (2009/2188b), South-Eastern Norway. All patients gave informed consent for participation. The research was completed in accordance with the Helsinki Declaration.

Participants and Procedures Thirty-eight participants diagnosed with SBM were recruited from TRS national resource center for rare disorders, Sunnaas Rehabilitation Hospital (Norway). The sample (58% female) had a mean age of 32 years (SD = 8.3), ranging from 19 to 45 years. All participants were White, and spoke Norwegian as their primary language. Table 1 shows the characteristics of both groups. No participants reported prior involvement with a neuropsychological rehabilitation program. Figure 1 shows the flow of participants through trial phases. All patients diagnosed with SBM (19-45 years) and registered in 2010 at the resource center (TRS) were requested to participate in the current study (n = 201). The information letter specifically solicited participants with subjective complaints of executive dys­ function. Accompanying the letter was a self-report questionnaire, the Behavior Rating Inventory of Executive Function-Adult Ver­ sion (BRIEF-A; Gioia, Isquith, Guy, & Kenworthy, 2000), which respondents were asked to complete and return. In addition to the reported difficulties from the information letter, inclusion of pa­ tients was also based upon an elevated score (T > 60) on at least one of the subscales constituting the BRIEF-A. However, the presence of emotional distress or impaired coping skills was not a prerequisite to participation in the study. Criteria for exclusion, based on review of medical records and baseline measures, included impaired essential linguistic, perceptual, or motor function that would interfere with capacity to participate in training. Additionally, patients with Axis I psychiatric disorders or IQ below 70, based on baseline IQ testing, were excluded. Six subjects met exclusion criteria; 4 had IQ below 70, and two had Axis I disorders. Eight additional subjects met inclusion criteria, but could not participate because of somatic illness and/or hospitalization, or

STUBBERUD ET AL.

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Table 1 Means (and SDs) o f Demographic Variables as Well as Cognitive Functioning, Emotional Health, and Coping at Baseline

Age Gender (M = men, F = female) Hydrocephalus n (%) Arnold Chiari malformation n (%) MMC level n (%) Sacral Lumbar Thoracic Education, years Paid work full time n (%) Taking medication (anxiety) n (%) Taking medication (depression) n (%) WASI FSIQ CPT-II omissions CPT-II commissions Tower total achievement score (D-KEFS) CWI condition 3 (D-KEFS) CWI condition 4 (D-KEFS) BRIEF-A Global Executive Composite DEX Positive affect Negative affect HSCL-25 Total Depression Anxiety GCQ Task-focused coping Avoidant-focused coping Emotion-focused coping SF-36 Physical functioning Role physical Bodily pain General health Vitality Social functioning Role emotional Mental health

GMT (n = 24)

Control (n = 14)

Total (n = 38)

Sign.

31.79 (8.38) 10M, 14F 21 (88) 17(71)

31.79 (8.50) 6M, 8F 12 (86) 8(57)

31.79 (8.31) 16M, 22F 33 (87) 25 (66)

ns ns ns ns

2(8) 20 (83) 2(8) 12.04 (1.71) 2(8) 1(4) 0 93 (13.41) 70.8 (56.4) 61.7(11.1) 7.8 (3.9) 5.7 (3.7) 5 (3.5) 64.1 (9.8)

0 14 (100) 0 12.71 (1.90) 1(7) 2(14) 2(14) 89.1 (15.41) 48.9 (7.9) 59.6(10.3) 6.4 (4.5) 7.4 (3.5) 5.5 (3.4) 64.1 (12.1)

2(5) 34 (89) 2(5) 12.3(1.78) 3(8) 3(8) 2(5) 91.5(14.1) 62.5 (46) 60.9 (10.7) 7.3 (4.1) 6.3 (3.7) 5.2 (3.4) 64.1 (10.5)

ns ns ns ns ns ns ns ns ns ns ns ns ns ns

4.8 (2.3) 3.5 (1.6)

4.6 (2.2) 3.3 (1.9)

4.7 (2.3) 3.4 (1.7)

ns ns

26.04(16.15) 19.21 (12.82) 6.83 (4.78)

25.79 (20.43) 17.5 (14.29) 8.29 (8.46)

25.94(17.57) 18.57(13.21) 7.37 (6.31)

ns ns ns

22.92 (3.7) 19.75 (3.23) 23.17 (4.34)

25.86 (3.08) 19.57 (2.95) 23.79 (2.55)

24 (3.73) 19.68 (3.1) 23.39 (3.75)

Sign. ns ns

37.92 (26.58) 68.23 (27.64) 62.42 (28.16) 58.13(29.36) 45.21 (14.71) 64.59 (27.5) 73.61 (29.15) 69.17(18.34)

43.57 (30.66) 59.82 (22.16) 51.14(29.87) 50.21 (23.78) 38.57 (14.20) 56.25 (26.74) 69.64 (21.58) 63.93 (20.30)

40 (27.88) 65.13(25.78) 58.26 (28.93) 55.21 (27.38) 42.76 (14.69) 61.51 (27.17) 72.15(26.37) 67.24(18.98)

ns ns ns ns ns ns ns ns

Note. All neuropsychological test scores reported are standardized scores. Higher neuropsychological scores represent better performance, except for scores in CPT-II where t scores between 40 and 60 are in the normal range. Questionnaire data are reported in raw scores except for SF-36 are reported in standardized scored. A higher score on DEX, BRIEF-A, and HSCL-25 represents a higher presence and intensity of symptoms or difficulties. A higher score on GCQ represents more use of the specific coping style. A higher score on SF-36 represents better health related quality of life. MMC = Myelomeningocele; GMT = goal management training; WASI FSIQ = Wechsler Abbreviated Scale of Intelligence Full Scale Intelligence Quotient (M = 100, SD = 15); CPT-II = Conners Continuous Performance Test II (M - 50, SD = 10); CWI = Color-Word Interference Test; D-KEFS = Delis Kaplan Executive Functioning System; Subtests from D-KEFS (M = 10, SD = 3); BRIEF = Behaviour Rating Inventory of Executive Function (M = 50, SD = 10); DEX = Dysexecutive Questionnaire; HSCL-25 = Hopkins Symptom Checklist; GCQ = General Coping Questionnaire; SF-36 = The Short Form (36) Health Survey (M = 50, SD = 10). Differences between groups were tested with x2 for dichotomous variables and f-tests for continuous variables (p < 0.05). Sign. = significance; n.s., not significant.

educational requirements (see Figure 1 for CONSORT diagram; Schulz, Altman, Moher, & the CONORT Group, 2010). The study included one treatment group (GMT) and one com­ parable wait-list control group (WL), and used a repeatedmeasures design across three time points (baseline, postinterven­ tion, and 6-month follow-up). Post hoc power analysis on the available number of participants using an effect size of 0.3 and an a level of 0.05 resulted in a power of 0.98 for this study (Faul, Erdfelder, Lang, & Buchner, 2007).

Randomization, Assessment, and Intervention/Waiting List Procedure Randomization in this parallel group study was by block design with block size 2, and stratification for both age (above/below 33 years) and education (above/below 12 years). An unequal random­ ization ratio of 2:1 was used to ensure maximum use of the available intervention and to gain GMT intervention experience (Dumville, Hahn, Miles, & Torgerson, 2006). The investigators were not in-

COGNITIVE REHABILITATION IN SPINA BIFIDA

Figure 1.

5

CONSORT diagram.

volved in the randomization process. The independent person who was responsible for randomly allocating participants to group assign­ ment did not determine participant eligibility, was not involved in training procedures, and had no information about persons participat­ ing in the trial. However, subsequent to random allocation, the study was not blinded. Thus, those administering the intervention and at some point assessing outcomes, were aware of group assignment. Participants were informed about the randomization procedure, and, if assigned to the WL group, told that they would receive GMT 1 year later. GMT consists of seven modules, with a mini­ mum of 3 hr being necessary to complete each module. Earlier GMT studies have conducted trials with weekly sessions (e.g., Levine et al., 2011). Because SBM is a rare disorder, participants had to be recruited from throughout Norway, necessitating cover­ age of the seven GMT modules (7 X 3 hr) during three 3-day inpatient intervention periods over 3 months, with 1 month be­ tween each intervention period (Stubberud et al., 2013). All par­ ticipants were assessed at the same time for baseline, postinter­ vention, and follow-up. None of the participants received other cognitive rehabilitation intervention during the study period.

Intervention GMT was administered according to a manualized protocol (see also Levine et al., 2011; Stubberud et al., 2013) consisting of

PowerPoint slides and participant workbooks. Group treatment was used to exploit the opportunity for individuals to receive peer-group interaction, feedback, and support. A clinical neuro­ psychologist and a nurse/social worker led group treatment ses­ sions. Each training group consisted of six members. Although the GMT program is standardized, it is also individualized, in that training involved interactive discussions and exercises intended to increase awareness of different features of goal management to meet the needs of each participant (see Table 2). In GMT, participants are trained to use strategies such as stopping and orienting to relevant information, partitioning goals into subgoals, encoding and retaining goals, monitoring performance (Levine et al., 2011), and use of mindfulness training (Kabat-Zinn, 1990). The objective of the first module was to introduce and define the concept of absentmindedness, using absentminded errors (slip-ups) in daily life as an example. In the second module, such errors were operation­ alized as inappropriate expressions of habit (i.e., the “automatic pi­ lot”), and the “stop” strategy was introduced (i.e., periodic suspension of ongoing activities to assess goal attainment). The stop strategy was given primary emphasis throughout Module 3, in which mindfulnessbased meditation was also introduced to improve awareness toward current behavior, emotions, and goals (Kabat-Zinn, 1990). The con­ struct of working memory (denoted the “mental blackboard”) was also presented in Module 3, with participants practicing a check of

STUBBERUD ET AL.

6 Table 2

Outline o f the Modules and Objectives in Goal Management Training (GMT) GMT modules 1. The absent mind and slip-ups

2. Stop the automatic pilot

3. The mental blackboard and presentmindedness

4. State your goal

5. Making decisions

6. Splitting tasks into subtasks

7. Checking (STOP!)

Objectives/key concepts Orientation to the GMT program Defining absentmindedness, absentminded errors (sbp-ups), and goals Discussing and raising awareness of consequences of sbp-ups Defining the automatic pilot Addressing automatic pilot errors Addressing how to stop the automatic pilot

Introducing the concept of working memory as a mental blackboard Introducing the “STOP!” technique (periodic suspension of ongoing behavior) to check the mental blackboard Introducing a mindfulness-based meditation technique to acquire an ability of bringing one’s mind to the present to monitor ongoing behavior, goal states, and the correspondence between them Teaching to state goal after stopping, and present-mindedness as a way to activate goal representations “STOP!” (present-mindedness)-STATE cycle Addressing competing goals Understanding emotional reactions to conflicting goals To-do lists in the “STOP”- STATE cycle Combatting indecision Defining overwhelming tasks. Defining tasks and subtasks (goal hierarchies) Splitting the task up“ STOP!”- STATE - SPLIT cycle Recognizing “STOP!”“-STATE-SPLIT errors Using “STOP!” to monitor output/error correction GMT review

their mental blackboards to ensure that behavioral output matched intention. The main objective in Module 4 was to teach participants to state a goal after stopping, and to retrain present-mindedness. In addition to the use of to-do lists, the focus of Module 5 was decision making, especially in situations with goal conflicts or emotional reactions to competing goals. Participants practiced splitting complex goals into subgoals in Module 6. The final module involved moni­ toring ongoing behavior to check if it corresponded to intended goals (Levine et al, 2011). Participants were continually reminded to mon­ itor and evaluate executive performance in real-life situations during training, and application of GMT strategies to life situations and difficulties were discussed and reviewed. Assignments designed to facilitate transfer from GMT to real-life between sessions included monitoring and recording of both absentminded slips and success­ ful activities, along with present-mindedness practice. Every next session, these assignments were extensively evaluated. These “awareness-exercises” were continued after Module 1 and incor­ porated in every subsequent training session.

Within-session exercises

Between-session assignments

Clapping task Clapping task-revisited

Record slips, remember workbook

Card-dealing task Clapping task with “STOP!” Card-dealing task with “STOP!” by trainer, card-dealing task with “STOP!” by participant Card-dealing task with “STOP!” by participant, card-dealing task with distraction

Record slips, 30-min daily STOP

Complex task I Complex task II Complex task with to-do list

Wedding task

Daily present-mindedness practice Record slips/things that went well

Daily present-mindedness practice, record slips/ things that went well 30-min daily STOPSTATE Get to-do list Daily present-mindedness practice, catalogue task 1 Log STOP-STATESPLIT scenarios Daily present-mindedness practice, catalogue tasks 2 and 3

Clapping task with “STOP!”

Baseline Instruments In the main study (Stubberud et al., 2013), a comprehensive battery of neuropsychological tests and questionnaires was admin­ istered to the participants. For the current supplementary study, the relevant instruments were included, to characterize cognitive func­ tioning of the sample: the Wechsler Abbreviated Scale of Intelli­ gence (WASI; Wechsler, 1999), the Tower Test (TT), and ColorWord Interference Test (CWI) from Delis Kaplan Executive Functioning System (D-KEFS; Delis, Kaplan, & Kramer, 2001), and Conners’ Continuous Performance Test II (CPT-II; Conners, 2000). The BRIEF-A was used as a screening instrument.

Outcome Measures The impact of GMT on perceived emotional health (i.e., psy­ chological distress, emotional regulation, and HRQoL) and coping was investigated by means of four primary outcome measures; Hopkins Symptom Checklist 25 (HSCL-25; Derogatis, Lipman,

COGNITIVE REHABILITATION IN SPINA BIFIDA

Rickels, Uhlenhuth, & Covi, 1974), Dysexecutive Questionnaire (DEX; Burgess, Alderman, Evans, Wilson, & Emslie, 1996), Short-Form 36 health survey version 2 (SF-36; Ware & Gandek, 1998), and the General Coping Questionnaire (GCQ; Joseph, Wil­ liams, & Yule, 1992). Each measure was selected for its capacity to assess the specific aspects of emotional health and coping of interest in this study (see Table 3). All measures have been translated to Norwegian in authorized versions. The present article reports a subset of data from a larger study investigating the efficacy of GMT for patients with SBM and EF deficits. Hence, additional EF data from neuropsychological tests (Stubberud et al., 2013) and self-report questionnaires (Stubberud et al., 2014) have been published elsewhere. Hopkins Symptom Checklist 25 (HSCL-25). The HSCL-25 is a 25-point self-report inventory of depressive and anxiety symp­ toms (Derogatis et al., 1974). The HSCL-25 is derived from the 90-item Hopkins Symptom Checklist (SCL-90; Derogatis, Lipman, & Covi, 1973), and includes a 15-item depressive symptoms scale and a 10-item anxiety symptoms scale. Items are scored on a Likert scale ranging from 0 (not at all) to 4 (very much). The HSCL-25 has proved to have satisfactory validity and reliability as a measure of psychological distress (Derogatis et al., 1974). The HSCL-25 has been validated for use as a screening instrument for psychological symptoms in a range of chronic health condition populations, and has previously been administered to adults with SBM (Kalfoss & Merkens, 2006). Dysexecutive Questionnaire (DEX). The 20-item DEX from the Behavioral Assessment of the Dysexecutive Syndrome (BADS; Wilson, Alderman, Burgess, Emslie, & Evans, 1996) is a standardized instrument to measure possible behavioral changes as a result of executive dysfunction. Various factor structures with different subscales have been proposed for DEX (e.g., Simblett & Bateman, 2011). The present study utilized a five-factor structure (Burgess, Alderman, Evans, Emslie, & Wilson, 1998). For the purpose of the study, only the Positive affect and Negative affect subscales were used, and they were combined to constitute the index Emotional regulation. Items are rated on the 5-point (0 -4 ) Likert scale with each point representing a greater level of problem severity (ranging from never to very often). The DEX has demon­ strated very high internal reliability (>.8) in both clinical and control samples (Mathias, 2003). Short-Form 36 health survey version 2 (SF-36). The SF-36 is a generic 36-point self-report inventory comprising eight con­ structs of physical and mental health that has proved to have high validity and reliability (Ware & Gandek, 1998). Except for the one single-item of health transition (HT; item 2), scores of the other 35 items are summated into eight multi-item scales, including phys­

7

ical function (PF), limitations because of physical health problems (role-physical [RP]), bodily pain (BP), general health (GH), vital­ ity (VT), social functioning (SF), limitations because of emotional health problems (role-emotional [RE]), and mental health (MH). The eight multi-item scales can be aggregated into a physical component summary and a mental component summary. For this article, only the scales constituting the mental health summary measure (i.e., VT, SF, RE, and MH) were used as outcome measures. For each construct, item scores are coded, summed, and transformed to 0-100 scales (0 = poorest health, 100 = best health) according to the SF-36 algorithm (t-score transformation with mean, 50 ± 10 [YD]; Ware, Kosinski, & Dewey, 2000). The Norwegian version of the SF-36 has been validated (Kvien, Kaasa, & Smedstad, 1998; Loge, Kaasa, Hjermstad, & Kvien, 1998). General Coping Questionnaire (GCQ). The 30-item Gen­ eral Coping Questionnaire (GCQ; Joseph et al., 1992) was con­ ceptually developed and represents the subject’s general, rather than specific, coping style. The questionnaire rates coping style from 1 (never) to 4 (very often) on three dimensions, task-focused (e.g., “I make a plan of action”), emotion-focused (e.g., “I let my feelings out”), and avoidant-focused coping (e.g., “I refuse to believe that it happened”), with 10 items on each dimension. For the purpose of this study, only the task- and avoidant-focused dimensions were used as outcome measures. The Norwegian ver­ sion of GCQ-30 has demonstrated good psychometric properties, with a Cronbach’s a of 0.81 and a 4-month test-retest reliability of r = .66 (Eid, Thayer, & Johnsen, 1999).

Statistical Analyses Differences between groups were analyzed using \ 2 for dichot­ omous variables and t tests for continuous variables. A general linear model with repeated measures analysis of variance (RM ANOVA) was used to examine treatment effects. Data were ana­ lyzed using a 2 X 3 mixed-design with Group (GMT, WL) as a between-subjects factor, and Session (baseline, postintervention, and follow-up) as a within-subjects factor. Furthermore, t tests were used to explore change scores (baseline to Session 2, and baseline to Session 3) between the two groups. The strength of experimental effects was interpreted with effect size statistics, including partial eta-squared for ANOVA results and T)2 for t tests. According to Cohen (1988), thresholds for interpreting r|2 are less than .06 (small), .06 to .14 (medium), and greater than .14 (large). To control for experiment-wise error, the a level for significance was adjusted to p < .01 in the RM ANOVA, all other tests were conducted with an a level of p < .05. As only one subject was

Table 3 Constructs in the Different Outcome Measures, Including the Dependent Variables Measure

Construct

Dependent variables

HSCL-25 DEX SF-36 GCQ

Emotional health Emotional health Emotional health Coping

Anxiety and depression symptoms Emotional regulation (i.e., composite score of Positive and Negative affect subscales) Mental health (MH), Vitality (V), Social functioning (SF), and Role emotional (RE) Task-focused coping and avoidant-focused coping

Note. HSCL-25 — Hopkins Symptom Checklist-25; DEX = Dysexecutive Questionnaire; SF-36 = Short-Form 36 Health Survey; GCQ = General Coping Questionnaire.

STUBBERUD ET AL.

8

missing at follow-up (because of death) intention-to-treat analysis was not used. Follow up analyses of covariance (ANCOVAs) were performed to control for the effect of gender, injury severity (i.e., hydroceph­ alus, Chiari-II, and MMC level), IQ, and problem-solving capacity (i.e., Total achievement score Tower Test). Post hoc analyses also evaluated clinical significance of changes in HSCL-25 scores using a statistical formula for assessing the reliability of individual change scores: the Jacobson and Truax (1991) Reliable Change Index (RCI). To calculate the RCI, the posttreatment observed score is subtracted from the pretreatment observed score. This result is divided by the SE of the differences between the two test scores. If the product is larger than the z-score desired level of significance, in this case 1.96 (p < .05) the change in pre- to posttreatment scores is said to occur beyond that of chance variation (Jacobson & Truax, 1991). In our study, the follow-up scores were used as posttreatment scores. As partici­ pants were taught to use compensatory strategies autonomously, the long-term presence of treatment effects at follow-up was considered as most important. The Trial is registered at ClinicalTrials.gov, number NCTO1302314.

Results Participants As seen in Figure 1, a total of 37 individuals with SBM were included in the 6-month follow-up analysis, with one person lost to follow-up because of death. Demographic variables as well as measures of cognitive functioning, perceived emotional health, and coping from the pretreatment baseline assessment are summa­ rized in Table 1. Most participants had brain abnormalities (i.e., hydrocephalus, Chiari-II) and a lumbar-level lesion. The majority of participants were female (58%) and had finished upper second­ ary school. Very few participants (8%) were employed full-time. A very small number of the participants used medication for symp­ toms of anxiety or depression. No significant differences in demo­ graphic, medical, or cognitive functioning were found at the pre­ treatment baseline assessment, nor were there any significant differences between the groups on questionnaires measuring psy­ chological distress, emotional regulation, or HRQoL (i.e., HSCL25, DEX, or SF-36; see Table 1). No significant group differences were found on emotion and avoidant coping at the pretreatment baseline assessment, although a significant difference was detected between groups on task-focused coping (p < .05), with the WL group showing more task-focused coping at baseline (i.e., GCQ). Overall, both groups displayed impaired EF relative to the stan­ dardization samples. Compared with normative data from the general Norwegian population, both groups showed lower scores for all subscales on HRQoL (SF-36; Loge & Kaasa, 1998). Finally, both groups presented total psychological distress and depressive symptoms above scores considered as clinically significant cutoff levels, 25 and 15, respectively, on the HSCL-25 (Ravndal & Amundsen, 2010).

Effects of Treatment Tables 4 and 5 provide mean scores on questionnaire outcome data by session for intervention and control group, with session-

and group-by-time effects. An examination of the pattern of scores across time and between groups revealed that the GMT group had greater improvements in perceived emotional health and coping after training in comparison with the control group. In the WL group, no significant changes were detected after training on either of the outcome questionnaires, except for two of the SF-36 sub­ scales. Furthermore, addition of IQ, gender, injury severity, and problem-solving capacity to the RM ANOVA model did not change treatment effects. Psychological distress (HCSL-25). The ratings on HCSL-25 showed a significant reduction in the presence and intensity of total anxiety and depression symptoms across sessions for the GMT group compared with the WL group that held at follow-up ip < .01). With regard to the subscales constituting HCSL-25, there was a significant main effect for the Depression scale (p < .001), but this was reliable only for the GMT group at baseline versus follow-up and baseline versus posttraining (p < .001). Nevertheless, within the GMT group there were statistically sig­ nificant improvements on both HCSL-25 outcome variables after training {p < .001), where the GMT group’s change from baseline to postintervention held at follow-up (p < .001; see Table 4). Of note, effect-size estimates indicated overall large training effects (>.14). The variability in the WL group is somewhat high compared with the GMT group across sessions (HSCL-25 total). Two sub­ jects in the WL are responsible for the high variability, reporting high values at all three time points. A RM ANOVA was performed excluding those two subjects, and a significant interaction effect (p < .05) was still detected. Furthermore, when removing these two subjects from the analysis the SD in the control group matched the SD in the GMT group. Of note, at pretreatment assessment 11 out of 24 participants in the GMT group reported total psychological distress above scores considered as clinically significant cutoff level (>25) on the HSCL-25 (WL group: 6/14; Ravndal & Amundsen, 2010). After treatment, all (100%) of the GMT participants with a clinically elevated HSCL-25 score improved to a score considered within normal limits (WL group: 1/6), and 8 out of the 11 (73%) main­ tained a score below clinically elevated at follow-up (WL group: 1/1). Additionally, post hoc analyses demonstrated that 12 (50%) participants in the GMT group, as opposed to 2 (15%) in the WL group, had a RCI above 1.96 after treatment, indicating a clinically significant amount of improvement. Emotional regulation (DEX). No interaction effects were found for the ratings regarding emotional regulation on DEX self-report. However, the results of t tests showed that the GMT group reported significantly less problems with positive and neg­ ative affects at posttraining (p < .01) and follow-up (p < .001) compared with baseline (see Table 4). Effect-size estimates indi­ cated large training effects (>.14). Mental components in HRQoL (SF-36). Table 4 also pro­ vides mean scores on HRQoL outcome data by session for inter­ vention and control groups, with session- and group-by-time ef­ fects. No interaction effects were found for the ratings on subscale MH. However, the results of t tests showed that both groups reported significantly better mental health posttraining compared with baseline on this scale (p < .05). The WL group also reported better mental health at follow-up compared with baseline (p < .01). Eta squared statistic indicated large effect sizes (>.14).

COGNITIVE REHABILITATION IN SPINA BIFIDA

9

Table 4 Mean Scores on Measures o f Emotional Health by Session fo r Intervention Group (GMT) and Wait List Control (WL) With Session and Group by Time Effects Group

Measure assessment HSCL-25 Total score Baseline Postintervention 6-month follow-up Anxiety Baseline Postintervention 6-month follow-up Depression Baseline Postintervention 6-month follow-up DEX Emotional regulation Baseline Postintervention 6-month follow-up SF-36 Mental health (MH) Baseline Postintervention 6-month follow-up Vitality (VT) Baseline Postintervention 6-month follow-up Social functioning (SF) Baseline Postintervention 6-month follow-up Role emotional (RE) Baseline Postintervention 6-month follow-up

Group by session and session effects

GMT

Control

M (SD) (n = 24)

M (SD) (n = 13)

F (dj) group by session effect

F (dj) session effect

26.04(16.15) 12.33 (6.88)*** 13 (9.96)***

27.15 (20.58) 26.23 (24.62) 24.15(21.1)

5.29** (2, 34)

9.76*** (2, 34)

6.83 (4.78) 3.79 (2.72)*** 3.67 (2.58)***

8.77 (8.6) 9.08 (8.87) 8.92 (7.7)

4.91 (2, 34)

3.72 (2, 34)

19.21 (12.82) 8.54 (4.87)*** 9.33 (8.26)***

18.38(14.47) 17.15(17.45) 14.92 (14.96)

3.61 (2, 34)

8.65*** (2, 34)

8.29 (3.16) 6.79 (2.52)* 6.54 (2.65)**

7.85 (3.83) 7.62 (4.87) 7(3.19)

0.78 (2, 34)

4.24 (2, 34)

69.17(18.34) 77.5 (11.89)* 73.54 (16.32)

62.69 (20.58) 66.92 (22.6)* 70.77 (25.4)**

1.57 (2, 34)

4.59 (2, 34)

45.21 (14.71) 52.68(13.51)* 48.13 (16.6)

37.31 (13.94) 38.46 (9.44) 47.31 (19.32)*

4.05 (2, 34)

3.69 (2, 34)

64.58 (27.5) 82.81 (22.06)** 72.92 (27.25)

54.81 (27.26) 46.15(31.2) 64.42 (37.79)

7.28** (2,34)

2.11 (2, 34)

73.61 (29.15) 85.42 (15) 80.56 (23.01)

69.23 (22.41) 73.72 (31.7) 70.51 (36.1)

.28 (2, 34)

1.22 (2, 34)

Note.

Scores are reported in raw scores except SF-36 scores where item scores are coded, summed, and transformed to 0-100 scales (0 = poorest health, 100 = best health) according to the SF-36 algorithm (t-score transformation with mean, 50 ± 10 [SO]). A higher score represents a higher presence and intensity of symptoms or difficulties in HSCL-25 and DEX. GMT = goal management training; HSCL-25 = Hopkins symptom checklist-25; DEX = Dysexecutive Questionnaire; Emotional regulation = composite score of Positive affect and Negative affect subscales in DEX; SF-36 = Short-Form 36 health survey. Significant effects in comparison to baseline *p < .05. **p < .01. ***p < .001. All F-tests use the Wilks’ X. statistic **p < .01. ***p < .001.

No interaction effects were found for ratings on subscale VT. The results of t tests showed that the GMT group reported signif­ icantly better vitality posttraining (p < .05), and the WL group reported better vitality at follow-up, compared with baseline (p < .05). Eta squared statistic indicated large effect sizes (>.14). There was a significant Group X Session interaction on the SF scale, because of self-reported improvement in social functioning across sessions for the GMT group (p < .01), but not the WL group. This was accounted for by a significant improvement in social functioning for the GMT group at posttraining as compared with baseline (p < .01). Effect-size estimates indicated large training effects (>.14).

No interaction or time effects were found for ratings on the RE scale. In addition, no changes in limitations because of emotional health problems after training were detected on the t tests in any of the two groups compared with baseline. Coping style (GCQ). No interaction effects were found for task-focused coping. The GMT group demonstrated a significant increase in task-focused coping at posttraining {p < .001) and follow-up compared with baseline (p < .01). In addition, effectsize estimates indicated overall large training effects (>.14). With regard to avoidant-focused coping, no interaction effects were found for the ratings on GCQ. However, results of t tests showed that the GMT group reported a significant reduction in avoidant-

STUBBERUD ET AL.

10 Table 5

Mean Scores on Measure o f Coping by Session for Intervention Group ( GMT) and Wait List Control (WL) With Session and Group by Time Effects Group by session and session effects

Group

Measure assessment GCQ Task-focused coping Baseline Postintervention 6-month follow-up Avoidant-focused coping Baseline Postintervention 6-month follow-up

GMT

Control

M(SD)

M (SD)

22.92 (3.71) 27.33 (3.24)*** 26.54 (3.43)** 19.75 (3.25) 18.46 (2.99)* 18.54 (2.87)

F (df) group by session effect

F (df) session effect

26 (3.16) 25.23 (4.51) 25.77 (3.75)

5.07 (2, 34)

2.81 (2, 34)

19.77 (2.98) 19.15 (3.76) 20.46 (3.91)

2.31 (2, 34)

2.09 (2, 34)

Note. Scores are reported in raw scores. A higher score represents more use of the specific coping style in GCQ. GMT = goal management training; GCQ = General Coping Questionnaire. Significant effects in comparison to baseline *p < .05. **p < .01. ***p < .001. All F tests use the Wilks’ X statistic ** p < .01. ***p < .001.

focused coping at posttraining compared with baseline (p < .05; see Table 5). The p 2 statistic indicated a large effect size (>.14).

Discussion The principal aim of this study, analyzing a subset of data from an RCT, was to determine the efficacy of GMT on perceived emotional health and coping in adults with SBM. Overall, several results supported our hypotheses. First, the GMT group showed significant improvement, compared with WL subjects, on a selfreport inventory of depressive and anxiety symptoms (HSCL-25) after training, lasting at least 6 months posttreatment. Noteworthy is the fact that improvements in HSCL-25 were in both anxiety and depression subscales, suggesting that the treatment may have been effective in improving management of intrusions from both mood states. Significant improvements were also found on SF-36 and DEX subscales after training compared with baseline report, sug­ gesting further improvements in areas of emotional health. Fur­ thermore, the GMT group showed a significant increase in taskfocused coping and a decrease in avoidant coping after training compared to pretreatment baseline assessment scores (GCQ).

Psychological Distress The present study found a significant reduction in the presence and intensity of total anxiety and depression symptoms across sessions for the GMT group compared with the WL group that held at follow-up. Additionally, as opposed to pretreatment results, the scores of total psychological distress and depression after GMT were below levels considered as clinically significant (Ravndal & Amundsen, 2010), and clinically significant amount of improve­ ment on total distress were detected in 50% of the participants (Jacobson & Truax, 1991). Intensive intervention programs, in­ volving daily group psychotherapy and individual therapy over an extended time period, such as those run by Ben-Yishay and col­ leagues (Ben-Yishay & Diller, 2011), as well as Prigatano and colleagues (Prigatano, 1999; Prigatano & Fordyce, 1986), have been found to improve emotional health outcome after brain in­

jury. In this regard our finding is novel, considering the low intensity of GMT compared with previous studies. Nevertheless, findings in the current study are in accordance with results from van Hooren et al.’s (2007) study involving 69 community-dwelling individuals, where the GMT group reported less anxiety symptoms than those in a waiting-list control group after training. It is possible, as suggested by van Hooren et al. (2007), that improved ability to structure activities and cope with cognitive complaints may cause a reduction in psychological dis­ tress. It might also be that after training, participants felt more able to control or influence outcomes. Studies have shown a relation­ ship between perceived control and improved emotional health (Haidt & Rodin, 1999). Perhaps of even more relevance, GMT emphasizes strengthen­ ing of sustained attention to maintain awareness of goal states and output monitoring. Accordingly, GMT has been associated with improved performance on neuropsychological measures of EF, and in particular attentional control (Levine et al., 2011; NovakovicAgopian et al., 2011; Stubberud et al., 2013). Conceptually and practically, attentional control has been demonstrated to have an ameliorating effect for symptoms of anxiety and depression (Rueda et al., 2004), and it has been hypothesized that attentional control plays an essential role in ability to regulate cognition, emotion, and behavior (Posner & Rothbart, 2007). As such, im­ proved EF, and in particular attentional control, demonstrated in our main study (Stubberud et al., 2013), might have influenced ones ability to manipulate the focus of attention and thus have implications for aspects of perceived emotional health.

Emotional Regulation Because attentional control has been regarded as a subcompo­ nent process of the self-regulation system (Rueda et al., 2004), the finding that the GMT group reported significantly fewer problems with emotional regulation at posttraining and follow-up compared with baseline may provide further support to the notion that attentional control supports capacity for self-regulation. One es­ sential ingredient in GMT is a mindfulness-based attention tech-

COGNITIVE REHABILITATION IN SPINA BIFIDA

nique (Kabat-Zinn, 1990), which possibly has an important role in enhancing attentional control (Tang et al., 2007), and consequently amplify one’s capacity for self-regulation (Shapiro & Schwartz, 2000). As such, attention may be redirected from maladaptive thinking to the experience of the present moment (Teasdale et al., 1995). Novakovic-Agopian et al. (2011) reported on a variant of GMT in which 16 participants with ABI took part in a pseudoran­ dom crossover design. The intervention particularly emphasized mindfulness. With regard to emotional health, comparison with an ABI education control condition linked training with self-reported improvements in the ability to stop and relax during stressful times, and in level of anxiety after training. However, dismantling studies are needed to better understand which components that have the most effect on particular outcomes. Nevertheless, robust EF is crucial for regulating emotional and behavioral responses to circumstances and achieving desired outcomes. Without adequate attentional control a person may be unable to recognize which contextual cues are of greatest prominence and which response sets are appropriate for a particular situation (Posner, 1995).

Mental Components in HRQoL With respect to mental health components of HRQoL, both groups showed improvements after the training period on sub­ scales constituting the mental health summary score in SF-36. The GMT group reported improvement posttraining compared with baseline report in mental health (MH), vitality (VT), and social functioning (SF). Of note, only the GMT group showed a signif­ icant improvement on the subscale social functioning after train­ ing. This finding is in accordance with previous SBM studies where EF has been found to be related to self-care independence (Heffelfinger et al., 2008; Tarazi et al., 2008), social participation (Tarazi et al., 2008), and everyday physical activity (Roebroeck et al., 2006), all areas central to social functioning. Although the study design precludes drawing conclusions about causality, the results from the present study suggest that by enhancing EF, and in particular attentional control, mental health components in HRQoL may also be improved. Attentional control may enable individuals to regulate emotion and behavior by allowing them to make shifts to more intentional, controlled processes (Rueda et al., 2004). This is in line with the notion that successful goal-directed behavior requires self-regulation to override competing impulses essential to health-promoting behaviors, such as medication management, dietary and lifestyle changes, self-monitoring of responses, and follow-up with health care professionals. No prepost changes were detected on limitations because of emotional health problems (RE) in either of the groups. This might be because of item content as 2 out the 3 RE items were work-related, and only 8% of the participants were employed full-time. The WL group reported improvement in MH at posttraining and follow-up, and in VT at follow-up compared with baseline. These results were surprising because the participants in the WL group did not receive any intervention during the study period. It is possible that the expectation of the participants assigned to the waiting list to receive an intervention in the near future, and contact with health professionals may have had positive effects on their functioning. In addition, when conducting a neuropsycholog­ ical assessment there is a component of psycho-education. In our study, both groups were exposed to neuropsychological assess­

11

ments during the study that might have increased the participants’ awareness of own functioning and, hence, contributed to the re­ sults (Donker et al., 2009).

Coping Style Significant GMT-related treatment effects were found on the GCQ, as the GMT group reported a significant increase in taskfocused coping and a decrease in avoidant coping after training when compared with results from the pretreatment baseline assess­ ment. The WL participants initially reported more task-focused coping, which may have made it more difficult to detect change within this group. Nevertheless, this is a novel finding, and is particularly noteworthy as it suggests that systematic training in strategies for improving attention and problem solving may have the potential to increase task-focused coping and decrease avoidant-focused coping. This finding is, however, in accordance with Anson and Ponsford’s (2006) results, where ABI participants reported using more active problem-focused coping styles after CBT (Coping Skills Group). Although GMT and CBT are different interventions, they have some common elements, such as relax­ ation and structured problem-solving skills training. In addition, both GMT and CBT have an emphasis on enabling patients to gain skills, record progress, challenge pessimism, and promote selfefficacy. Approach-oriented coping is commonly viewed as an adaptive mode of coping that involves actively planning or engaging in a particular behavior to surmount problems causing distress (Carver et al., 1989). As participants in GMT are trained to “stop and think” about problems and goals before and during task execution, in addition to training in mindfulness and decision making, it can be argued that GMT promotes greater approach-oriented coping. The results from the present study are in accordance with findings from studies where better EF have been related more to use of approach coping strategies, whereas poorer EF have been related more to use of avoidance coping strategies (Campbell et al., 2009; Krpan et al., 2007; Spitz et al., 2013; Winocur et al., 2007). Adequate coping strategies require substantial attentional focus and may be less effectively used, or even inaccessible, when executive and attentional capacities are compromised. By enhanc­ ing EF, and in particular attentional control, the initiation of coping responses may be more adaptive as it allows individuals to orient toward and better evaluate potential stressors, and to appraise the status of the stressor when coping styles have been engaged (Compas & Boyer, 2001). Research has shown that persons with chronic illness or disabil­ ity who use active coping styles (i.e., approach-oriented coping) as opposed to avoidant coping styles have been shown to have more successful psychosocial adaptation (Lewis & Kliewer, 1996). Cop­ ing style has also been suggested as a key concept in explaining the effects of stress, productivity, social activity, psychological func­ tioning, and QoL after ABI (Anson & Ponsford, 2006; Finset & Andersson, 2000). In general, our results are in line with evidence supporting structured group-based compensatory interventions involving problem solving and GMT that have incorporated “stop-and-think training” to manage EF problems (e.g., Levine et al., 2000; Miotto et al., 2009; Rath et al., 2003). These studies have, however, mainly shown an impact on neuropsychological measures in stud-

STUBBERUD ET AL.

12

ies involving subjects with ABI. We were able to extend this evidence by demonstrating that beneficial effects also can be achieved with regard to aspects of perceived emotional health and coping in patients with congenital brain dysfunction when using GMT.

Strengths, Limitations, and Future Research The study had a number of strengths, including randomized assignment to GMT or WL, follow-up assessment, and inclusion of measures related to perceived emotional health and coping that have not previously been examined in studies of GMT. The study also had a number of limitations. One limitation is that all out­ comes were assessed only by self-report. Although several factors (e.g., awareness, demand characteristics, cognitive deficits, or so­ cial desirability bias) may affect the accuracy and validity of self-report, these measurement methods offer the advantage of giving access to participants’ own perspectives. Moreover, non­ specific intervention effects cannot be ruled out as contributing to results without having an active control group. Accordingly, de­ mand characteristics in the GMT group cannot be ruled out as contributing to the positive effects. Another possible limitation of the present study was that the persons who carried out the assess­ ments posttreatment were not blind to group membership. Finally, the present findings need to be cross-validated in a larger and more representative SBM sample considering the relatively small sam­ ple size.

Conclusions Overall, our findings indicate that by using a compensatory intervention to manage executive dysfunction, effective and lasting benefits can be achieved with regard to several aspects of per­ ceived emotional health and coping in SBM patients. These find­ ings include areas of psychological distress, emotional regulation, HRQoL, and coping style.

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Appendix CONSORT 2010 Checklist of Information to Include When Reporting a Randomised Trial3

Section/topic Title and abstract Introduction Background and objectives Methods Trial design Participants

Checklist item

la lb

Identification as a randomised trial in the title Structured summary of trial design, methods, results, and conclusions (for specific guidance see CONSORT for abstracts)

i 2

2a 2b

Scientific background and explanation of rationale Specific objectives or hypotheses

3-9 9

3a 3b

Description of trial design (such as parallel, factorial) including allocation ratio Important changes to methods after trial commencement (such as eligibility criteria), with reasons Eligibility criteria for participants Settings and locations where the data were collected The interventions for each group with sufficient details to allow replication, including how and when they were actually administered Completely defined prespecified primary and secondary outcome measures, including how and when they were assessed Any changes to trial outcomes after the trial commenced, with reasons How sample size was determined When applicable, explanation of any interim analyses and stopping guidelines

9-11 11

Interventions

4a 4b 5

Outcomes

6a

Sample size

6b 7a 7b

Randomisation: Sequence generation Allocation concealment mechanism Implementation

Reported on page no.

Item no.

8a 8b 9 10

Method used to generate the random allocation sequence Type of randomisation; details of any restriction (such as blocking and block size) Mechanism used to implement the random allocation sequence (such as sequentially numbered containers), describing any steps taken to conceal the sequence until interventions were assigned Who generated the random allocation sequence, who enrolled participants, and who assigned participants to interventions

(Appendix continues)

9-10 9 9-11 13-16 na 10 na

n ii ii ii

STUBBERUD ET AL.

16 Appendix (continued)

Reported on page no.

Item no.

Checklist item

Blinding

11a

ii

Statistical methods

lib 12a 12b

If done, who was blinded after assignment to interventions (for example, participants, care providers, those assessing outcomes) and how If relevant, description of the similarity of interventions Statistical methods used to compare groups for primary and secondary outcomes Methods for additional analyses, such as subgroup analyses and adjusted analyses For each group, the numbers of participants who were randomly assigned, received intended treatment, and were analyzed for the primary outcome For each group, losses and exclusions after randomisation, together with reasons Dates defining the periods of recruitment and follow-up Why the trial ended or was stopped A table showing baseline demographic and clinical characteristics for each group For each group, number of participants (denominator) included in each analysis and whether the analysis was by original assigned groups For each primary and secondary outcome, results for each group, and the estimated effect size and its precision (such as 95% confidence interval) For binary outcomes, presentation of both absolute and relative effect sizes is recommended Results of any other analyses performed, including subgroup analyses and adjusted analyses, distinguishing prespecified from exploratory All important harms or unintended effects in each group (for specific guidance see CONSORT for harms)

10, 17

Trial limitations, addressing sources of potential bias, imprecision, and, if relevant, multiplicity of analyses Generalizability (external validity, applicability) of the trial findings Interpretation consistent with results, balancing benefits and harms, and considering other relevant evidence

26-27

Section/topic

Results Participant flow (a diagram is strongly recommended)

13a

Baseline data Numbers analyzed

13b 14a 14b 15 16

Outcomes and estimation

17a

Recruitment

17b Ancillary analyses

18

Harms

19

Discussion Limitations

20

Generalizability Interpretation

21 22

Other information Registration Protocol Funding

23 24 25

Registration number and name of trial registry Where the full trial protocol can be accessed, if available Sources of funding and other support (such as supply of drugs), role of funders

na 16-17 na

10, 17 10 na 10 17 17-21 na 18-19 na

26-27 21-27

17 na 28

a We strongly recommend reading this statement in conjunction with the CONSORT 2010 Explanation and Elaboration for important clarifications on all the items. If relevant, we also recommend reading CONSORT extensions for cluster randomised trials, non-inferiority and equivalence trials, nonpharmacological treatments, herbal interventions, and pragmatic trials. Additional extensions are forthcoming: for those and for up to date references relevant to this checklist, see www.consort-statement.org. Received February 10, 2014 Revision received October 19, 2014 Accepted October 23, 2014 ■

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