Child Neuropsychology, 2015 Vol. 21, No. 2, 191–209, http://dx.doi.org/10.1080/09297049.2014.889110

Improving executive functioning in children with fetal alcohol spectrum disorders Kelly Nash1,2, Sara Stevens1,3, Rachel Greenbaum4, Judith Weiner2, Gideon Koren1, and Joanne Rovet1,2,3 1

The Hospital for Sick Children, Neuroscience and Mental Health, Toronto, Ontario, Canada 2 The Ontario Institute of Studies in Education, University of Toronto, Toronto, Ontario, Canada 3 The Hospital For Sick Children, The University of Toronto, Toronto, Canada 4 Surrey Place Centre, Toronto, Canada An extensive body of literature has documented executive function (EF) impairments in children with fetal alcohol spectrum disorders (FASD); however, few studies have aimed specifically at improving EF. One treatment program that shows promise for children with FASD is the Alert Program for SelfRegulation®, which is a 12-week treatment specifically designed to target self-regulation, a component of EF. The present study sought to examine if Alert would produce improvements in selfregulation that would generalize to other aspects of EF, behavior, and social skills in children with FASD. Twenty-five children aged 8–12 years diagnosed with an FASD were assigned in alternating sequence to either an immediate treatment (TXT) or a delayed treatment control (DTC) group. Both groups received a comprehensive evaluation of EF at baseline and upon completing therapy (TXT), or after a 12- to 14-week interval from baseline (DTC). Parents also completed questionnaires assessing EF and behavior at both time points. For the TXT group only, parent questionnaires were readministered at 6-month follow-up. At the 12-week follow-up, the TXT group displayed significant improvements in inhibitory control and social cognition. Parents of children in the TXT group reported improved behavioral and emotional regulation, as well as reduced externalizing behavior problems. These behavioral improvements along with further improved parent-rated inhibitory control was maintained at the 6-month follow-up. The EF disabilities in children with FASD can be remediated through a targeted treatment approach aimed at facilitating self-regulation skills. Keywords: Treatment; Fetal alcohol spectrum disorders; Self-regulation; Executive functioning; Alert.

Fetal alcohol spectrum disorder (FASD) is the umbrella term to denote the range of conditions that arise from prenatal exposure to alcohol and affect as many as 1–2.5% of newborns in North America. Of the several diagnostic subtypes FASD encompasses, the best known is Fetal Alcohol Syndrome (FAS), which involves a triad of features that include a distinctive dysmorphic face, growth abnormalities, and marked neurobehavioral impairments; partial Address Correspondence to Kelly Nash, The Hospital for Sick Children, 555 University Avenue, Toronto ON M5G1X8 Canada. E-mail: [email protected]

© 2014 Taylor & Francis

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FAS is similar but involves fewer and less severe physical features (Institute of Medicine of the National Academy of Sciences Committee to Study Fetal Alcohol Syndrome, 1996). However, the most prevalent form of FASD is Alcohol-Related Neurodevelopmental Disorder (ARND), which is an invisible disorder involving no associated physical features but the full set of neurobehavioral characteristics (Chudley et al., 2005). Regardless of subtype, most children with FASD show a diversity of severe cognitive impairments (Nash, Sheard, Rovet, & Koren, 2008) that include memory difficulties (Mattson, Gramling, Delis, Jones, & Riley, 1996; Rasmussen, 2005; Willoughby, Sheard, Nash, & Rovet, 2008), language delays (Coggins, Olswang, Carmichael Olson, & Timler, 2003), visuospatial weaknesses (Mattson, et al., 1996), attention problems (Mattson, Calarco, & Lang, 2006), and reduced IQ (Mattson et al., 1997; Rasmussen, Horne, & Witol, 2006). Notably, most children with FASD also have deficient executive functioning (EF) skills, which have come to be recognized as a hallmark deficit of the disorder (Kodituwakku, 2007, 2010). EF is the umbrella term used to describe a broad range of mental abilities that encompasses cognitive and socioaffective domains. Cognitive EFs include the following: anticipation and deployment of attention; impulse control and self-regulation; initiation of action; working memory; mental flexibility; utilization of feedback; planning and organization; and the selection of efficient problem-solving strategies (Anderson, Jacobs, & Anderson, 2008; Prencipe et al., 2011; Stuss & Levine, 2002; Wu et al., 2011). Socioaffective EFs include social cognition and emotion processing (Damasio, Grabowski, Frank, Galaburda, & Damasio, 1994; Zelazo, Craik, & Booth, 2004). Importantly, cognitive and socioaffective EF domains are not independent. Rather, they work in concert, rely on the same mechanisms and may be best viewed as falling along a continuum (Hongwanishkul, Happaney, Lee, & Zelazo, 2005; Prencipe et al., 2011; Stuss & Levine, 2002). Indeed, recent neuroimaging findings provide support for this interdependence showing significant overlap, albeit with some distinctions, between brain regions necessary for cognitive and socioaffective EF (Amodio & Frith, 2006; Happaney, Zelazo, & Stuss, 2004; Sabbagh, 2004; Yeates et al., 2007). Unfortunately, children with FASD show difficulties in nearly all aspects of EF (Burden, Jacobson, Sokol, & Jacobson, 2005; Connor, Sampson, Bookstein, Barr, & Streissguth, 2000; Fryer, Tapert, et al., 2007; Greenbaum, Stevens, Nash, Koren, & Rovet, 2009; Green et al., 2009; Kodituwakku, Kalberg, & May, 2001; Mattson et al., 2006). Cognitive and socioaffective EFs are also assumed to be dependent on self-regulation ability (Barkley, 2001; Rothbart, Ziaie, & O‘Boyle, 1992). Studies of children with brain injury other than FASD have shown that, when damage occurs between 0 and 3 years of age, the selfregulation component of EF is primarily affected, whereas brain damage during later stages of development (4–6 years) affects other brain functions (Anderson et al., 2008; Dennis, 1989). Given the timing of the early brain insult in children with FASD, it is not surprising that several researchers have proposed that self-regulation may represent a core deficit in this population (Kodituwakku, 2010; Kodituwakku, Handmaker, Cutler, Weathersby, & Handmaker, 1995). In children with FASD, deficits in self-regulation are evident throughout development and first appear as abnormal sleep-wake cycles and variable arousal states in infancy (Coles, Kable, Drews-Botsch, & Falek, 2000; Landesman-Dwyer, Keller, & Streissguth, 1978), next as heightened reactivity and high levels of distractibility and hyperactivity and sensory processing in toddlerhood (Nulman et al., 2004; Steinhausen, Nestler, & Spohr, 1982), and then as significant attention (Nanson & Hiscock, 1990) and higher order EF deficits at school age (Connor et al., 2000; Green et al., 2009; Kodituwakku, 2009; Kodituwakku et al., 2001; Mattson, Goodman, Caine, Delis, & Riley, 1999; Mattson et al., 2010).

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Self-regulation deficits are also shown to predict the high levels of behavior disturbance as well as the abnormal and delayed social skills frequently observed in children with FASD (Fryer, McGee, Matt, Riley, & Mattson, 2007; Greenbaum et al., 2009; Schonfeld, Mattson, & Riley, 2005; Schonfeld, Paley, Frankel, & O‘Connor, 2006). Indeed these children are at high risk for behavior problems and psychopathology, including conduct disorder, oppositional defiant disorder (ODD), and attention deficit/ hyperactivity disorder (ADHD). When compared with other pediatric populations, such as ADHD, children with FASD are more likely to engage in maladaptive behaviors, such as lying and stealing, and lacking guilt and acting younger than their ages (Nash et al., 2006; Nash, Koren, & Rovet, 2011). Researchers have also found that, as children with FASD get older, they lag further behind their same-age peers in self-regulatory and social abilities (Thomas, Kelly, Mattson, & Riley, 1998; Whaley, O‘Connor, & Gunderson, 2001). Given that the self-regulation deficits in this population are pervasive and worsen with age, researchers have proposed that it may represent a core target of EF for intervention (e.g., Kodituwakku, 2010). One effective therapy for facilitating children’s self-regulation behaviors is the Alert Program for Self-Regulation®, a 12-week manualized intervention that helps children to monitor, to maintain, and to regulate their levels of “alertness” to match environmental needs (Williams & Shellenberger, 1996). Previous research on Alert has reported significantly improved self-regulation for children with emotional disturbance and conduct disorder (Barnes, Vogel, Beck, Schoenfeld, & Owen, 2008; Israel, Guile, Baker, & Silverman, 1994), while one recent study reported improvement in children with FASD (Wells, Chasnoff, Schmidt, Telford, & Schwartz, 2012). However, as this study was limited by not examining their abilities but directly relying only upon parent- and childreport questionnaire measures (Wells et al., 2012), further research is required. The current study sought to circumvent the above limitations by evaluating the Alert program® in children with FASD within the context of a wait-list control design. In particular, different aspects of social and cognitive EF were directly assessed using measures of attention, set shifting, planning, and social cognition, as well as parentrated questionnaires of EF, behavior problems, and social skills. This broad range of EF measures was used to examine if self-regulation abilities are a “keystone” to improving other aspects of EF in children with FASD and, similar to work with other populations of children (Yeates et al., 2007), to shed light on whether a self-regulation intervention can produce widespread positive change in other domains of functioning. Based on previous research using the Alert program®, we hypothesized that the immediate treatment group of children, compared to the wait-list group would show improvements in self-regulation and these would generalize to parent-reported behaviors and social skills.

MATERIALS AND METHODS Participants Originally enlisted were 25 children with FASD between the ages of 8 and 12 years old with a mean age of 10.3. All children had a previous diagnosis of FASD from two sources, the Motherisk Clinic at The Hospital for Sick Children (“SickKids” n = 16) or an accredited FASD diagnostic facility in Ontario (n = 9). All children had confirmation of heavy maternal alcohol consumption during pregnancy as per maternal report, Children’s Aid Society documentation when child was removed from the mother, or adoption

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records. At SickKids, the diagnostic process involved a team consisting of a pediatrician, psychologist, psychometrist, and speech language pathologist, who over several days saw the child and family with the final diagnosis being based on consensus of the pediatrician and psychologist. The Motherisk Clinic used the Canadian Diagnostic Guidelines system (Chudley et al., 2005); this system was derived from the Washington and Institute of Medicine methods (Astley & Clarren, 2001; Institute of Medicine of the National Academy of Sciences Committee to Study Fetal Alcohol Syndrome, 1996) but included more detailed criteria for designating the ARND category whereby a child must show a minimum of three significant deficit areas to receive an ARND diagnosis. The 9 children diagnosed elsewhere were diagnosed using the Washington four-digit code system: 5 children fulfilled criteria for partial FAS and 4 had ARND exclusively. Because this study was part of a larger study involving experimental tests of greater cognitive complexity (to be reported subsequently) and the manualized guidelines proposed by Alert, only children with an IQ of 70 or above were included. There were no children with FAS from any of the above-described clinical facilities. Procedures Initially, letters were mailed to parents of children meeting age criteria who had been previously diagnosed in the Motherisk clinic. Interested families contacted the project coordinator, who conducted a screening interview by telephone to determine initial eligibility. The parents/caregivers of children not diagnosed at SickKids, who had heard about our project via FASD support groups, to which postings had been sent, directly contacted the study coordinator. After enrolment, children were assigned on an alternating-sequence basis to either the immediate-treatment (TXT) or delayed-treatment control (DTC) conditions. This approach, rather than strict randomization, was used for practical purposes in scheduling testers and the MRI scanner (data presented in Nash, 2012). Two families contributing 2 children to the study were allowed to have both assigned to the same condition, which turned out to be the TXT group. Although children were individually seen, they were studied in cohorts of 10 (5 per condition), all of whom were seen in the same single school semester or summer period. All study-related activities took place at SickKids and were approved by the Research Ethics boards at SickKids and the University of Toronto. The design involved baseline testing (pretest), treatment of the TXT group, posttesting, and treatment of the waitlist group. Baseline testing took place over two days separated by 2–30 days. On the first day of baseline testing, children received an extensive neuropsychological test battery and parents completed questionnaires. On the second day, children were scanned and completed any remaining tests. Approximately one week later, the TXT group started therapy, which consisted of 12 one-hour sessions over a period of around 14 weeks, allowing for illnesses and holiday interruptions. Posttesting was provided within 2 weeks of completing therapy (TXT group) or a comparable period of time (DTC group) and consisted of a modified test battery and a posttreatment scan on a single day. Children in the DTC group commenced therapy within ~1 month of posttesting. All parents/caregivers were also invited back for a 1-hour feedback session to discuss the child’s results and to receive psychoeducational recommendations and techniques for translating the therapeutic tools into home and classroom practice. Families were compensated for all travel expenses. At 6 months posttreatment, parents/caregivers of the TXT group were mailed questionnaires as a long-term follow-up. As part of their

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participation in a larger study, children also completed structural and functional neuroimaging at baseline and posttreatment and those results are reported independently (Nash, 2012). The Alert Program for Self-Regulation® therapy was provided on an individual basis in a specially designated therapy room that contained floor mats, therapy balls, inner tubes, large pillows, a tent, a caterpillar tunnel, and a variety of manipulanda, as specified by the program. The room was devoid of any extraneous visual or auditory distractions. The program itself was originally developed by occupational therapists (Williams & Shellenberger, 1996) and focuses on improving self-regulation through sensory integration and cognitive processing activities via the analogy of a car engine. There are 12 sessions organized into three successive stages with a child not proceeding to the next stage until the previous one is mastered as indicated by “mile markers” set after each stage of therapy. Stage One consists of four sessions during which children learn to identify and label their “engine levels” such as engines running high, low, or just right and thus to gain awareness of their “engine speeds.” Stage Two consists of four sessions in which children experiment with changing their engine speeds by acquiring self-regulation strategies involving the five senses in order to either “gear up or down” to obtain a “just right” engine level. Stage Three teaches children to choose strategies independently and to use them outside the therapy over three sessions while the fourth session provides a review of concepts learned. Thus, this approach both ensured an individualized treatment program to meet each child’s unique needs and to allow for a positive, success-based learning environment. It is important to note that the Alert intervention included metacognitive strategies (thinking about engine levels), mind-body awareness (what is the body doing in different engine levels), and sensory-based coping strategies, all of which were interwoven throughout the 12 sessions. Table 1 provides a detailed description of each Alert session. Quality control was ensured by having (a) the two senior doctoral-level graduate student therapists (KN, SS) receive training in Alert from its developers, (b) a registered clinical psychologist (RG) observe video of select sessions, and (c) biweekly supervisory meetings to adapt the treatment to each child’s needs and deficit profile. The latter involved only how the therapy was delivered (e.g., use of visuals, play-based learning of concepts), rather than changes in the content of the intervention, thus preserving the integrity of the therapy. Measures Demographics. Background information was obtained via caregivers who completed the NEPSY History questionnaire. Socioeconomic status (SES) was determined from information collected within this questionnaire using the Hollingshead system (Hollingshead, 1975). Intelligence was assessed with the Vocabulary and Matrix Reasoning subtests of the Weschler Abbreviated Scale of Intelligence (WASI; Weschler, 1999), while language was assessed with the Peabody Picture Vocabulary Test–fourth edition (PPVT-4; Dunn & Dunn, 2007). Cognitive and Socioaffective EF Measures. Cognitive EF was measured using select subtests from the NEPSY-II (Korkman, Kirk, & Kemp, 2007), the Test of Everyday Attention for Children (TEA-Ch), and the Cambridge Neuropsychological Test Automated Battery (CANTAB) that targeted inhibitory control, visual and auditory

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Table 1 Overview of Alert Sessions. Session Number Stage 1 1

2

3

4

Stage 2 5

6 7

8

Stage 3 9

10

11

12

Session Description

How does your engine run: Therapeutic rules and issues pertaining to confidentiality are discussed. Using the metaphor of a car engine for the human brain, the clinician introduces the concept of self-regulation. Just as a car engine runs low, just right, and high, so does our brain. Once we learn how to identify what gear our engine speed is in we can learn to make adjustments depending on the situation. Self-Regulation—Feelings and Behaviors: The child participates in experiential activities that will enable one to learn to identify when one’s engine is running in low, just right and high gear. Throughout the session, the clinician will reinforce the idea that, while we sometimes need to adjust our engine speed to fit the demands of a situation, there are times when we need our engines to be in low or high gears. The clinician labels the child’s engine levels. Self-Regulation-Feelings and Behaviors Stage II: The clinician introduces the engine speed chart to help the child label his/her “engine levels” while participating in various activities throughout the course of a session. Planning and Behavior: The child begins to identify their own engine levels through experiential awareness. The child learns different subtypes of the engine levels. Beginning of generalization: The child begins to label one’s engine levels outside of the therapy sessions, using visual aids. The child begins with experimenting with different oral motor tools to change their engine levels. Methods to change engine speeds continued: The child learns engine tools for the body and hands. Methods to change engine speeds continued: The child learns engine tools for the eyes and ears. Previous engine tools are reviewed and the clinician comments on what tools she has observed work for the child. Problem Solving: Via pictures, the child begins to strategize with appropriate engine tools in different academic and social situations with the assistance of the clinician. Problem-Solving II: Via pictures, the child begins to strategize with appropriate engine tools in different academic and social situations with the assistance of the clinician. The child identifies triggers that cause one’s engine to up or down regulate. Generalization III: The child begins to generalize changing how alert they feel by charting “what works and what bothers them” in different situations independently. Emotions and generalization: The child shares stories from home and school where they encountered emotional situations where they used their engine tools or had difficulty using their engine tools. The clinician prepares the client for the last session, including a discussion about the many feelings we might have when it is time to say good-bye. Conclusion and Graduation: The final session is conducted with the child and concludes with the child and their parents present. The client and their parents review and practice concepts learned over the course of 11 sessions. Children are awarded a master mechanic certificate to celebrate their growth and achievement. Children are presented with their strengths binder, which is a compilation of learning exercises and art projects they completed in each of the sessions. A photograph is taken with the child and clinician, which is then mailed to the child within one month of completing therapy.

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attention, set shifting, and planning abilities. The NEPSY-II Inhibition subtest (Korkman, Kurt, & Kemp, 2007), a timed test requiring the child to inhibit an automatic responses in favor of a novel response, served to measure inhibitory control. Visual attention was assessed using Sky Search from the TEA-Ch (Manly, Roberston, Anderson, & NimmoSmith, 1998) that required children to find the “target” ships in an array of distracting ships. Auditory attention was assessed using the TEA-Ch Score subtest, which required children to track the number of sounds as they heard them on a tape recording. Set shifting was assessed with the Intra/Extra dimensional Shift task (IED) from the CANTAB (Robbins et al., 1994), which required children to learn new rules as the former ones were changed intermittently. Planning was assessed with the Stockings of Cambridge (SOC) subtest from the CANTAB (Robbins et al., 1994). Across tests, lower scores signified poorer performance. The particular cognitive EF measures were chosen because they have strong reliability and validity and previous research has shown that they are reflective of the cognitive EF deficits in this population (e.g., Green et al., 2009; Mattson et al., 1999). In the socioaffective EF domain, emotion recognition and social cognition abilities were specifically assessed based on previous findings by Greenbaum et al. (2009) showing children with FASD are impaired in these abilities. Emotion recognition was evaluated using the NEPSY-II Affect Recognition subtest, in which children are required to match the emotions in pictures of children’s faces. Social cognition was assessed using four subtests from The Test of Social Cognition, each of which presented stories read by the examiner (Saltzman-Benaiah & Lalonde, 2007). The False Beliefs subtest contained eight stories each describing an interpersonal situation involving a false belief, sarcasm, or deception. Strategic Control of Emotions contained four stories about situations in which story characters hid their emotions in order to either protect other people or oneself from being hurt (e.g., from embarrassment). Personalized Thoughts consisted of four brief scenarios in which a child doll informed a second infant or adult doll about the location of a hidden prize using ambiguous or unambiguous information. Personalized Emotions contained two short stories describing a character involved in two events whereby the first event might influence the character’s feelings about the second event (e.g., seeing a scary moving about snakes and then being scared of a snake). On all tests, lower scores reflected poorer performance.

Parent Questionnaires. The Behavior Rating Inventory of Executive Functioning (BRIEF; Gioia, Isquith, Guy, & Kenworthy, 2000) was used to evaluate children’s executive functioning abilities in their daily functioning. For the present purposes, we used two of the BRIEF composite scores, the Behavioral Regulation Index and the General Executive Composite, and the three subscales of the BRIEF Behavioral Regulation Index. On this task, scores for multiple scales are provided as T scores (M = 50, SD = 10), with scores of 65 or higher marking the clinically elevated range. The Child Behavior Checklist (CBCL; Achenbach & Rescolora, 2001) was used to assess the children’s behavior problems. This is a widely used instrument for 6- to 18-year-old children that contains 118 items, which provides a number of broad and narrow band scales. It is scored as T-scores with scores of 65 or higher signifying the clinically elevated range. For present purposes, we used results from the Externalizing Problems and Total Problem scores broadband scales. The Social Skills Improvement System questionnaire (SSIS; Gresham & Elliot, 2008) for 3- to 18-year-old children assesses both social skills and

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behavior problems via 60 items, which are scored as standard scores (M = 100, SD = 15). For the present purposes, we used only the Social Skills subscale, which evaluates communication, cooperation, assertion, responsibility, empathy, engagement, and self-control abilities. Higher scores on the BRIEF and CBCL signify more behavior problems while lower SSIS Social Skills scores signify poorer social-skill functioning. Data Analyses. Baseline comparisons of TXT and DTC groups on demographic variables were conducted using chi-square and analysis of variance (ANOVA). The shortterm efficacy of the therapy in improving self-regulation and EF was evaluated using analysis of covariance (ANCOVA) with therapy condition (TXT vs. DTC) as the grouping factor and baseline scores as covariates. Six-month follow-up data for self-regulation results were analyzed using paired t-tests, comparing these data with comparable scores from the first posttreatment. Since we had specific hypotheses, one-tailed ANCOVAs were used. RESULTS Participant Attrition and Demographic Data From the original 29 children, 14 were assigned to the TXT condition and 15 to the DTC condition. The mean age of the study sample was 10.3 years. Of these, 12 in the TXT group and 13 in the DTC group completed the baseline and 12-week posttreatment or wait phases of the study. Of the 4 noncompleters, 3 (1 TXT, 2 DTC) had custody access issues and did not continue after baseline testing, and 1 child was lost to follow-up between the initial screening interview and scheduling of baseline testing. Although the 13 children in the DTC were all offered therapy after posttesting, it was declined for two cases when services closer to home became available. Nine of the 12 parents from the TXT group returned questionnaires at 6-month follow-up. Since Alert is divided into three hierarchical stages of mastery, treatment success was also evaluated based on children’s ability to move through each stage of Alert. Twenty-four children successfully mastered all three levels of Alert. One child, with an IQ of 70 and a significant trauma history, remained in Stage 2. Table 2 presents the demographic characteristics of the children who completed the study. At baseline, groups differed significantly only on two indices: frequency of ADHD diagnoses and exposure to both alcohol and drugs. Significantly more children in the DTC than TXT group were diagnosed with ADHD, 85% versus 42%, χ2(1) = 5.0, p < .05, while significantly more children in the TXT than DTC group were exposed to both alcohol and drugs in utero, 67% versus 23%, χ2(1) = 3.8, p = .05. Treatment Effects on Cognitive and Socioaffective EF Domains Table 3 presents results for the cognitive EF indices. ANCOVA revealed a significant treatment effect for the Inhibition-Naming score from the NEPSY-II Inhibition subtest, F(2, 20) = 6.12, p = .001. As shown in Figure 1, the TXT group made significant gains on this subtest with scores now falling within the normal range, whereas the DTC group did not make gains and in fact exhibited a slight decline remaining in the impaired range. Effects were not significant for the Inhibition-Inhibition score, F(2, 18) = 3.27, p = .15, or Inhibition-Switching, F(2, 18) = 2.12, p = .30, subtests. Regarding attention, a trend-level effect was observed for the TEA-Ch Score subtest, F(2, 22) = 2.89, p = .15.

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Table 2 Sample Characteristics of the Treatment (TXT) and Delayed Treatment Control (DTC) Groups. TXT Group

DTC Group

(n = 12)

(n = 13)

50 50 10.3 (1.7)

46 54 10.4 (1.3)

ns ns ns

75 17 8

54 8 38

ns ns ns

42 42 16

69 8 23

ns ns ns

58 67

77 23

ns p = .05

42 25 50 0 8

85 15 38 15 8

p < .05 ns ns ns ns

86.3 (12.7) 97.3 (17.1)

92.7 (15.3) 99.5 (11.7)

ns ns

Variables Sex (%) Females Males Age M (SD) Placement Status (%) Adopted Foster Kinship Caregiver Socioeconomic Status (%) Low Medium High Exposure History (%) Alcohol and Cigarettes Alcohol and Secondary Drugs Comorbidities (%) ADHD diagnosis ODD diagnosis LD diagnosis Anxiety diagnosis Sensory Processing Delay Diagnosis Cognitive Profile M (SD) Child Composite IQ Receptive Language

p value

Note. ODD = Oppositional defiant disorder; LD = Learning disability.

Table 3 Change in Cognitive EF Domain Scores as a Function of Self-Regulation Therapy.

Cognitive EF Measure Inhibition-Naminga Inhibition-Inhibition Inhibition-Switching Sky Search Score! CANTAB IEDb CANTAB SOC a

TXT group

DTC Group

M (SD)

M (SD)

p value

Effect size

Baseline: 6.63 (3.5) Posttest: 9.80 (3.2) Baseline: 6.45 (3.4) Posttest: 7.40 (4.0) Baseline: 5.80 (2.30) Posttest: 7.22 (2.0) Baseline: 4.12 (2.6) Posttest: 6.42 (2.7) Baseline: 7.91 (3.3) Posttest: 7.73 (3.3) Baseline: 0.02 (1.0) Posttest: –0.10 (0.93) Baseline: –0.74 (0.59) Posttest: –0.47 (1.0)

Baseline: 6.69 (3.9) Posttest: 6.23 (3.5) Baseline: 6.67 (3.3) Posttest: 5.73 (3.6) Baseline: 5.17 (3.1) Posttest: 6.55 (2.0) Baseline: 4.50 (2.0) Posttest: 7.9 (3.2) Baseline: 5.92 (3.2) Posttest: 6.00 (3.1) Baseline: –0.40 (0.87) Posttest: –0.41 (1.2) Baseline: –0.55 (0.91) Posttest: –0.52 (1.0)

.01

.283

.15

.060

.30

.010

.20

.033

.15

.047

.30

.014

.40

.003

Results presented as scaled scores (M = 10, SD = 3). Results presented as z-scores (M = 0, SD = 0.5). Note. IED = Intra/extra dimensional shift; SOC = Stockings of Cambridge. b

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Scaled Scores

NEPSY Naming 10 9 8 7 6

TXT DTC Posttreatment

Baseline

Time Figure 1 Change in NEPSY Inhibition-Naming Score as a function of therapy.

Table 4 Change in Socioaffective EF Domain Scores as a Function of Self-Regulation Therapy. TXT group

DTC Group

Socioaffective EF Measure

M (SD)

M (SD)

p value

Effect Size

NEPSY Affect Recognition

Baseline: 6.63 (3.5) Posttest: 9.80 (3.2) Baseline: –1.28 (1.3) Posttest: –0.98 (1.3) Baseline: –3.01(2.3) Posttest: –2.76 (2.2) Baseline: –2.86 (2.6) Posttest: –2.61 (3.1) Baseline: –0.75 (1.2) Posttest: –0.42 (1.2)

Baseline: 6.69 (3.9) Posttest: 8.15 (3.5) Baseline: –1.4 (1.5) Posttest: –2.35 (3.2) Baseline: –3.43 (1.9) Posttest: –2.77 (1.4) Baseline: –3.05 (3.2) Posttest: –2.00 (2.3) Baseline: 0.09 (0.8) Posttest: –0.22 (1.0)

.05

.103

.07

.004

.39

.101

.09

.002

.43

.083

Strategic Control of Emotions False Belief, Deception, and Sarcasm Personalized Emotions Personalized Thoughts

No group differences were observed at posttest on indices of attention switching or planning from the CANTAB. Table 4 presents the results for the socioaffective EF domain. A significant treatment effect was observed for NEPSY-II Affect Recognition, F(2, 21) = 4.82, p = .05, reflecting scores improving into the normal, from clinical, range for the TXT group. While the DTC group also showed some improvement, scores were lower than the DTC group and at the bottom of the normal range. On the Test of Social Cognition, trend-level effects were seen for two subtests: Strategic Control of Emotions, F(2, 21) = 6.49, p = .07, and Personalized Emotions, F(2, 21) = 5.46, p = .09. On Strategic Control of Emotions, the TXT group showed improved performance with the DTC group declining, whereas, on Personalized Emotions, only the DTC group showed improvement. Groups did not differ on False Beliefs or Personalized Thoughts. Parent-Rated EF Treatment Effects Table 5 contains parent-questionnaire results. On the BRIEF, a significant treatment effect was observed for the Behavioral Regulation Index, F(2, 21) = 22.6, p = .01, and a trend-level effect was observed for the General Executive Functioning Index, F(2, 21) = 21.7, p = .06, due to improvements in the TXT group only. However, the scores from this group still remained in the clinical range. Results for individual BRIEF Behavioral Regulation subscales revealed a significant treatment effect for Emotional Control, F(2, 21) = 4.29 , p = .03, (see Figure 2), and a

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Table 5 Change in Parent-Rated EF, Behavior, and Social Skills as a Result of Therapy. TXT group

DTC Group

M (SD)

M (SD)

p value

Effect Size

Baseline: 78.3 (7.0) Posttest: 75.1 (9.7) Baseline: 81.1 (11.7) Posttest: 75.5 (11.9) Baseline: 77.7 (8.9) Posttest: 75.9 (11.6) Baseline: 77.1 (14.7) Posttest: 73.7 (13.8) Baseline: 74.6 (11.2) Posttest: 67.0 (13.4) Baseline: 67.6 (10.5) Posttest: 64.7 (9.5) Baseline: 69.6 (7.2) Posttest: 68.4 (6.9) Baseline: 65.5 (12.3) Posttest: 67.0 (14.9)

Baseline: 83.2 (6.0) Posttest: 83.0 (6.0) Baseline: 84.5 (8.3) Posttest: 83.8 (7.5) Baseline: 79.2 (8.7) Posttest: 80.3 (9.2) Baseline: 82.5 (9.1) Posttest: 80.6 (9.9) Baseline: 78.4 (6.2) Posttest: 75.8 (5.8) Baseline: 73.2 (6.1) Posttest: 72.5 (8.4) Baseline: 73.8 (5.7) Posttest: 72.9 (7.5) Baseline: 69.0 (15.5) Posttest: 72.5 (22.3)

.06

.103

.01

.189

.09

.085

.15

.050

.03

.170

.08

.095

.25

.020

.25

.028

Measures (T-Scores) BRIEF General Executive Composite BRIEF Behavioral Regulation BRIEF Inhibit BRIEF Shift BRIEF Emotional Control CBCL Externalizing Problems CBCL Total Problems SSIS Social Skills

Standard Scores

(a) 90 85 80 75 70

Behavioral Regulation

TXT DTC

Baseline

Posttreatment

Time

T-Scores

(b) 80 75 70 65 60

BRIEF Emotional Control

TXT DTC

Baseline

Posttreatment

Time Figure 2 Change in (a) Behavioral Regulation and (b) Emotional Control scores as a function of therapy.

trend-level effect for Inhibition, F(2, 21) = 1.96, p = .09, whereas Shifting Behavior remained unchanged (see Table 4). For the CBCL, a trend-level effect was observed for the Externalizing Behavior Problems scale, F(2, 21) = 34.6, p = .08, reflecting fewer problems at posttest than baseline in TXT, compared with DTC whose scores were unchanged. Despite this improvement in the TXT group, scores still remained in the clinical range. No treatment effects were observed for the CBCL Total Behavior Problems or SSIS Social Skills scores.

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Table 6 Mean (SD) Scores in TXT Group at 6-Month Follow-Up. TXT group M (SD) Measuresa BRIEF Behavioral Regulation BRIEF General Executive Composite CBCL Externalizing Problems BRIEF Inhibit BRIEF Shift BRIEF Emotional Control

Posttest 6-month follow-up 79.1 79.3 69.4 78.9 76.6 70.6

(12.1) 78.9 (11.8) (7.2) 79.6 (8.7) (6.0) 65.1 (9.3) (8.7) 74.6 (10.6) (16.3) 78.8 (15.5) (14.0) 72.9 (12.6)

p value ns ns ns .01 ns ns

a

Results presented as T-scores (M = 50; SD = 10).

Results from parent-questionnaire data obtained at 6-month follow-up in nine TXT cases revealed that treatment effects observed at first posttest were sustained after 6 months, while an improvement on the Inhibit subscale of the BRIEF was also noted (Table 6). DISCUSSION The current study was designed to determine whether a treatment program targeting self-regulation would result in improvements in self-regulation that would also generalize to other areas of EF, behavior, and social skills in children with FASD. Our main finding suggests that this therapeutic approach resulted in parent-rated improvements in selfregulation with some gains seen on the direct child measures. On the parent-reported measures, positive outcomes were described in the TXT group’s general behavior, as well behavioral regulation. The most robust finding reflected the improvements in emotion regulation according to parent, thus suggesting that aspects of emotion regulation may be the most sensitive to Alert treatment. Notably, these results were maintained at the 6month follow-up, with parents reporting further improvement in inhibitory control. On the child measures, the TXT group improved on one of the cognitive EF measures, which involved inhibiting a cognitive response. However in the socioaffective EF domain, children showed improved ability in recognizing the emotions of others and solving social cognitive problems requiring emotional understanding. Results from the present study are consistent with a previous study that used Alert with children with FASD (Wells et al., 2012). Our results similarly indicated improvements in parent-rated EF abilities along with significantly improved behavioral regulation, particularly emotional control. Although children in the TXT group showed improvements in several areas of EF, not all aspects improved following treatment. In fact, in both cognitive and socioaffective domains, it appears that children showed the greatest improvements on simple (e.g., inhibition naming) rather than complex EF tasks. The TXT group also showed improved ability to recognize basic emotions in others with their scores on the NEPSY Affect Recognition subtest coming into the average, from clinical, range of functioning after Alert therapy. Interestingly, scores for the DTC group also improved on the False Belief and Personalized Emotions subtests, suggesting some practice effects; however, these scores remained well below age expectations in both groups. A failure to demonstrate improvements in parent-rated social skills is surprising when considered alongside the well-documented relationship between EF and social

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competence, especially since some aspects of children’s EF improved following treatment (Yeates et al., 2007). One reason may be that children with FASD need “hands-on” direct instruction with specific social situations, rather than abstracting general therapeutic concepts to real-world situations (Bertrand, 2009; Green, 2007; Kodituwakku et al., 2006). This seems especially plausible when considering the extremely poor baseline performance of children on the various socioaffective EF tasks and the difficulties with concept formation and abstraction documented in children with FASD (McGee, Fryer, Bjorkquist, Mattson, & Riley, 2008). Current findings also support research showing that children with FASD are particularly challenged by multicomponent tasks, as children did not show improvements on the more complex tasks in our battery. As task demands increase, children with FASD may need more domain-specific cognitive problem-solving strategies (Green, 2007). The issue of “task” complexity is particularly relevant when considering the broader treatment literature for children with FASD. For example, a recent study observed “domain specific” improvements in the hostile attributions of children with FASD following a social skills treatment, with the children who benefited the most from the therapy being those with the most intact EF abilities at baseline (Keil, Paley, Frankel, & O‘Connor, 2010; Schonfeld, Paley, Frankel, & O’Connor, 2009). Combining a “domain specific” approach (Coles, Strickland, Padgett, & Bellmoff, 2007; Kable, Coles, & Taddeo, 2007; Keil et al., 2010) with a “domain general” approach, as used presently, may in fact provide a more holistic treatment that meets the unique needs of children with FASD. While statistically significant results were found, the clinical significance of the results remains unclear. On measures of basic inhibitory control, recognizing emotions in others, and solving social cognitive problems, scores for the TXT group improved into the normal range, whereas on parent-rated measures, improvements were seen but scores remained in the problematic range. According to O’Connor et al. (2006), these findings should not be considered surprising given that children with FASD are characterized as having significant brain pathology that may restrict the degree of improvement they can receive from any type of intervention. Nevertheless, the question of degree of improvement in children with FASD is important and warrants examination, especially when considering that other populations of children with brain injury often do not display a complete return to functioning following injury, particularly if it occurred early in development (Eslinger, Flaherty-Craig, & Benton, 2004). In addition, the current study did not examine important variables related to the treatment process that may have contributed to treatment improvements, such as the therapeutic relationship. Discussion is also warranted as it pertains to our conceptualization of self-regulation and EF. For the present study self-regulation was conceptualized as reflecting a biologically driven cognitive and socioaffective component of EF, albeit distinct. However, as is pointed out by Blair and Diamond (2008), self-regulation and executive functioning may in fact be distinct constructs working in a bidirectional manner that is mediated by the child’s environment, particularly caregiver response. In other words, children with FASD, who come in to the world with poor EF, may be expected to do poorly by caregivers and teachers. Therefore, an alternative explanation for the current findings that reflect greater behavioral than cognitive improvements is the potential dual impact of this therapeutic approach that allows for positive shift in the caregiver/child environment. Specifically, the guidance/support provided to caregivers as cofacilitators of change, by equipping them with knowledge of Alert tools, allowed reinforcement of these tools beyond the context of therapy allowing for generalization. In so doing, a new relational template may have been

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created for the child/parent, which may be equally as important as the acquisition of new skills in children with FASD. It will be important for future studies to examine potential mediators of treatment outcomes in children with FASD. Despite these significant findings, some limitations need to be addressed. First, few of our child-administered results approached statistical significance and results were not corrected for multiple comparisons. Given our small sample size, we would have needed a very large effect to use the correction, which would likely have washed out these findings. Second, although our sample was sufficient to detect treatment effects, we were unable to examine additional important factors related to outcome, such as IQ, gender, and placement status, which also undoubtedly contributed to outcome. Third, as we used standardized scores rather than raw scores, it is possible that some treatment effects may have been washed out if more children in TXT moved into a higher age category than the DTC group over time. Fourth, it is possible that the current findings were influenced by the differential group differences at baseline in terms of the number of cases with ADHD and secondary drug exposures. Regarding ADHD, all of the children were being medically treated and the baseline scores did not differ between groups. A sixth concern is that our sample may not generalize to the larger FASD population since we excluded children with an IQ of 70 or below and had no cases with FAS. We also did not exclude for comorbidities. Further modifications to the protocol will need to be made and evaluated to accommodate children with FASD with lower cognitive functioning. Although the literature on psychotherapeutic approaches to childhood disorders typically excludes children with comorbidities, because most children with FASD are diagnosed with other conditions, this is inherent to the FASD profile and so it is not possible to obtain a “pure” FASD sample. However, a larger sample size would have allowed us to examine the impact of other conditions on treatment outcome. In conclusion, although this study represents only the second controlled treatment approach for improving the self-regulatory and EF abilities of children with FASD, it is the first to use a broad array of outcome measures and a theory-driven perspective. Most promisingly, it was demonstrated that a relatively brief intervention can lead to significant improvements within a clinically complex sample. Furthermore, the current findings highlight that children with FASD indeed benefit from Alert therapy and provide hope that best practices for treatment can be developed for children with FASD. Original manuscript received April 18, 2013 Revised manuscript accepted January 25, 2014 First published online March 28, 2014

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Improving executive functioning in children with fetal alcohol spectrum disorders.

An extensive body of literature has documented executive function (EF) impairments in children with fetal alcohol spectrum disorders (FASD); however, ...
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