Psychosocial issues for children with epilepsy

Epilepsy & Behavior 22 (2011) 47–54

Contents lists available at ScienceDirect

Epilepsy & Behavior j o u r n a l h o m e p a g e : w w w. e l s ev i e r. c o m / l o c a t e / ye b e h

Review

Psychosocial issues for children with epilepsy☆ Roos Rodenburg a, b, Janelle L. Wagner c, d, Joan K. Austin g, 1, Michael Kerr e, David W. Dunn f,⁎ a

Research Institute of Child Development and Education, University of Amsterdam, Amsterdam, The Netherlands SEIN-Epilepsy Institute in the Netherlands Foundation (SEIN), Heemstede, The Netherlands c Departments of Nursing and Pediatrics, Medical University of South Carolina, Charleston, SC, USA d Comprehensive Epilepsy Center, Medical University of South Carolina, Charleston, SC, USA e Welsh Centre for Learning Disabilities, School of Medicine, Cardiff University, Cardiff, UK f Departments of Psychiatry and Neurology, Indiana University School of Medicine, Indianapolis, IN, USA g Department of Environments for Health, Indiana University School of Medicine, Indianapolis, IN, USA b

a r t i c l e

i n f o

Article history: Received 20 April 2011 Accepted 21 April 2011 Available online 25 June 2011 Keywords: Childhood epilepsy Psychosocial problems Family problems in epilepsy Academic difficulties and epilepsy Emerging adulthood and epilepsy

a b s t r a c t Epilepsy is a pervasive disorder that consists not only of seizures, but of behavioral, academic, and social difficulties. Epilepsy has an impact on the entire family and may have a significant effect on the interrelationships between child and parent. Epilepsy also has a potentially deleterious effect on academic functioning that may be the result of central nervous system dysfunction, seizures, antiepileptic drugs, or child and family response to illness. Early assessment for psychosocial problems and appropriate interventions can be beneficial for the child and family. Particular attention should be paid to periods of transition such as the move from adolescence to adulthood. © 2011 Elsevier Inc. All rights reserved.

1. Introduction Epilepsy in children is more than recurrent seizures and antiepileptic drugs. Epilepsy may have a direct effect on the behavior and cognition of a child, but understanding the response of the child requires an awareness of the effects of epilepsy on the family and surrounding community. In the next three sections, we review current research and future directions for assessment and treatment of the psychosocial problems of children with epilepsy. We begin with reviews of the psychosocial issues for the child within the family (Section 2) and the child in the academic setting (Section 3). This is followed by an examination of the important issue of transition from childhood into adolescence and emerging adulthood, including both risk factors and interventions that will determine a successful outcome for children with epilepsy (Section 4). The fourth and fifth sections highlight the current work of two young investigators. Dr. Rodenburg discusses the bidirectional association of changes in the behavior of the child with the relationships in the family and the response of the parent to the challenge of epilepsy that helps to ☆ From a special issue of Epilepsy & Behavior: "The Future of Clinical Epilepsy Research" in which articles synthesize reviews from senior investigators with the contributions and research directions of promising young investigators. ⁎ Corresponding author at: Departments of Psychiatry and Neurology, Indiana University School of Medicine, 702 Barnhill Drive, ROC 4300, Indianapolis IN 46202 USA. Fax: + 1 317 948 0609. E-mail address: [email protected] (D.W. Dunn). 1 Distinguished Professor Emerita, Indiana University School of Nursing. 1525-5050/$ – see front matter © 2011 Elsevier Inc. All rights reserved. doi:10.1016/j.yebeh.2011.04.063

shape the emotional and cognitive outcome of the child. Dr. Wagner presents initial data on attempts to intervene by teaching the child and parents management techniques to limit adverse effects of epilepsy. 2. Family environment and parenting 2.1. Joan K. Austin The family environment has been found to be important in the psychological development of children with a chronic illness. Children with epilepsy also have behavior problems at rates higher than those in children with other chronic conditions [1]. Children with epilepsy are especially at risk for behavioral disorders related to attention and internalizing behaviors, such as anxiety and depression symptoms. Among the risk and protective factors associated with these problems are those related to the family environment and parenting. Findings from studies investigating both seizure and family variables as predictors of child behavior commonly show that family variables are the more strongly associated [2]. Research on the nature of the relationship of the family environment and parenting with child behavior problems, however, is still in its early stages and many issues remain to be studied. We propose that the issues surrounding the complex interrelationship of family and parenting factors with child behavioral outcomes currently shape some of the most important clinical research questions in our field. We identified three broad areas for discussion: (1) identifying the impact of epilepsy on the family and parenting; (2) delineating the interrelationship of family variables

48

R. Rodenburg et al. / Epilepsy & Behavior 22 (2011) 47–54

and child behavior problems; and (3) developing interventions that enhance family functioning and parenting as well as improve behavior in children. 2.1.1. Impact of epilepsy on the family Dealing with a child's epilepsy can be stressful for the entire family. Although the level of stress may vary over time, it appears to be especially high at onset. Parents observing their child's first seizure report fearing that their child was dying [3]. Comorbidities such as cognitive deficits and learning problems can be an additional stressor for the family. Living with an episodic and unpredictable condition such as epilepsy has been found to be more disruptive for families than conditions that are more stable and predictable [4]. Stress in the family environment has the potential to disrupt parenting behaviors and erode parents’ confidence in their ability to parent their child. Comparison studies show that families of children with epilepsy have more relationship and parenting difficulties and, thus, more disrupted environments than families of children with other chronic conditions [5]. Because most research has used a cross-sectional design, we do not have a good understanding of the natural history of the processes that families use to successfully manage childhood epilepsy. Further work is needed to identify family variables that serve as risk and protective factors for child behavior problems. A recent study by Thornton et al. [6] suggests that children who are at higher biological risk do less well in poorly functioning families. Comprehensive, prospective studies are needed to identify which families and children are most at risk and, hence, most in need of interventions. 2.1.2. Delineating the interrelationship of family variables and child behavior problems Until recently, studies investigating the relationship of family variables to child behavior problems focused on children with chronic epilepsy, making it difficult to determine the causal sequence of the relationship. It was commonly assumed that a negative family environment and dysfunctional parenting were major causes of child behavior problems. However, recent prospective studies of children with new-onset seizures show that child behavior problems can precede the onset of epilepsy [7]. These findings suggest that behavior problems in children might contribute to disruption in the family environment. There is a need to increase our understanding of the complex interaction between the family environment and the child's behavior to provide a foundation for developing family interventions. Using family theoretical frameworks to guide the selection of family variables should help identify family variables that should be targeted. It is especially important to identify specific family and parenting variables that both are amenable to intervention and reduce child attention difficulties and internalizing problems. 2.1.3. Developing family interventions The ultimate goal is to have family interventions that are effective in enhancing the family environment and parenting behaviors while diminishing child behavior problems. Epilepsy is difficult for families because of its unpredictable nature, potential for injury, and associated cognitive and behavioral comorbidities. Childhood epilepsy also varies widely, with some children having no problems and others experiencing uncontrolled seizures and developmental delay. Moreover, parents react differently to their child's epilepsy, with some having more difficulty in coping than others. Major parenting tasks include: managing the epilepsy and its treatment, facilitating the child's accomplishment of developmental tasks, and helping the child successfully cope with having a seizure condition. To successfully meet these challenges, parents will need to make a healthy adjustment to the epilepsy themselves. Interventions are needed that will facilitate parents in their accomplishment of these tasks. A few interventions have been tested and others are currently under development. Tested interventions show parent improvement

in epilepsy knowledge and management of seizures; however, they have been less successful in improving child behavior [8]. Such a diverse population suggests that one family intervention will not work for all families and that interventions will need to be tailored to the needs of the child and the family. For example, interventions might need to differ with seizure variables (e.g., new onset, intractable epilepsy), child development (normal, delayed), or the type of behavior problems experienced by the child (attention problems, internalizing problems). It will be important to identify which families need which type intervention at what point in the epilepsy trajectory. 3. School and academic achievement 3.1. David W. Dunn A primary developmental task of childhood is successful participation in the academic arena. Children 6–18 years of age spend approximately half their waking hours in school and their academic performance has a major impact on self-esteem and subsequent entry into higher education and employment. Children with epilepsy are at risk for academic underachievement. Even those children with epilepsy and normal intelligence have an increased risk of academic underachievement. In a recent study of children with epilepsy, Fastenau et al. [9] found at least one learning disability in 48% using the IQ-discrepancy model and one learning disability in 41–62% using the low achievement model. In studies of children with new-onset seizures, McNelis et al. [10] and Fastenau et al. [11] found no difference between children with seizures and siblings at baseline. However, by 24 months, McNelis et al. [10] found lower scores in comparison to national norms, and by 36 months, Dunn et al. [12], in a follow-up of the sample described by Fastenau et al. [11], documented lower achievement scores in children with epilepsy as compared with siblings. No areas are spared. Children with epilepsy have lower scores than siblings, chronic disease controls, and general population children in reading, writing, math, and general information. Just as seen with behavioral problems, the potential causes for underachievement are multiple, and academic problems are probably caused by the interactions of a variety of factors. Potentially important factors are neurological dysfunction, seizures, medications used to suppress seizures, and psychosocial factors. Biological factors that result in cognitive problems may predate or follow the onset of seizures. Berg et al. [13] found that children with symptomatic seizures were more likely to require special education services than children with idiopathic or cryptogenic seizures, and in 54% of the sample of children with symptomatic seizures, special education services were begun prior to the first seizure. Academic underachievement may also be the result of seizures. Early age at onset and persistent seizures are risk factors for cognitive difficulties found in many studies [10,14], but not all [15]. Aldenkamp and Arends [16] found that frequent epileptiform discharges and nonconvulsive seizures are associated with slowing of processing speed, difficulty with attention, and impairment of short-term memory. This same group also suggested that seizure syndrome, specifically symptomatic generalized and localization-related epilepsies, adversely affect psychological function, which, in turn, results in academic underachievement; an association was not found with idiopathic epilepsies [17]. Assessment of new-onset seizures over time is limited. Most studies do not show significant decline over time [18], particularly in those children with no additional comorbidity [19,20]. Related to neuropsychological functioning is the association of symptoms of attention-deficit/hyperactivity disorder (ADHD) with academic underachievement. Williams et al. [21] showed that symptoms of ADHD are a better predictor of academic underachievement than memory function or socioeconomic status. Fastenau et al.

R. Rodenburg et al. / Epilepsy & Behavior 22 (2011) 47–54

[9] found that ADHD is the strongest predictor of reading and math disability using a 1.0 SD low-achievement model. Hermann et al. [20] found that children with epilepsy and no comorbidity are similar to controls in cognitive function, but those with epilepsy and ADHD had significantly worse cognitive function. Sleep disruption may be a comorbidity of epilepsy and can be the result of seizures and epileptiform activity during sleep, antiepileptic drugs (AEDs), or behavioral problems. Sleep disturbance can also contribute to academic difficulties. Byars [22] found an association between worse neuropsychological functioning and sleep disturbance in children with new-onset seizures. Parisi et al. [23] offer the hypothesis that epileptiform activity may disrupt the slow wave sleep that is essential for learning. Academic difficulties have been blamed on AEDs; however, data on the cognitive effects of AEDs are limited. Phenobarbital has an adverse effect on cognition. Of the newer AEDs, topiramate has been associated with impaired attention, and in a recent study, children with absence seizures had more problems with attention while on valproate compared with ethosuximide or lamotrigine [24]. More often, the adverse effect of AEDs on academic function are idiosyncratic and require the physician to monitor all school-aged children with epilepsy for adverse cognitive effects of an AED. The child with epilepsy exists within a family and in the larger community. Multiple psychosocial factors may affect academic function. In the child with epilepsy, negative attitudes toward illness, low self-esteem, and poor motivation have been associated with poorer academic achievement [25–27]. Family factors are major contributors to academic performance. Family competence has been shown to be a moderating factor in academic achievement [27–29]. Dunn et al. [12] found that anxiety in parents moderated the association between change in language and attention/executive/ construction neuropsychological function and change in writing achievement. Perception of academic success may be influenced by the stigma associated with epilepsy. Katzenstein et al. [30] found that there was no association between knowledge of a child's diagnosis and performance on standardized tests, but teacher knowledge that the child had epilepsy was associated with lower rating of academic performance. Several questions remain unanswered. Most reports are crosssectional and very few follow children prospectively with adequate measures of neuropsychological function and academic achievement. The long-term studies are mostly clinic-based and not populationbased studies. Predictive factors have been studied in children with normal IQ, but there needs to be better assessment of the effects of epilepsy-related and psychosocial factors on academic achievement in children with intellectual disability. Beyond knowing factors that predict academic underachievement, we need studies that show when children should be assessed and what interventions are most likely to be effective. Genetic and pharmacogenetic studies should help identify children who might be at increased risk for adverse cognitive effects of specific AEDs. The significant contribution of psychosocial factors suggests that behavioral interventions could be efficacious in this population. 4. Transition of people with epilepsy through adolescence into adulthood 4.1. Michael Kerr Although childhood epilepsy has many unique characteristics and for many, up to 60%, it is a self-limiting condition [31], for others it is associated with an inevitability of growing older and transitioning into adulthood. Even for those without seizures it appears that lingering negative social and cognitive concerns remain. Furthermore, this progression into adulthood occurs across the range of childhood epilepsy and across the range of children, that is, those with and

49

without physical or cognitive disabilities. The adolescent epilepsy population can be predicted to have an excess of certain risk factors such as: associated intellectual disability, previous neonatal seizures, and a high pretreatment seizure frequency. The importance of transition for those with complex disabilities reflects an increasing life expectancy in this population. We believe that issues around this transition constitute some of the key clinical and research questions in our field. We expand on these in more detail later, but in summary the issues are: (1) reducing the impact of mental illness through adolescence and transition, (2) identifying factors to reduce the impact of chronic disease on social integration and participation, and (3) understanding and developing service continuity into adulthood. Although the population with childhood epilepsy is by no means homogenous the themes apply to all though their delivery will need adaptation depending on, in particular, the individual's cognitive ability. 4.1.1. Risk prediction and reduction Adolescents with epilepsy are at a particularly high risk of mental ill health and associated suicidality, both as compared with the general population and as compared with adults with older-onset epilepsy [32]. The risk seems to be associated with the severity of the epilepsy, but other factors are likely to be important. This is an important area for further investigation and advances in clinical practice. More work is needed to identify risk factors for mental ill health and associated suicidality and to develop programs of care to identify and treat illness at its onset rather than allow the development of chronicity. Key research themes are aimed at understanding the development of psychopathology from childhood through adolescence and into adulthood, to identify amenable factors and predictors and to develop integrated care packages to support these vulnerable individuals. The work should specifically address the interface between psychiatric and neurological practice, an area where this population needs focused shared care. 4.1.2. Identifying factors to reduce the impact of chronic disease The impact of chronic disease on health-related quality of life is seen across a range of pediatric chronic conditions [33]. Epilepsy is perceived by adolescents as having a greater negative social impact then other chronic diseases [34]. Individuals with epilepsy often suffer from multiple comorbidities, with psychiatric problems prevalent in those of higher IQ and a range of complex psychiatric and physical problems present in those with intellectual disabilities and epilepsy. Despite this we know little of how to reduce the impact of chronic disease in these young individuals. Interventions designed to ameliorate the impact of chronic disease in childhood are therefore needed to reduce the lifelong impact. For those with more complex problems, greater individualization of outcome is needed. A greater recognition of the etiology of the intellectual disability through advances in genetics is allowing a greater number of etiologies to be identified in individuals progressing into adulthood. Prospective studies are needed to predict the occurrence of comorbidities in such individuals. This is reasonably clear in conditions such as Down syndrome, but remains less so for the majority of conditions. 4.1.3. Service continuity into adulthood Transition into adult care is fragmented and patchy. Although varied service models have been identified, such as transition clinics and adolescent clinics (see, e.g., Stephen et al. [35]), there is no consistency. Barriers to the transition of persons with neurological disease into adult care have been identified [36]: (1) failure of the pediatric team to let go, feeling adult services are inferior; (2) the difficulty faced by adolescents in coming to terms with adult doctors; (3) the family's concerns over the adult environment and loss of control; and (4) the adult team's lesser familiarity with pediatric

50

R. Rodenburg et al. / Epilepsy & Behavior 22 (2011) 47–54

conditions and multidisciplinary team work. This transition occurs while issues of mental ill health and chronic disease impact are high, compounding any negative impact. Detailed work is needed to develop and track models of service delivery that can achieve continuity yet also deliver the elements of chronic disease management that are necessary to provide appropriate support (see Fig. 1). Of particular importance in adolescents with epilepsy are self-management, decision support, and design of the care delivery system. 4.1.4. Conclusion Transition into adulthood is a time of risk and uncertainty for people with epilepsy and their families—a risk reflected in psychopathology and social outcomes. Reduction of this risk is possible with research that can encompass basic science to understand genetic susceptibility, epidemiology identifying environmental and epilepsy-related variables, through to the social sciences informing the experiential pathway of those with the condition. All of this will impact our clinical care delivery. 5. Promising Areas of Research and Young Investigators 5.1. Roos Rodenburg Children with epilepsy: Child development and family perspectives- Toward long-term outcomes and treatment efficacy It is well known, particularly since the Isle of Wight study [1,37– 39], that children with epilepsy are at high risk for behavioral problems. It was less clear; however, which behavior problems were commoner in children. I started my research with the overall goal of clarifying the issue of which behavior problems predominate. With this in mind, the results of studies of children with epilepsy were meta-analyzed and compared with respect to the severity of behavioral problems with children from the normative population and general populations, children with other chronic conditions, and siblings [40]. When compared with children from the normative population, children with epilepsy were particularly at high risk for attention and social problems and somatic complaints. Internalizing problems (e.g., anxiety/depression) seemed more severe than externalizing problems (e.g., rule-breaking behaviors). Comparisons with children with other chronic conditions and siblings suggested that the risk for behavioral problems was actually decreased. This may indicate that psychopathology in children with epilepsy is likely due

to the chronicity of the condition and family factors may contribute. Attention, thought, and social problems, however, seemed more specific to epilepsy: children with epilepsy were at higher risk for these problems when compared with children with other chronic conditions and siblings. The role of psychosocial and family factors in behavioral problems in childhood epilepsy has long been recognized [41–44]. As each family factor is differentially influential on child outcome [45,46], with my colleagues, I developed a distinct conceptual family factor framework on the basis of social ecology principles [47,48]. Depending on the proximity to the child's everyday life, proximal factors (parenting and parent–child relationship quality), distal factors (parental characteristics, e.g., parental depression), and contextual factors (other family relationships) were distinguished [49]. Applied in a review of family factors in families with a child with epilepsy [50] less positive family factor outcomes (e.g., lower parent–child relationship quality, more parental depression) were found when compared with control families. At the proximal level, negative relationships were found between parental psychological control (parenting aimed at the person of the child by, e.g., inducing guilt or shame) and child behavior problems. Negative relationships between distal and contextual factors and behavioral problems were also found. To examine whether proximal family factors contribute most to child behavior problems, I and colleagues subsequently made a simultaneous investigation of epilepsy factors, parent–child relationship quality, parental characteristics, and other family relationships in relation to child behavior problems [5]. Children with epilepsy and normal intelligence were at high risk for internalizing, attention, and thought problems. Proximal family factors, but not epilepsy factors, significantly and to the largest extent predicted child behavioral problems. Parent–child relationship quality, in particular, mediated the effects of distal and contextual family factors on behavioral problems, indicating that parental characteristics and poor family relationships spill over into less adequate parent–child interactions. This in turn, contributed to higher levels of behavioral problems. Of interest was that family factors were also strong predictors of the behavioral problems identified earlier as being more specific to having epilepsy (e.g., attention problems) [40]. This may point to a contribution of family factors later in the course of epilepsy where epilepsy may have contributed earlier around epilepsy onset. Parenting is thus an important contributor to child behavioral problems in epilepsy. As parenting stress is a determinant of parenting

Fig. 1. The Chronic Care Model.

R. Rodenburg et al. / Epilepsy & Behavior 22 (2011) 47–54

[51], we investigated in depth the role of parenting using the ABCX model of stress and adaptation [52]. Parenting stress was studied as a mediator between, on the one hand, stressors, resources, and coping behaviors and, on the other hand, parenting [53]. Parenting stress and stressors contributed most to different types of parenting (support, behavioral control, psychological control, and parent–child relationship quality). Parenting stress most consistently mediated the effects of stress, resources, and coping behaviors on parental behavioral control. It thus seems that in the context of epilepsy, parental control is especially sensitive to parenting stress. Important for clinical practice is that if a child with epilepsy has behavioral problems, parenting stress and parenting are targets that can be intervened with relatively easily. Our group is currently examining which parenting behaviors are most impaired in the context of raising a child with epilepsy. As parenting is multidimensional [54], knowledge of the particular dimension(s) of parenting with which parents experience the most problems will help us to fine-tune the guidance for parents. We have substantial (macro-level) knowledge about the contributors to behavior problems, but this does raise new questions. For example, parenting is an important contributor, but why is it? We have now also included parental perceptions of epilepsy and child vulnerability [55–57] to examine whether these affect parenting practices. In addition, knowing that parenting stress affects parenting [58] begs the question: What happens in real time when parents feel stressed? To this end, we are currently examining daily stress in mothers of children with epilepsy to capture the micro-level antecedents and consequences of maternal parenting stress. From this knowledge, we will develop an intervention that aims to reduce parenting stress. The assumption is that this positively affects parenting practices and, in turn, children's behaviors. Interestingly, if epilepsy factors are simultaneously investigated with family factors, family factors are mostly detected as significant contributors [5,59,60]. Why do we find much more consistent evidence for the contribution of family factors? It may be due to the lack of conceptual distinctions in measuring the impact of epilepsy. Epilepsy impact itself can be disentangled into brain-related factors (e.g., central nervous system dysfunction) and non-brain-related epilepsy factors (e.g., parental fear for seizures) [61]. Furthermore, different measures have been used to investigate epilepsy impact and mixed results have thus far been obtained. Some have pleaded for a more unified measurement of condition severity, which should include a measurement of epilepsy syndrome severity (to measure central nervous system impact) along with other indices of epilepsy factors such as seizure frequency and severity [62]. Syndrome severity has been little investigated in childhood epilepsy, but it is considered to be of importance for the prediction of child behavior problems. New perspectives should help us to investigate further, beyond behavior problems. For example, children with epilepsy are at high risk for anxiety [63–67], including specific phobias, panic disorder, and posttraumatic stress symptoms (PTSD) [68]. Parenting appears to be a major contributor to such problems, but the role of epilepsy needs to be elucidated. That is, high rates of anxiety may reflect seizure-related anxiety symptoms, a phenomenon that has been reported, but not examined, to revolve around the seizures /epilepsy in 20–30% of patients [69]. Anxiety, stress, and emotions can trigger seizures and increase seizure frequency [70]. These, in turn, can also trigger anticipatory anxiety and evoke stress, anger, and frustration. This may lead to a vicious circle of anxiety and seizures maintaining each other [71]. Thus, the challenge is to investigate in how many children have anxiety-related seizures related. We conducted a pilot study to estimate the percentage of children with a clinically diagnosed relationship between anxiety and seizures/epilepsy [72]. Twelve of 31 children (38%) had clinical anxiety, and 6 of them had seizure/epilepsy-related anxiety. Children with an established relationship between seizures and anxiety are currently being treated with eye movement desensitization and

51

reprocessing (EMDR), an effective anxiety/trauma treatment for children [73,74]. We choose to screen for seizure-related anxiety and the subsequent treatment of anxiety as a research gap exists regarding the testing of treatment efficacy in children with epilepsy [75,76], as is true for children from other pediatric populations [77,78]. Pediatric epilepsy researchers are encouraged to investigate the efficacy of evidence-based treatments for nonpediatric populations (e.g., behavioral therapy or EMDR) in pediatric epilepsy populations. Case study designs are valuable to start with testing for treatment efficacy [79,80]. An N = 9 experimental group and N = 9 control group design is sufficient for determining treatment efficacy [81,82]. Much childhood epilepsy research to date is cross sectional in nature. Longitudinal studies such as routine outcome measurement (ROM) are needed to gain more insight into long-term developmental outcomes [83–85] of psychopathology, cognition, family factors, healthrelated quality of life, epilepsy factors, and medical/neurological parameters. Hermann and Whitman's conceptual model [61] is valuable for defining the outcome parameters on distinct levels. As an advantage, this model can integrate the child developmental framework of the researchers’ choice [e.g., 86]. Testing for treatment efficacy can be incorporated into clinical practice by using ROM. As such, ROM may help answer the research issues raised here: the life-span course of epilepsy [87]; social ecological and family factors explaining child developmental outcomes [20,29,88–90]; the use of unified measurements and epilepsy impact conceptualizations; the relationship between seizures and anxiety; and psychological treatment efficacy. I hope that researchers and clinicians worldwide will be able to address these issues so that our knowledge will expand and will be applied rapidly in pediatric epilepsy clinical practice.

6. Promising Areas of Research and Young Investigators 6.1. J.L. Wagner Assessments and interventions for children with epilepsy and their families My program of research encompasses evidence-based psychosocial assessment and intervention within the context of pediatric epilepsy self-management. Self-management has been defined not solely as adherence to a treatment regimen, but as encompassing personal and family resources, including cognitive and behavioral coping skills, needed to manage a chronic condition in the context of everyday life [91]. As a pediatric psychologist, I have the background to specialize in cognitive and behavioral evaluation and treatment and have chosen to apply these skills in researching psychosocial adjustment and self-management intervention effectiveness for youth with epilepsy (YWE). For the past 7 years, I have had the pleasure of collaborating with individuals in various disciplines in the field of pediatric epilepsy, most notably Ms. Gigi Smith at the Medical University of South Carolina (MUSC). As a doctoral fellow, I contributed to the epidemiological study South Carolina Health Outcomes in Persons with Epilepsy (SC HOPE) funded by the Centers for Disease Control and Prevention. My role was to assist in development of a comprehensive telephone survey to measure mood, behavior, access to care, quality of life, and other factors [92]. Qualitative findings from focus groups revealed that YWE in South Carolina face many physical and psychosocial challenges, compounded by stigmatization [93]. During my fellowship, I also examined psychological referrals in the MUSC pediatric epilepsy clinic [94] and critically reviewed the extant literature on psychosocial interventions for YWE [95]. Unfortunately, my review revealed that few studies have examined the effectiveness of psychosocial interventions for YWE and only two focused on self-management. Many of the studies cited design limitations and recruitment barriers, resulting in small sample sizes [95].

52

R. Rodenburg et al. / Epilepsy & Behavior 22 (2011) 47–54

6.1.1. Study findings. As an early career investigator, grant support from the Epilepsy Foundation (EF) has been critical to my success as a researcher. Building on my experiences with SC HOPE, with funding through the Targeted Mood Initiative, I examined caregiver and youth perceptions of epilepsy and how these perceptions related to mental health symptoms in an effort to identify targets for psychosocial selfmanagement intervention. Eighty-two youth completed evidencebased assessment. Only 6 participants reported clinical depressive symptoms; however, 28% endorsed suicidal ideation. Further, selfreport of hopelessness mediated the attitude toward the epilepsy– depressive symptom relationship, and attitudes toward epilepsy and self-efficacy were independent predictors of depressive symptoms [96]. Thus, these cognitive appraisal mechanisms related to epilepsy experiences are important to consider when intervening with YWE. More specifically, these results suggest interventions that assist youth in identifying epilepsy-related aspects of functioning over which they can realistically exercise control and challenging negative thoughts about situations they cannot control. This investigation also revealed that a longer recovery time from the last seizure and longer seizure duration influence how severe caregivers judge seizures, suggesting that caregiver perceptions are important considerations when rating seizure severity [97]. Epilepsy health care providers are encouraged to consider this when assessing seizure severity during clinical visits. A second study funded by the Partnership for Pediatric Epilepsy Research [EF, Parents Against Childhood Epilepsy (PACE), and the American Epilepsy Society (AES) ] received the William R. Turk, M.D. Pediatric Epilepsy Research Award. This study examined the development of a youth depression screening measure and the facility of this tool in informing mental health referrals. We revised the Neurological Disorders Depression Inventory for Epilepsy (NDDI-E) and created the 11 item NDDI-E-Y for YWE aged 12–17. Data collection was just completed with 93 YWE; however, preliminary analyses indicate strong test–retest reliability [98]. Validity indices indicated the NDDIE-Y has poor sensitivity but strong specificity in predicting a diagnosis of depression on the Kiddie Schedule for Affective Disorders and Schizophrenia (K-SADS). Because of the atypical clinical presentation of depression in YWE [99], we are currently looking at the ability of the NDDI-E-Y to predict individual depressive symptoms instead of a DSM-IV depression diagnosis. Results also revealed that almost half of caregivers endorsed the presence of mood and behavior symptoms in YWE. On the basis of depression and behavior screening measures, 35% of youth had significant symptoms, were not receiving mental health services, and thus were referred. These findings support routine psychosocial screening in pediatric epilepsy clinics. On the basis of our results, which identified attitudes toward epilepsy and self-efficacy for seizure management as targets for psychosocial self-management intervention, and DiIorio's work demonstrating that self-efficacy for seizure management is related to actual practice of self-management behaviors [100,101], we created Coping with Epilepsy (COPE), an epilepsy self-management group intervention for YWE and their caregivers. The core skills included in COPE were derived from two empirically supported interventions, Weisz and colleagues’ [102] Primary and Secondary Control Enhancement Training (PASCET) and Tim Wysocki's Behavioral Family Systems Therapy (BFST) [103]. COPE presents and applies these skills within the context of social learning theory-driven models of self-management in epilepsy [104]. Primary coping skills include changing behaviors in situations in which one has control over the outcome (e.g., taking prescribed medications as directed, maintaining healthy sleep habits, eating nutritional foods, setting goals, problem solving specific to epilepsy treatment regimen adherence, engaging in pleasurable activities, stress reduction/relaxation). Secondary coping skills involve changing one's perceptions of the situation when the situation itself cannot be changed, or restructuring perceptions of what it means to have epilepsy, and looking for any positive impact of epilepsy.

The COPE pilot study was funded by PACE. We enrolled nine YWE 10–15 years of age and their caregivers. Caregivers and youth endorsed satisfaction with the program, and intervention content delivered in a brief, group format was feasible [105]. Moreover, youth reported an increase in seizure knowledge and self-efficacy for seizure management, and caregivers reported enhanced coping skills in youth [106]. Improvements in attitudes toward epilepsy and caregiver distress were not significant; however, trends in the expected direction were observed. Limitations of our pilot study are similar to those of other pediatric chronic illness intervention studies [107] and include (1) suboptimal use of skills outside of sessions (33% of youth, 16% of caregivers “often used skills”); (2) difficulties with single-site recruitment necessitating deletion of control group in the study design; and (3) small sample size limiting the power to detect potential pre–post treatment differences. 6.1.2. Implications. Given the increased risk of psychosocial and behavioral symptoms in YWE, evidence-based psychosocial assessment and intervention are critical to the comprehensive care of these youth. Though many YWE do not meet DSM-IV criteria for depressive disorder, higher rates of suicidal ideation and atypical depressive symptom presentation must be recognized by clinicians. Depression screening in epilepsy clinics is necessary to identify those youth with increased risk who would benefit from a more thorough psychological evaluation and treatment. The NDDI-E-Y may be a brief instrument for depressive symptom identification; however, further analyses and future studies are necessary before the psychometrics of this measure are established. In a field where there are no evidence-based self-management programs for YWE, COPE may provide benefit to YWE and their caregivers by improving cognitive and behavioral coping skills. COPE is designed to improve self-efficacy for seizure management by helping youth and caregivers identify epilepsy-related aspects of functioning over which they can realistically exercise control and challenge negative thoughts about epilepsy-related situations they cannot control. Preliminary results are promising. Such an intervention could have remarkable significance in the comprehensive treatment of pediatric epilepsy and follows priority recommendations of Living Well with Epilepsy II [108] and consensus guidelines for mental health treatment in YWE [99]. However, a randomized, controlled clinical trial to further test the effectiveness of COPE is necessary. 6.1.3. Future directions. My research team will continue to analyze the results of the NDDI-E-Y and apply for future funding to further evaluate the psychometric properties of this instrument. In addition, my research team has applied for funding from the National Institutes of Health to conduct a three-site randomized clinical trial to examine the effectiveness of COPE. Recognizing the limitations of our pilot study, we have enhanced the research design to include (1) interactive and multimodal learning computer modules to promote practice and mastery of skills outside of group, (2) a multisite design with fewer face-to-face visits to improve recruitment and access to intervention, and (3) two structured, interactive booster sessions and follow-up assessments to closely track skill mastery and sustainability of the effect of the intervention. These changes have been made in an effort to reduce participant burden, enhance skill mastery by using computer module skill practice instead of manual self-initiated practice between groups, track treatment fidelity, and improve recruitment. 7. Conclusion Epilepsy in children can have a disruptive effect on the child, the family, and the community. The most immediate effect is on the child and the family. An initial goal should be to assist the family with the new challenge of coping with a chronic illness. The discussions by

R. Rodenburg et al. / Epilepsy & Behavior 22 (2011) 47–54

Dr. Austin, Dr. Rodenburg, and Dr. Wagner indicate the importance of the family environment in determining the long-term behavioral outcome of the child with epilepsy and the need and feasibility of child- and family-focused interventions to prevent problems. Multiple factors contribute to academic difficulties in children with epilepsy. Children with seizures must be monitored for the emergence of school problems and careful assessments should be made for biological, psychological, and social factors that could be addressed to limit longterm problems. As the child nears completion of his or her academic career, a critical and often unmet need is assistance in navigating the transition into adulthood. Collaborative research between medical and social scientists could lead to substantial improvements in the quality of life of people with epilepsy. Acknowledgments (J.L. Wagner) On the basis of my brother Aaron's personal experience with epilepsy, I chose a career in which I could potentially improve the lives of youth with epilepsy. I am grateful to all of the families who have participated in our research studies. They continue to inspire me. The AES, EF, and PACE have made it possible for my research team to conduct the aforementioned studies. I also thank the following individuals for their mentorship and/or collaboration: Gigi Smith, Dr. Joan Austin, Dr. Braxton Wannamaker, Dr. Pam Ferguson, Dr. Jonathan Edwards, Dr. Anbesaw Selassie, Karen van Bakergem, and Stephanie Hrisko. References [1] Davies S, Heyman I, Goodman R. A population survey of mental health problems in children with epilepsy. Dev Med Child Neurol 2003;45:292–5. [2] Austin JK, Caplan R. Behavioral and psychiatric comorbidities in pediatric epilepsy: toward an integrative model. Epilepsia 2007;48:1639–51. [3] Besag FMC, Nomayo A, Pool F. The reactions of parents who think that a child is dying in a seizure: in their own words. Epilepsy Behav 2005;7:517–23. [4] Rolland JS. Families, illness, and disability. New York: Basic Books; 1994. [5] Rodenburg R, Meijer M, Dekovic M, Aldenkamp AP. Family predictors of psychopathology in children with epilepsy. Epilepsia 2006;47:601–14. [6] Thornton N, Hamiwka L, Sherman E, Tse E, Blackman M, Wirrell E. Family function in cognitively normal children with epilepsy: Impact on competence and problem behaviors. Epilepsy Behav 2008;12:90–5. [7] Austin JK, Harezlak J, Dunn DW, Huster GA, Rose DF, Ambrosias WT. Behavior problems in children before first recognized seizures. Pediatrics 2001;107:115–22. [8] May TW, Pfafflin M. Psychoeducational programs for patients with epilepsy. Dis Manage Health Outcomes 2005;13:185–99. [9] Fastenau PS, Shen J, Dunn DW, Austin JK. Academic underachievement among children with epilepsy: proportion exceeding psychometric criteria for learning disability and associated risk factors. J Learn Disabil 2008;41:195–207. [10] McNelis AM, Dunn DW, Johnson CS, Austin JK, Perkins SM. Academic achievement in children with new-onset seizures and asthma: a prospective study. Epilepsy Behav 2007;10:311–8. [11] Fastenau PS, Johnson CS, Perkins SM, et al. Neuropsychological status at seizure onset in children. Neurology 2009;73:526–34. [12] Dunn DW, Johnson CS, Perkins SM, et al. Academic problems in children with seizures: relationships with neuropsychological functioning and family variables during 3 years after onset. Epilepsy Behav 2010;19:455–61. [13] Berg AT, Smith SN, Froblish D, et al. Special education needs of children with newly diagnosed epilepsy. Dev Med Child Neurol 2005;47:749–53. [14] Schoenfeld J, Seidenberg M, Woodard A, et al. Neuropsychological and behavioral status of children with complex partial seizures. Dev Med Child Neurol 1999;41: 724–31. [15] Jones JE, Siddarth P, Gurbani S, Shields WD, Caplan R. Cognition, academic achievement, language, and psychopathology in pediatric chronic epilepsy: short-term outcomes. Epilepsy Behav 2010;18:211–7. [16] Aldenkamp AP, Arends J. The relative influence of epileptic EEG discharges, short nonconvulsive seizures, and type of epilepsy on cognitive function. Epilepsia 2004;45:54–63. [17] Aldenkamp AP, Weber B, Overweg-Plandsoen CG, Reijs R, van Mil S. Educational underachievement in children with epilepsy: a model to predict the effects of epilepsy on educational achievement. J Child Neurol 2005;20:175–80. [18] Austin JK, Huberty TJ, Huster GA, Dunn DW. Does academic achievement in children with epilepsy change over time? Dev Med Child Neurol 1999;41:473–9. [19] Berg AT, Langfitt JT, Testa FM, et al. Residual cognitive effects of uncomplicated idiopathic and cryptogenic epilepsy. Epilepsy Behav 2008;13:614–9. [20] Hermann BP, Jones JE, Seth R, et al. Growing up with epilepsy: a two-year investigation of cognitive development in children with new onset epilepsy. Epilepsia 2008;49:1847–58.

53

[21] Williams J, Phillips T, Griebel ML, et al. Factors associated with academic achievement in children with controlled epilepsy. Epilepsy Behav 2001;2:217–23. [22] Byars AW, Byars KC, Johnson CS, et al. The relationship between sleep problems and neuropsychological functioning in children with first recognized seizures. Epilepsy Behav 2008;13:607–13. [23] Parisi P, Bruni O, Villa MP, et al. The relationship between sleep and epilepsy: the effect on cognitive functioning in children. Dev Med Child Neurol 2010;52: 805–10. [24] Glauser TA, Cnaan A, Shinnar S, et al. Ethosuximide, valproic acid, and lamotrigine in childhood absence epilepsy. N Engl J Med 2010;362:790–9. [25] Sturniolo MG, Galletti F. Idiopathic epilepsy and school achievement. Arch Dis Child 1994;70:424–8. [26] Austin JK, Huberty TJ, Huster GA, Dunn DW. Academic achievement in children with epilepsy or asthma. Dev Med Child Neurol 1998;40:248–55. [27] Adewuya AO, Oseni SBA, Okeniyi JAO. School performance of Nigerian adolescents with epilepsy. Epilepsia 2006;47:415–20. [28] Fastenau P, Shen J, Dunn D, Perkins, et al. Neuropsychological predictors of academic underachievement in pediatric epilepsy: moderating roles of demographic, seizure, and psychosocial variables. Epilepsia 2004;45:1261–72. [29] Oostrom KJ, van Teeseling H, Smeets-Schouten A, Peters ACB, Jennekens-Schinkel A. Three to four years after diagnosis: cognition and behavior in children with ‘epilepsy only’. A prospective, controlled study. Brain 2005;128:1546–55. [30] Katzenstein JM, Fastenau PS, Austin JK, Dunn DW, Bigatti SM, Johnson KE. Teacher's assessments of the academic performance of children with epilepsy. Epilepsy Behav 2007;10:426–31. [31] Sillanpää M, Jalava M, Kaleva O, et al. Long-term prognosis of seizures with onset in childhood. N Engl J Med 1998;338:1715–22. [32] Nilson L, Ahlom A, Bahman Y, et al. Risk factors for suicide in epilepsy: A case control study. Epilepsia 2002;43:644–51. [33] Ingerski L, Modi A, Hood K, et al. Health related quality of life across paediatric chronic conditions. J Paediatr 2009;156:639–44. [34] Cheung C, Wirrell E. Adolescents’ perception of epilepsy compared with other chronic diseases: “through a teenagers eyes.”. J Child Neurol 2006;21:214–22. [35] Stephen L, Maxwell J, Brodie M. Outcomes from a nurse-led clinic for adolescents with epilepsy. Seizure 2003;12:539–44. [36] Tuffrey C, Pearce A. Transition from paediatric to adult medical services for children with chronic neurological conditions. J Neurol Neurosurg Psychiatry 2003;74:1011–3. [37] McDermott S, Mani S, Krishnawami S. A population-based analysis of specific behavior problems associated with childhood seizures. J Epilepsy 1995;8:110–8. [38] Rutter M, Tizard J, Yule W, Graham P, Whitmore K. Isle of Wight studies, 1964–1974. Psychol Med 1976;6:313–32. [39] Dunn DW. Neuropsychiatric aspects of epilepsy in children. Epilepsy Behav 2003;4:101–6. [40] Rodenburg R, Stams GJ, Meijer AM, Aldenkamp AP, Deković M. Psychopathology in children with epilepsy: a meta-analysis. J Pediatr Psychol 2005;30:453. [41] Grunberg F, Pond DA. Conduct disorders in epileptic children. Br Med J 1957;20:65. [42] Hartlage LC, Green JB. The relation of parental attitudes to academic and social achievement in epileptic children. Epilepsia 1972;13:21–6. [43] Seidenberg M, Berent S. Childhood epilepsy and the role of psychology. Am Psychol 1992;47:1130–3. [44] Sullivan EB, Gahagan L. On intelligence of epileptic children. Genet Psychol Monogr 1935;17:309–76. [45] Belsky J. The determinants of parenting: a process model. Child Dev 1984;55: 83–96. [46] Cicchetti D, Toth SL. Transactional ecological systems in developmental psychopathology. In: Luthar SS, Burack JA, Cicchetti D, Weisz JR, editors. Developmental psychopathology: perspectives on adjustment, risk, and disorder. New York: Cambridge Univ. Press; 1997. p. 317–49. [47] Bronfenbrenner U. The ecology of human development: experiments by nature and design. Cambridge, MA: Harvard Univ. Press; 1979. [48] Bronfenbrenner U. Ecology of the ramily as a context for human development: research perspectives. Dev Psychol 1986;22:723–42. [49] Deković M, Janssens JMAM, As N. Family predictors of antisocial behavior in adolescence. Fam Process 2003;42:223–35. [50] Rodenburg R, Meijer AM, Deković M, Aldenkamp AP. Family factors and psychopathology in children with epilepsy: a literature review. Epilepsy Behav 2005;6:488–503. [51] Abidin RR. The determinants of parenting behavior. J Clin Child Adolesc Psychol 1992;21:407–12. [52] McCubbin HI, Patterson JM. The family stress process. Marriage Fam Rev 1983;6: 7–37. [53] Rodenburg R, Meijer AM, Deković M, Aldenkamp AP. Parents of children with enduring epilepsy: predictors of parenting stress and parenting. Epilepsy Behav 2007;11:197–207. [54] Darling N, Steinberg L. Parenting style as context: an integrative model. Psychol Bull 1993;113:487–96. [55] Green M, Solnit AJ. Reactions to the threatened loss of a child: a vulnerable child syndrome: pediatric management of the dying child, part III. Pediatrics 1964;34:58. [56] Thomasgard M, Metz WP. The vulnerable child syndrome revisited. J Dev Behav Pediatr 1995;16:47. [57] Chapieski L, Brewer V, Evankovich K, Culhane-Shelburne K, Zelman K, Alexander A. Adaptive functioning in children with seizures: impact of maternal anxiety about epilepsy. Epilepsy Behav 2005;7:246–52.

54

R. Rodenburg et al. / Epilepsy & Behavior 22 (2011) 47–54

[58] Modi AC. The impact of a new pediatric epilepsy diagnosis on parents: parenting stress and activity patterns. Epilepsy Behav 2009;14:237–42. [59] Pianta RC, Lothman DJ. Predicting behavior problems in children with epilepsy: child factors, disease factors, family stress, and child mother interaction. Child Dev 1994;65:1415–28. [60] Sbarra DA, Rimm-Kaufman SE, Pianta RC. The behavioral and emotional correlates of epilepsy in adolescence: a 7-year follow-up study. Epilepsy Behav 2002;3:358–67. [61] Hermann BP, Whitman S. Behavioral and personality correlates of epilepsy: a review, methodological critique, and conceptual model. Psychol Bull 1984;95: 451–97. [62] Dunn DW, Buelow JM, Austin JK, Shinnar S, Perkins SM. Development of syndrome severity scores for pediatric epilepsy. Epilepsia 2004;45:661–6. [63] Alwash RH, Hussein MJ, Matloub FF. Symptoms of anxiety and depression among adolescents with seizures in Irbid, northern Jordan. Seizure 2000;9:412–6. [64] Caplan R, Arbelle S, Magharious W, et al. Psychopathology in pediatric complex partial and primary generalized epilepsy. Dev Med Child Neurol 1998;40: 805–11. [65] Caplan R, Siddarth P, Gurbani S, Hanson R, Sankar R, Shields WD. Depression and anxiety disorders in pediatric epilepsy. Epilepsia 2005;46:720–30. [66] Caplan R, Siddarth P, Gurbani S, Ott D, Sankar R, Shields WD. Psychopathology and pediatric complex partial seizures: seizure related, cognitive, and linguistic variables. Epilepsia 2004;45:1273–81. [67] Jones JE, Watson R, Sheth R, et al. Psychiatric comorbidity in children with new onset epilepsy. Dev Med Child Neurol 2007;49:493–7. [68] Dunn DW, Austin JK, Perkins SM. Prevalence of psychopathology in childhood epilepsy: categorical and dimensional measures. Dev Med Child Neurol 2009;51: 364–72. [69] Beyenburg S, Mitchell AJ, Schmidt D, Elger CE, Reuber M. Anxiety in patients with epilepsy: systematic review and suggestions for clinical management. Epilepsy Behav 2005;7:161–71. [70] Mattson RH. Emotional effects on seizure occurrence. In: Smith D, Treiman D, Trimble M, editors. Advances in neurology. New York: Raven Press; 1991. p. 453–60. [71] Lathers CM, Schraeder PL. Stress and sudden death. Epilepsy Behav 2006;9: 236–42. [72] Dautovic E, de Roos C, van Rood YR, et al. Seizure-related anxiety in children with epilepsy: treatment efficacy (unpublished thesis). [73] Rodenburg R, Benjamin A, Meijer AM, Jongeneel R. Eye movement desensitization and reprocessing in an adolescent with epilepsy and mild intellectual disability. Epilepsy Behav 2009;16:175–80. [74] Rodenburg R, Benjamin A, de Roos C, Meijer AM, Stams GJ. Efficacy of EMDR in children: a meta-analysis. Clin Psychol Rev 2009;29:599–606. [75] Ramaratnam S, Baker GA, Goldstein LH. Psychological treatments for epilepsy. Cochrane Database Syst Rev 2008;3CD002029. [76] Kassam-Adams N. Introduction to the special issue: posttraumatic stress related to pediatric illness and injury. J Pediatr Psychol 2006;31:337–42. [77] Kazak AE, Kassam-Adams N, Schneider S, Zelikovsky N, Alderfer MA, Rourke M. An integrative model of pediatric medical traumatic stress. J Pediatr Psychol 2006;31:343–55. [78] Kean EM, Kelsay K, Wamboldt F, Wamboldt MZ. Posttraumatic stress in adolescents with asthma and their parents. J Am Acad Child Adolesc Psychiatry 2006;45:78–86. [79] Drotar D. Case studies and series: a call for action and invitation for submissions [editorial]. J Pediatr Psychol 2009;34:795–802. [80] Rapoff M, Stark L. Journal of Pediatric Psychology statement of purpose: section on single-subject studies [editorial]. J Pediatr Psychol 2008;33:16–21. [81] Chambless DL, Hollon SD. Defining empirically supported therapies. J Consult Clin Psychol 1998;66:7–18. [82] Chorpita BF, Yim LM, Donkervoet JC, et al. Toward large-scale implementation of empirically supported treatments for children: a review and observations by the Hawaii Empirical Basis to Services Task Force. Clin Psychol Sci Pract 2002;9: 165–90. [83] Garralda EM. Accountability of specialist child and adolescent mental health services. Br J Psychiatry 2009;194:389–91. [84] Johnston C, Gowers S. Routine outcome measurement: a survey of UK child and adolescent mental health services. Child Adolesc Ment Health 2005;10: 133–9.

[85] Varni JW, Burwinkle TM, Lane MM. Health-related quality of life measurement in pediatric clinical practice: an appraisal and precept for future research and application. Health Qual Life Outcomes 2005;3:1–9. [86] Kazak AE, Rourke MT, Navsaria N. Families and other systems in pediatric psychology. In: Roberts MC, Steele RG, editors. Handbook of Pediatric Psychology. 4th Ed. New York: Guilford Press; 2009. p. 656–71. [87] Hermann B, Seidenberg M, Jones J. The neurobehavioural comorbidities of epilepsy: can a natural history be developed? Lancet Neurol 2008;7:151–60. [88] Baum KT, Byars AW, deGrauw TJ, et al. The effect of temperament and neuropsychological functioning on behavior problems in children with newonset seizures. Epilepsy Behav 2010;17:467–73. [89] Berg AT, Vickrey BG, Testa FM, Levy SR, Shinnar S, DiMario F. Behavior and social competency in idiopathic and cryptogenic childhood epilepsy. Dev Med Child Neurol 2007;49:487–92. [90] Austin JK, Dunn DW, Johnson CS, Perkins SM. Behavioral issues involving children and adolescents with epilepsy and the impact of their families: recent research data. Epilepsy Behav 2004;5:33–41. [91] Schilling LS, Grey M, Knafl KA. The concept of self-management of type 1 diabetes in children and adolescents: an evolutionary concept analysis. J Adv Nurs 2002;37: 87–99. [92] Wagner JL, Smith G, Ferguson PL. Psychosocial correlates in pediatric epilepsy: preliminary findings from an epidemiological study in South Carolina. Presented at the Child Health Conference; April 2007. Cincinnati OH. [93] Wagner JL, Sample PL, Ferguson PL, Pickelsimer EE, Smith GM, Selassie AW. Impact of pediatric epilepsy: voices from a focus group and implications for public policy change. Epilepsy Behav 2009;16:161–5. [94] Wagner JL, Smith G. Psychological services in a pediatric epilepsy clinic: feasibility and referral patterns. Epilepsy Behav 2007;10:129–33. [95] Wagner JL, Smith G. Psychosocial intervention in pediatric epilepsy: a critique of the literature. Epilepsy Behav 2006;8:39–49. [96] Wagner JL, Smith G, Ferguson P, Horton S, Wilson E. A learned hopelessness model of depressive symptoms in youth with epilepsy. J Pediatr Psychol 2009;34:89–96. [97] Wagner JL, Smith G, Ferguson PL, Wannamaker B. Caregiver perceptions of seizure severity in pediatric epilepsy. Epilepsia 2009;50:2102–9. [98] Smith G, Ferguson P, Wagner J, Hall V, Wannamaker B. Testing the validity of a depression screening instrument for youth with epilepsy. Presented at the American Epilepsy Society Meeting; December 2010. San Antonio, TX. [99] Barry JJ, Ettinger AB, Friel P, et al. Consensus statement: the evaluation and treatment of people with epilepsy and affective disorders. Epilepsy Behav 2008;13(Suppl 1):S1–S29. [100] DiIorio C, Shafer PO, Letz R, Henry TR, Schomer DL, Yeager K. Behavioral, social, and affective factors associated with self-efficacy for self-management among people with epilepsy. Epilepsy Behav 2006;9:158–63. [101] Robinson E, DiIorio C, DePadilla L, et al. Psychosocial predictors of lifestyle management in adults with epilepsy. Epilepsy Behav 2008;13:523–8. [102] Weisz JR, Southam-Gerow MA, Gordis EB, Connor-Smith J. Primary and secondary control enhancement training for youth depression: applying the deploymentfocused model of treatment. In: Kazdin AE, Weisz JR, editors. Evidence-based psychotherapies for children and adolescents. New York: Guilford Press; 2003. p. 165–83. [103] Wysocki T, Harris MA, Greco P, et al. Randomized, controlled trial of behavior therapy for families of adolescents with insulin-dependent diabetes mellitus. J Pediatr Psychol 2000;25:23–33. [104] DiIorio C, Escoffery C, Yeager KA, et al. WebEase: development of a Web-based epilepsy self-management intervention. Preventing Chronic Dis 2009;6:1–7 PMCID: 2644585. [105] Wagner JL, Smith G, Ferguson PL, Wannamaker B. Coping openly and personally with epilepsy: a CBT program for youth with epilepsy and their caregivers. Presented at the American Epilepsy Society Annual Meeting; December 2009. Boston, MA. [106] Wagner JL, Smith G, Ferguson PL, van Bakergem K, Hrisko S. Pilot study of an integrated cognitive-behavioral and self-management intervention for youth with epilepsy and caregivers: Coping Openly and Personally with Epilepsy (COPE). Epilepsy Behav 2010;18:280–5. [107] Karlson CW, Rapoff MA. Attrition in randomized controlled trials for pediatric chronic conditions. J Pediatr Psychol 2009;34:782–93. [108] Austin J, Carr D, Hermann B. Living Well II: a review of progress since 2003. Epilepsy Behav 2006;9:386–93.