Psychosocial treatment programs in epilepsy: A review

Epilepsy & Behavior 16 (2009) 371–380

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Review

Psychosocial treatment programs in epilepsy: A review Robert James Mittan Private Practice, 7316 Oxford Bluff Drive, Stanley, NC 28164-6318, USA

a r t i c l e

i n f o

Article history: Received 21 May 2009 Revised 17 August 2009 Accepted 28 August 2009 Available online 26 September 2009 Keywords: Psychosocial treatment Psychoeducational Quality of life Compliance Outcomes Cost Refractory epilepsy Coping Adjustment Patient education

a b s t r a c t Psychosocial problems are a major unmet treatment need in epilepsy. The purpose of this review was to describe published psychosocial treatment interventions in the English medical literature. Seventeen outcome studies for 15 treatments were found. Survival past proof of concept was a problem, with only four programs making it into current use. With important exceptions, treatments met with limited success. Several were successful with medical education, improving quality of life, social adjustment, and adjustment to seizures. A psychological intervention for seizure control for patients with refractory epilepsy was outstanding. Research problems included few control groups, a dependence on paper-and-pencil measures, limited variables, and only one study with serum determination for compliance. Low costeffectiveness was a problem. Only one approach showed economic promise. Low participation by patients was an unexpected barrier. Psychosocial treatments should be targeted for development and needs to be integrated into the treatment flow of specialty clinics. Ó 2009 Elsevier Inc. All rights reserved.

1. Introduction ‘‘Possibly the least understood and most neglected aspects of epilepsy are the social, psychological, and behavioral problems that are so common. . . [These] can be more handicapping than the actual seizures, yet these problems often are unrecognized and little skilled assistance is available”. The Commission for the Control of Epilepsy and Its Consequences made this critical observation in 1975 [1]. Regrettably, this situation remains true today. There is a broad and extensive literature documenting the psychiatric, behavioral, and psychosocial problems of epilepsy. However, research evaluating formal psychosocial interventions to ameliorate these problems is rare. The problems are well known. The solutions are not. This article reviews the current state of published psychosocial and/or educational treatment programs for persons affected by epilepsy. It is hoped this discussion will provide a launching point for further innovation and inspire greater effort and funding for such important endeavors. 2. Procedure Psychosocial treatment programs published since 1990 were investigated through the Epilepsy Foundation’s National Library E-mail address: [email protected] 1525-5050/$ - see front matter Ó 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.yebeh.2009.08.031

database, a survey of epilepsy journals, and through additional citations found in discovered articles. Emphasis was placed on finding published patient education and psychosocial treatment programs that had formal outcome studies or that had been implemented beyond an initial pilot demonstration. Programs from epilepsy associations were not included because no English-based epilepsy association programs were found to be published in peer review journals or to be subject to peer review outcome studies. A total of 14 psychosocial or educational treatment interventions were found, with two of these interventions having two outcome studies. A total of 17 studies were reviewed [2–18] (see Table 1). A list was made of the most common categories of psychosocial and educational variables measured in these studies. Six ‘‘Medical Management” or medical self-management variables were identified. Fifteen ‘‘Psychosocial” variables were identified. From these outcome variables a matrix was constructed plotting these 21 variables against 17 individual studies in Table 1. No study measured all of the variables. Many studies assessed a very limited set of variables. If a variable was not measured, its location in the matrix was left blank. If a measurement was entirely subjective, an ‘‘s” was entered across from that variable for that study. Findings considered significant (marked with a ‘‘+” in Table 1) were those listed at P 6 0.05 in the original study. If a trend was found toward a positive finding (P 6 0.1), a ‘‘t” was entered for that variable. If a variable was measured without a good result, a ‘‘ ” was entered into

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Intervention Program namea Theoretical modelb Intervention methodc Intervention group sized Target audiencee In general usef Outcome study Research author

Research designg Research N Research groupsh Follow-up (months)i Research done byj Medical management Knowledge of epilepsy Acceptance of epilepsy Medical selfmanagement Compliance: serum levels Seizure frequency Health care utilization costs Psychosocial Fear harm from seizures

MOSE Educ PE

FAMO Educ PE

Pengu Educ PE

SEE1m IM PE

SEE2 IM PE

FLIP Bipsyso Educ

PEPE Educ PE

ACT1 ACT Ind/G

ACT2 ACT Ind/G

ISPE SE PE

BSzS PE Ind/G

CEP1 Couns GRx

CEP2 Couns GRx

OGA Couns PE

GIL Psy PE

ACIN Piag Educ

TCE CBT PE

10

7–12

6

200

200

8

10

1/8

1/8

12

1/8

7–10

7–10

10

1

8–10

5

Adult Yes

C&P Yes

Child Yes

Adult Yes

C&P Yes

C&P No

Adult No

Adult No

Adult No

Adult No

C&P No

Child No

Parent No

Adult No

Adult No

C&P No

Adol No

May and Wohlrab Amdam, Helgeson Shore Jantzen Huber Pfafflin, et al., 2001 [4] et al., et al., et al., and 2002 [2] 2007 [3] 1990 [5] 2008 [6] 2009 [7] Seidel, 2001 [8] RC n/a n/a RC SC WLC UNC 242 38 29 15 2 2 1 2 2 6 4 6 6 0

SC 18 2 12

RC 27 2 12

RC 38 2 1

SC 20 2 0.5

RC 236 2 5

RC 365 2 5

UNC 10 1 IPost

SC 59 3 6

RC 355 2 12

SC 7 1 1

Auth

Auth

Auth

Auth

Auth

Auth

Auth

Auth

Auth

Auth

Auth

+

+

+

s

IND

IND

Auth

Auth

10/10k +

+

+

+

so

1/2

+ +

+

3/4

+

1/1 5/6 2/2

4/6

Auth

Auth

+

Lundgren Lundgren Pramuka Austin Lewis Lewis Ogata and Gillham, TieffenSnead, et al., et al., et al., et al., et al., et al., Kouichiro, 1990 [16] berg et al., 2004 [18] 2008 [9] 2006 [10] 2007 [11] 2002 [12] 1990 [13] 1991 [14] 2000 [15] 2000 [17]

+

+ +

+

+

+

t

+

+

+

s

+ +

+

R.J. Mittan / Epilepsy & Behavior 16 (2009) 371–380

Table 1 Psychosocial treatment programs and research outcomes.

Anxiety Depression Stigma Adjustment to seizures Mental health/ coping Memory/ cognition Behavior Self-esteem Self-efficacy Locus of control Quality of life Family adjustment Social adjustment Vocation/ school adjustment

a b

c d e f g h i j k l m n o

+

+

+

+

+

3/4

+

+

s

+

+ +

+

+

0/1 3/3 1/3 1/3 1/2 5/8 3/5 5/7 3/6

+ + +

+

t

+ +

+ +

tn t

140

383

100

8

+

+ +

+

8

+

200

s

320

+ +

675

663

240

310

180

180

s s

+

160

700

+

250

360

Abbreviation for the name of the therapeutic intervention. ‘‘1” and ‘‘2” indicate separate articles for the same program. The theoretical model underpinning the intervention: Educ, educational; IM, information medicine; Bipsyso, biopsychosocial; ACT, Acceptance and Commitment Therapy; Couns, counseling; Psy, psychotherapy; Piag, Piaget; CBT, cognitive behavior therapy. How the treatment was delivered. PE, psychoeducational therapy; Ind/G, combination of individual and group treatments; GRx, group therapy; Educ, educational therapy. Size of the treatment group used in the intervention. Adult, adults; C & P, children and parents; Child, children; Parent, parents; Adol, adolescents. Yes, the intervention is currently and regularly being used in epilepsy treatment; No, not regularly used. RC, randomized control; n/a, not researched; WLC, wait list control; SC, self-control; UNC, uncontrolled. Number of research groups included in the study, e.g., control + experimental = 2. Length of follow-up in the outcome study. Whether the outcomes research was done by the authors of the intervention (Auth) or by independent researchers (IND). Number of studies with a positive finding for that variable/total number of studies measuring that variable. Number of professional hours required to treat 100 patients using the intervention. Columns in boldface type indicate interventions where two separate outcome studies were performed (SEE and ACT), CEP1 and CEP2 were the same study reported in two different articles. t, a positive trend where P 6 0.1; +, means P 6 0.05; , means the variable was measured and did not show significance or a positive trend. s, a purely subjective rating showing a positive trend.

R.J. Mittan / Epilepsy & Behavior 16 (2009) 371–380

Professional hours/100 patientsl

2/3 3/6 0/1 5/7

373

374

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the corresponding cell. The two sets of columns in boldface type represent the only two psychosocial treatment programs to have more than one outcome study performed. Because the variables are based on a heterogeneous collection of measures, the categories are not defined past the common descriptions listed. There may appear to be some degree of overlap among them, which is a sign of the imprecision of measurement concepts used by the various studies. For more precise definitions, the reader is encouraged to read the summaries of each program in Section 7 at the end of this review and refer to the original studies for greater detail. The purposes of this article are to provide an overview of psychosocial treatment interventions, review their apparent effectiveness based on outcomes research, and make recommendations on future psychosocial treatment development. 3. What outcomes appear promising The letters in square brackets refer to the name given to each specific study in the Program Name row of Table 1. 3.1. Medical self-management Most of the interventions showed a clear strength in improving subjects’ knowledge about epilepsy. Of the 10 psychosocial and educational programs that measured knowledge about epilepsy, all 10 showed a significant improvement at follow-up. Programs demonstrating significantly improved knowledge on objective measurement scales include MOSES [MOSE, Table 1], both Seizure & Epilepsy Education (SEE) programs [SEE1, SEE2], FLIP&FLAP [FLIP], Be Seizure Smart [BSzS], Children’s Epilepsy Program [CEP1, CEP2], and ACINDES [ACIN]. Both PEPE [PEPE] and Ogata’s program [OGA] found significant improvement in knowledge based on subjects’ subjective ratings. These consistent results suggest improvement in subjects’ knowledge about epilepsy appears strongly amenable to intervention. Highlighting a much underappreciated outcome of psychosocial treatment programs, five of six studies showed a significant reduction in seizure frequency as an outcome. These included MOSES [MOSE], Acceptance and Commitment Therapy (ACT) [ACT1, ACT2], Gillham’s program [GIL], and ACINDES [ACIN]. These nondrug methods of seizure control clearly deserve greater attention in the care of persons with refractory epilepsy. Two programs appeared to have a broader range of impact on medical management than the other programs. These programs included ACINDES [ACIN] and the Seizures & Epilepsy Education (SEE, formerly named Sepulveda Epilepsy Education) program. SEE participants showed significantly improved knowledge and medical self-management [SEE1, SEE2], and improved acceptance of epilepsy and improved medication compliance as determined by serum levels [SEE1]. These programs were also the only two to show potential cost savings, with ACIN showing a significant reduction in health care utilization and SEE showing a trend in that direction. 3.2. Psychosocial outcomes Positive psychosocial outcomes appear to be somewhat limited among the various interventions with the exceptions of ACINDES, Gillham’s program [GIL], and the SEE program [SEE1, SEE2]. This may be due to the limited numbers of variables assessed in many studies, the insensitivity of the outcome measures to actual patient changes, limited therapeutic goals for the intervention, and small subject groups that underpowered statistical analyses. Although the measurements were sparse, successes suggest that many psychosocial problems in epilepsy are amenable to treatment.

Quality of life has been a major concept in epilepsy care in recent years. This concept spanned the time frame of the reviewed studies. Eight studies measured quality of life and five found significant improvements. These included the SEE program [SEE1, SEE2], the ACT programs [ACT1, ACT2], and Gillham’s program [GIL]. It would appear that well-constructed psychosocial treatments have the potential to make patients’ and families’ lives better. Three of three programs for children or adolescents found improvements in ‘‘behavior”—SEE program [SEE2], Children’s Epilepsy Program [CEP1], and Take Charge of Epilepsy [TCE]—suggesting the behavioral problems of children with epilepsy might be effectively treated through psychosocial or educational programs. Five of seven studies found significant improvement in ‘‘adjustment to seizures”: SEE [SEE1, SEE2], Be Seizure Smart [BSzS], and Children’s Epilepsy Program (adult) [CEP2], with PEPE showing significant subjective improvement. Five of seven found improvement in ‘‘fear of harm from seizures”: SEE [SEE1, SEE2], Gillham’s [GIL], and Ogata’s [OGA] programs, with FLIP&FLAP [FLIP] showing a positive trend. This suggests that the common anxieties that stem from seizures and epilepsy may also respond to psychosocial treatments. Depression has been recognized as the most common psychiatric comorbidity of epilepsy [19]. Half of the six studies that addressed this problem demonstrated significant improvement in self-report tests, including SEE1, SEE2, and GIL. Only two of these, SEE1 and SEE2, specifically looked at outcomes for patients who scored in the clinically depressed range. All of the ‘‘depressed” adult patients and all but one of the ‘‘depressed” child patients’ scores moved into the ‘‘normal” range after exposure to the program. All patients moving to the ‘‘normal” range after the intervention stayed in the ‘‘normal” range at long-term follow-up. These results are a promising development considering the significant problem of depression in epilepsy. Psychosocial treatment programs may also influence social functioning. Six of eight studies found trends toward improved social adjustment: FLIP, CEP1, and GIL on objective measures; PEPE and OGA on subjective measures; and SEE1 showing a positive trend. Three of five, including SEE2, BSzS, and ACIN, showed significant improvement in ‘‘family adjustment”. The self-efficacy measures ‘‘self-efficacy” and ‘‘locus of control” were measured five times. Only one study each showed improvement in one of these variables; the SEE program [SEE2] showed significant improvement in ‘‘self-efficacy” and ACINDES [ACIN] in ‘‘locus of control”. These much-discussed constructs [20] may be less easy to influence therapeutically and/or more effective therapeutic regimens remain to be discovered. Remarkably, ‘‘social stigma”, the iconic psychosocial problem of epilepsy, was measured only once, in Take Charge of Epilepsy [TCE], without a positive finding. Of the programs reviewed, three appeared to be more effective overall: ACINDES [ACIN], Gillham’s program [GIL], and the SEE program [SEE1, SEE2]. In part this limited success may be due to the relatively few outcome measures used in many of the other studies, though not in all. For example, ISPE had a total of 59 scales included in its design. Gillham’s program [GIL] showed significant improvement in psychological problems, as one might expect with one-on-one therapy. ‘‘Anxiety” and ‘‘depression” were both significantly improved along with ‘‘family adjustment”. This undoubtedly contributed to a significant improvement in ‘‘quality of life” for the study participants. These findings were obtained in a moderately powered study of 59 subjects. ACINDES [ACIN] was one of the few relatively strongly powered studies, with 355 subjects. ACINDES showed significant improvements in both medical management and psychosocial adjustment. In terms of management, ACINDES had the useful result of

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significantly reduced ‘‘seizure frequency” and significantly improved ‘‘knowledge”. It was the only intervention to show significant reduction in ‘‘health care utilization costs”. Psychosocially the program showed significant reduction in ‘‘fear of harm from seizures” and significant improvements toward an internal ‘‘locus of control” in ‘‘family adjustment” and ‘‘school adjustment”. The SEE program studies (SEE1, SEE2) were statistically underpowered, requiring large clinical effects to reach statistical significance. With respect to medical management, the SEE program resulted in significant ‘‘knowledge of epilepsy”, ‘‘acceptance of epilepsy”, and improved ‘‘medical self-management”. SEE was the only program to demonstrate significant improvement in compliance using blood serum determinations. With respect to psychosocial variables, the two studies found significant decreases in ‘‘fear of harm from seizures” and ‘‘depression”. There were significant improvements in ‘‘quality of life”, ‘‘adjustment to seizures”, ‘‘mental health/coping”, ‘‘behavior”, ‘‘self-esteem”, ‘‘self-efficacy”, and ‘‘family adjustment”, with trends toward improvement in ‘‘social adjustment” and ‘‘vocational adjustment”. SEE has the distinction of being the only large group format among all of the interventions reviewed. The apparent effectiveness of the SEE program with its treatment efficiency (8 professional hours to treat 100 patients) and positive outcomes with adults [SEE1], parents, and children (SEE2) offers a potentially practical means of meeting psychosocial treatment needs economically. Thus, this unique large audience treatment format warrants further research, and the SEE program should be tried in larger studies to see if the initial results may be generalizable. 4. Intervention paradigm discussion The theoretical frameworks of most interventions (if they are articulated) are still in their infancy. Only a few differences in treatment delivery methods exist among the studies reviewed. These conditions expose the nascent status of psychosocial treatment in epilepsy. Rather than a criticism, these situations should be appreciated as a significant beginning in an effort of great importance: the quest for psychosocial treatments to ensure the welfare and quality of life of people with epilepsy and their families. 4.1. Behavior therapy model Behavioral therapy models are among the oldest psychosocial treatment techniques used in epilepsy and they are clearly the best defined. The use of behavioral techniques to control seizures has a long history with many empirical studies [21,22]. In recent years publications using this approach have become rare, despite repeated findings of effectiveness [9,10,21,22]. Examples of this approach can be found in studies that look at relaxation, seizure interruption, and yoga as means of increasing seizure threshold or improving seizure control [9,10]. The behavior therapy models include behavior modification (which has long been used for epilepsy), cognitive behavior therapy, and the newer ACT. ACT appears in two studies in this review [ACT1, ACT2]. These ACT studies demonstrate striking effectiveness, with significant reductions in seizures for adults with refractory epilepsy. 4.2. Self-efficacy model The self-efficacy (SE) model is a theoretical foundation to healthrelated psychosocial treatment that is gaining popularity in a variety of chronic health programs. It has recently been adapted for epilepsy by DiIorio and colleagues [20]. SE remains mostly theoretical with respect to epilepsy psychosocial interventions;

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however, one study in this review was based entirely on the SE model: ISPE [ISPE]. ISPE had only one significant finding (improved mental health/coping) out of 11 measures. The idea is that self-efficacy, the belief in one’s ability to control or influence health-related outcomes, is the basis of compliance and psychosocial coping. Self-efficacy is usually measured by self-report questionnaires, including those measuring SE directly and those measuring locus of control (LOC), the degree to which persons perceive health outcomes are due to one’s own efforts versus due to outside forces over which one has little control. Of the four studies showing significantly improved compliance either by self-report or serum measurements (MOSE, SEE1 [serum determinations], SEE2, and FLIP), only one showed an improvement in either SE or LOC (SEE2). The extent to which the theoretical construct of ‘‘self-efficacy” influences actual compliance remains unclear in the available studies. 4.3. Information medicine/psychoeducational model Created in 1983, the SEE program [SEE1, SEE2] pioneered information medicine and the psychoeducational treatment method in epilepsy. The SEE program was called ‘‘psychoeducational” because it was designed to deliver psychotherapy through educational methods. Some version of the psychoeducational approach is now included in many health interventions, including 10 of the 16 interventions reviewed (see Table 1). The SEE program assumes that medical self-management and psychosocial coping are learned behavioral skills, just like playing the piano is a learned behavioral skill. For example, coping and compliance are not only supported by learning how to properly take medications (learned behavioral skill), but are maintained in daily practice by patients clearly understanding why medications are dosed and must be taken in a certain way (motivation induced by information medicine.) Through this psychoeducational/information medicine approach, the necessary motivation and practical behavioral skills for coping are taught. As comprehensive understanding is presumed sufficient for motivation of compliance, the SEE program proposes knowledge is the basis of compliance rather than a more ‘‘affective” approach (e.g., the belief system proposed in the selfefficacy model). Overall, programs using the psychoeducational model had significant positive findings in 40 of 66 variables measured, for a rate of 61% of therapeutic goals achieved. The SEE program’s combined rate was 23 of 25 variables (92%) showing improvement, that for PEPE was 3/3, that for Ogata’s program [OGA] was 4/4, that for Gillham’s program [GIL] was 5/5, and that for MOSES [MOSE] was 3/8 (38%). Two programs did poorly and accounted for most failures in the psychoeducational approach. These included ISPE [ISPE], with 1 of 11 variables significant, and Take Charge of Epilepsy [TCE], with 1 of 10 variables significant. It appears the psychoeducational approach may be effective if appropriately constructed. 4.4. Small group education method Small group education is more a treatment delivery method than a theoretical approach to treatment. This method is used as the exclusive means of treatment delivery in 11 of the 14 interventions reviewed here (see Table 1). Two additional interventions used a combination of small groups with individual treatment: ACT [ACT1, ACT2] and Be Seizure Smart [BSzS]. Small group interventions are broken into separate modules containing a specific set of topics that are delivered in a group interaction format. The theoretical underpinnings lie in the concept that group interaction is believed to allow for socialization and peer support, which reinforces learning and helps to therapeutically lessen the stigma and social isolation that accompany epilepsy. MOSES [MOSE] and

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FAMOSES [FAMO] are the most widely known examples of this model, though ACINDES [ACIN] appeared to achieve greater overall efficacy. Most interventions for children and adolescents use a modified small group education model. Parallel groups are conducted for children and parents separately. In most interventions the educational units appear similar in content for adults and children, though with age-appropriate modifications. Parent groups often include further information regarding childrearing and family dynamics issues not covered in the children’s groups. These child/parent groups, including Be Seizure Smart [BSzS], CEP [CEP1, CEP2], and ACINDES [ACIN], appeared successful. 4.5. Other treatment delivery methods In the series of studies reviewed, only two interventions do not use either a group or group/individual therapeutic method. Gillham’s program [GIL] used a one-on-one psychotherapeutic approach for her intervention. The SEE program [SEE1, SEE2] uses a large audience format (up to 850 people), which includes adults, parents, and children in the same audience. 5. Critique of programs 5.1. Issues in program development The greatest problem among psychosocial treatment and educational programs was that only a handful of these interventions were ever put into general use. The usual pattern is an intervention was given a feasibility study and then was abandoned at that point. Development ends as a ‘‘demonstration of concept”. Only 4 of the 14 different interventions reviewed appear to be in ongoing use. Part of this may be explained by the fact that most demonstrations either were not successful or were only moderately successful. A very significant practical matter interfering with implementation is economics. The professional hours required to treat 100 patients are listed for each educational and psychosocial intervention (Table 1). The professional time required for 100 patients was used instead of time per single patient because this quantity provides a better appreciation of the economic cost of implementing the program in clinical practice. Unfortunately psychosocial care is viewed as expensive and at best adjunctive to medical care. Reimbursement for psychosocial treatment is a problem in the United States. Budget constraints in nationalized health services also relegate these interventions to a low priority. One of the challenges of implementation is to create an intervention that can deal with economic realities. ACT [ACT1, ACT2] appears outstandingly effective in seizure control among refractory patients; however, it costs nearly 700 professional hours to treat 100 patients. This makes the program prohibitively expensive despite its obvious advantages for the patient. (An argument might rightly be made that the cost savings in reduction of seizures more than offsets the initial cost of treatment.) MOSES [MOSE] and FAMOSES [FAMO] require 140 and 383 hours per 100 treatments, respectively. This would also be prohibitive in the United States and possibly for other nationalized health care systems. The other interventions average between 200 hours and 300 hours per 100 treatments. The only economically practical exception to this problem is the SEE program [SEE1, SEE2], which uses a large audience format for treatment delivery. In an audience of 200 people, the treatment cost is only 8 hours of professional time per 100 treatments. There is further encouragement in that this novel treatment approach also appears effective with respect to both medical self-management and psychosocial treatment outcomes (Table 1).

Despite the widely acknowledged importance of psychosocial issues in the quality of life and medical outcomes of persons with epilepsy, there is only one example of formal psychosocial treatment programs specifically integrated with epilepsy specialty centers. This is the MOSES [MOSE] adult program, which is required in certain specialty clinics in Germany. The United States has no example of a published program that is integrated into ongoing care delivery in an epilepsy specialty practice. Over the past 26 years the SEE program (SEE1, SEE2) has been presented to approximately 30,000 patients and families across North America, Australia, and New Zealand, but is primarily sponsored by epilepsy associations, with only a few comprehensive epilepsy centers using it regularly. Although implementation is a major problem, the theoretical basis to psychosocial care is still in early stages of development. The field has not matured enough for well-defined theoretical models for epilepsy to emerge. Two theoretical models are tested in a few of the studies: the ACT [ACT1, ACT2] and SE [ISPE, SEE1, SEE2] models. The ACT model appeared to achieve its stated goals of seizure control and improved quality of life; however, that intervention was the most labor intensive of the interventions reviewed. A more nebulous model, information medicine, is represented by the SEE program [SEE1, SEE2]. Of the studies reviewed here, the SEE program appears to have the broadest range of effectiveness for both medical self-management and quality-of-life variables. However, beyond the ‘‘knowledge is power” foundation, there is little theory to explain the range of benefit that would make the SEE method applicable to other program development. Considering the encouraging outcomes of the SEE program, it is hoped a more detailed explanation will be forthcoming. There is a very troubling problem that was not formally included in any research design, but was a nearly universal theme emerging in many of the results: Patient recruitment and attendance were major problems. It appears difficult to get people to participate in psychosocial treatment. Despite the clearly identified need (by epilepsy professionals and by epilepsy associations), actual motivation for treatment was significantly lacking among available patient populations. For example, a program designed and piloted in Los Angeles, (CEP [CEP1, CEP2]), had to go to Santiago, Chile, to obtain sufficient subject numbers. This introduced the potential problem of cultural bias. Similarly, programs authored in Sweden (ACT [ACT1, ACT2]) were tested in South Africa and India. Although the problem of recruitment was frequently observed, the causes of this problem met with limited explanation. Weekly group meetings, the most common intervention format used in the reviewed studies, suffered from poor attendance because of the competing personal schedules of potential participants. One study, BSzS, got around this problem by conducting treatment sessions over the phone. However, their single ‘‘group meeting” by phone suffered scheduling conflicts. The SEE program avoids weekly schedule conflicts by taking place during a 2-day weekend. Attendance typically runs between 100 and 300 people. Although this appears a much more successful attendance strategy, there are still complaints that an entire weekend can be difficult for families. The problems with recruitment and attendance bring up a fundamental question: What is the point of creating programs if no one will attend? No effort to experimentally address this critical issue was made in any of the studies. Recruitment and attendance are emergent needs of study. 5.2. Lack of epilepsy association research With the scarcity of formal psychosocial treatment programs in epilepsy specialty practices, much of the burden of psychosocial intervention and patient education has fallen to epilepsy associa-

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tions. This condition seems worldwide with the exception of Germany, where the MOSE program is mandatory in several clinics. Although consumer-based epilepsy associations are sustaining the lion’s share of the burden for patient and family education and psychosocial treatment programs in the English-speaking world, virtually no empirically based outcome studies of epilepsy association programs have been published in peer-reviewed journals. The only exception found was a public education program (not a psychosocial treatment program) from Canada, Thinking about Epilepsy, which taught fifth graders about epilepsy and the acceptance of people with epilepsy. The study found significant improvements in knowledge about epilepsy and attitudes toward persons with epilepsy following the 30-minute classroom intervention [23]. 5.3. Research methodology problems The largest problem in research is the lack of outcome studies in the face of so many interventions. These interventions include books, pamphlets, and a host of epilepsy association programs. Internet resources, which are burgeoning, have increasingly authoritative sources yet are untested with respect to their impact on patient knowledge or quality of life. It is unclear to what extent epilepsy specialty centers carry on education or psychosocial treatments, as evaluations of these simply do not appear in the literature. There are only a small number of organized interventions that have proceeded to formal outcome evaluations and to this review. Virtually all were designed in connection with epilepsy centers, nursing, or psychology services with academic ties. In the real world of everyday medical practice, no studies could be found. Some programs reviewed here were implemented, but are only described and not assessed for outcome. These include two interventions that appear widely used: FAMOSES [FAMO] and The Penguin [Pengu]. A second substantial problem is that research is performed with too few subjects. Only four studies were done with sufficient numbers, including MOSES [MOSE] with 242 adult subjects, FLIP&FLAP [FLIP] with 160 children and 152 adult subjects, CEP [CEP1, CEP2] with 236 children and 365 parent subjects (published separately), and ACINDES [ACIN] with 355 combined children and parent subjects. The remaining studies had fewer than 40 subjects, including both experimental and control groups, with the single exception of Gillham’s program [GIL], which included 59 subjects. A third problem is the frequent lack of randomized control trials. Only 7 of 17 studies met this criterion. The rest used a self-control design or no controls whatsoever. There is already precedence for ‘‘significant” findings in uncontrolled studies to disappear when the intervention is subject to a controlled trial [24]. A fourth problem is the assessment measures employed. With few exceptions, the main measures used were paper-and-pencil inventories. There are reasonable concerns whether these inventories measure the constructs they are intended to measure in a meaningful way. For example, exactly how does one ‘‘measure”, ‘‘quality of life”, ‘‘stigma”, or ‘‘depression” in a series of yes–no questions or Likert rating scales? When a ‘‘significant” change is found statistically on such measurements, does it actually reflect a clinically significant change? These concerns are being addressed in current instrument development, but more in-depth correlations between scale scores and direct measurement of real-life adaptation are needed [25]. Objective measures are available for a few constructs. Most important are serum level determinations as a measure of compliance and seizure counts as a measure of seizure control. Compliance is almost always cited as a goal in these interventions, yet only one study (SEE1) measured blood levels. In that study the

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experimental group had a notable mean blood serum increase of 70% at long-term follow-up (no one was in the toxic range), whereas the control group had an 18% decline over the same period. Seizure counts fared better, being used in 6 of 15 studies. Three studies showed remarkable reductions in seizures 1 year following treatment of refractory patients: in ACT1 seizures decreased by 78%, in ACT2 the decrease was 84%, and in ACIN it was 83%. In MOSE an index was used that showed a significant decrease at 6 months; however, the change did not appear as dramatic as in the first three studies. The SEE1 reductions at 4 months were not significant. An additional problem with measures was they were not standardized across studies. For example, ‘‘quality of life”, ‘‘depression”, ‘‘knowledge about epilepsy”, etc., were measured by different scales in different studies. This makes comparison of the effectiveness of different approaches difficult at best. Further complicating this matter is the limited range of assessments made. Referring to Table 1, there are many blank spaces in the matrix indicating the variable was not assessed for that particular study. ACT1 and ACT2 measured only two variables: seizure frequency and a unitary ‘‘quality of life” measure. It may well have had other positive effects, but the limited assessment makes further comparison impossible. A potentially significant problem was that the authors of the interventions conducted outcomes research. This has the potential to introduce bias in terms of study populations, outcome measures, and especially in interpretation of results. To be fair, this condition is almost universal in medical research, and blinded studies only partly compensate for this potential confounder. Psychosocial interventions should be sponsored and researched by independent epilepsy specialty centers to determine effectiveness for their patient populations. The only exceptions to this problem were SEE1 and SEE2. Both SEE program studies were conducted, analyzed, and reported by independent third parties. Finally, in 12 of 14 programs, the evaluation of effectiveness is based on a single study on a single sample population. Given the small number of subjects involved, this severely limits a reader’s ability to extend the findings to other populations. Only two interventions have more than one outcome study on more than one population. ACT1 and ACT2 were performed on separate adult populations in South Africa and India. The findings were the same for both—significant reductions in seizure frequency and improvement in ‘‘quality of life”—suggesting ACT may be effective for adults cross-culturally. The second is the SEE program (SEE1, SEE2), where the first study population was adults with epilepsy and the second population was adolescents (aged 12–18) and their parents. This suggests the SEE program may be appropriate for children aged 12 or older, adolescents, adults, and parents in a US population.

6. Recommendations for the future The development of psychosocial treatment programs in epilepsy has not kept pace with the development of medical treatments. With notable exceptions, the results of these outcome studies are modest. This is a very troubling state of affairs given the extent of treatment need identified within the community. Formal psychosocial treatment interventions need the same program and funding incentives that were provided with the National Institutes of Health initiatives for epilepsy treatment in the late 1970s. This had been identified as a top priority by the Commission for the Control of Epilepsy and Its Consequences [26], but failed to materialize in the Centers for Excellence, despite the mandate.

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The most promising intervention approaches appear to be the psychoeducational model for broad information and psychosocial treatment and the ACT model for behavioral control of seizure frequency. The large group intervention model of the SEE program holds some promise for economic efficiency. Outcome research with randomized controlled trials, much larger subject samples, and more consistent outcome measures need to be made. Whenever possible, clinically defined outcomes such as seizure frequency and blood serum determinations should be used in place of pencil-and-paper self-report inventories. The unexplored and critical problem of patient and family recruitment and attendance needs to be made a research priority. Most of all, patient education and psychosocial treatment programs that are shown to be effective in well-designed research studies need to become integral parts of the treatment stream in epilepsy specialty practices.

7. Summary of specific programs Please refer to Table 1 for a comparison of these studies. The letters in square brackets refer to the name given to each study found in the Program Name row of Table 1.

7.1. MOSES [MOSE] (May and Pfafflin [2]) MOSES presents nine modular content units to groups of 10 adults with epilepsy in 14 one-hour lessons. The goals of MOSES are to improve patients’ knowledge and understanding about their epilepsy, its treatment, and their ability to cope with psychosocial consequences. MOSES covers living with epilepsy, epidemiology, basic knowledge, diagnostics, therapy, self-control, prognosis, psychosocial aspects, and networking. German language with recent translation to English; has also been used in Lithuania and the Czech Republic.

7.2. FAMOSES [FAMO] (Wohlrab et al. [3]) FAMOSES is an adaptation of the MOSES modular educational program for children with epilepsy and their parents. The goals of FAMOSES are to improve children’s and their parents’ knowledge about epilepsy and to help patients of childhood age and their parents achieve a better understanding of their disease, gain more self-confidence, and reduce specific fears regarding epilepsy. Two parallel sets of lessons are conducted for groups of 6 children and for groups of 10 parents. The child module includes orientation, basic facts, causes, diagnosis, therapy, social, and summary. The parent module includes overview, basic knowledge, diagnosis, therapy, prognosis, and living with epilepsy. German language.

7.3. The Penguin [Pengu] (Amdam et al. [4]) The Penguin uses cartoon penguins to help children aged 3–15 years learn and talk about epilepsy in four 1½-hour sessions. The goals of The Penguin are to guide children through their symptoms and seizures, to explain the causes of their epilepsy and their treatment, and to show them how to overcome and reduce anxiety and live as normal as possible with family and friends. These include what epilepsy is, seizure triggers, first aid, diagnostic tests, compliance, safety, and telling others. The program has been translated into English, German, Dutch, Finnish, Polish, and Hungarian.

7.4. Seizures & Epilepsy Education (SEE) program [SEE1, SEE2] (Helgeson et al. [5], Shore et al. [6]) The SEE program (formerly called Sepulveda Epilepsy Education) is a 2-day (16-hour) weekend seminar using an information medicine model that is delivered through psychoeducational techniques. The goal of the SEE program is to provide psychotherapy for patients with epilepsy and their family members using an economically practical delivery system. It is the only large audience intervention, with a typical attendance of 100–300 patients, family members, and professionals at a time. SEE is appropriate for people aged 12 years or older. SEE provides education about the medical aspects of epilepsy, including physiology, causes, epidemiology, diagnosis, details of medication treatment, surgery, lifestyle and triggers, first aid, and prognosis. Common problems and coping skills for the psychological and emotional challenges of epilepsy, impact on the family, effects on social functioning, and vocational adjustment are covered. SEE1 and SEE2 are two separate studies with different patient populations. English language. 7.5. FLIP&FLAP [FLIP] (Jantzen et al. [7]) FLIP&FLAP consists of three parallel sets of educational sessions for children with epilepsy over age 8, adolescents with epilepsy, and their parents. The program uses age-appropriate training materials, including cartoons, and manuals. A physician and psychologist present the 2½-day sessions. The goals are to improve (1) disease knowledge, (2) disease-related emotions, (3) communication, (4) self-responsibility, (5) self-management, (6) social participation, and (7) educational insecurity. German language. 7.6. PEPE [PEPE] (Huber and Seidel [8]) PEPE uses cartoons and an adult story line to teach self-care and independence to adults with epilepsy with limited intellectual functioning or other learning disabilities. The goals are to improve compliance, assist acceptance of epilepsy as a chronic illness, and support the process of becoming independent. The course consists of eight 2-hour sessions presented by a physician and an educator that cover common causes of epilepsy, types of seizures, treatment of epilepsy, comorbid learning and physical disabilities, prejudices, stigmatization, seizure triggers, work and employment, leisure, and social partnership. German language. 7.7. Acceptance and Commitment Therapy (ACT) program [ACT1, ACT2] (Lundgren et al. [9, 10]) ACT is an extension of cognitive behavior therapy. The goals are to increase psychological flexibility around the chain of seizure development, decrease the epilepsy-related experiential avoidance, broaden the person’s behavior repertoire, and motivate activity in the person’s chosen life direction. It involves teaching people to accept their predisposition to seize along with the fears, thoughts, and memories associated with epilepsy and to actively choose coping actions. The treatment is a combination of individual and small group therapy aimed at seizure reduction and improved quality of life. ACT1 and ACT2 are two separate studies with different patient populations. Used in India or South Africa. 7.8. Intervention in Self-Management for Persons with Epilepsy [ISPE] (Pramuka et al. [11]) ISPE uses the self-efficacy model of medical self-management and psychosocial coping in epilepsy. The intervention is conducted in six 2-hour sessions for 6–12 adults. Its goals are to improve selfefficacy and locus of control through goal-setting activities related

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to medical care and stress reduction, thereby resulting in improved quality of life. Topics include taking charge of your medical care, self-advocacy, stress, schedules and goals, relationships, and future. English language. 7.9. Be Seizure Smart [BSzS] (Austin et al. [12]) Be Seizure Smart is an educational intervention for children with epilepsy and their parents via telephone. The goal is to provide individually tailored interventions for each family member by (1) providing information about epilepsy, treatment, and seizure management according to the individual’s knowledge base; (2) addressing unique concerns and fears; and (3) providing emotional support. A trained nurse provides three 1-hour family sessions plus one group session with several families over a conference call. English language. 7.10. Children’s Epilepsy Program (CEP) [CEP1, CEP2] (Lewis et al. [13,14]) CEP consists of two parallel sets of four 90-minute lessons for groups of 7–10 children and adults. For the children, the goals are enhancing knowledge, self-esteem, self-care, and social communication skills, promoting responsibility on the part of the child, decreasing parental anxiety, and making it easier for both the children and their parents to live with the attendant stresses of epilepsy. For the parents, the goals are to deal with their anger, resentment, and grief related to the loss of a normal child; increase their knowledge about caring for their child; reduce anxieties related to having a child with a seizure disorder; and improve their decision-making skills. The children’s track, led by two professionals, includes understanding body messages, controlling seizures with medicines, telling others matter-of-factly, coping, and balancing my life. The adult track, led by one professional, consists of sharing the experience of having a child with epilepsy, making decisions and problem solving, working as a family system, and coping and adapting. Results of this single study were published in two separate parts: child and parent. English and Spanish language. 7.11. Ogata’s program [OGA] (Ogata and Kouichiro [15]) The authors used eight 2-hour group psychotherapy sessions led by a psychologist to treat family problems among 10 adults with epilepsy. Their goal was the prevention and amelioration of family problems. Japanese language. 7.12. Gillham’s program [GIL] (Gillham [16]) Gillham created two separate one-on-one, seven-session interventions. The goal was reduction in seizure frequency and psychological symptoms. In the Self-Control of Seizures track, adults are taught how to abort seizures, control seizure triggers, and use relaxation. In the Alleviation of Psychological Disorder track, adults are counseled for a target problem individually identified for each participant. English language. 7.13. ACINDES [ACIN] (Tieffenberg et al. [17]) ACINDES is a small group treatment designed for children with epilepsy and their parents. The goals are to train children to assume a leading role in the management of their health; train parents to learn to be facilitators; and train physicians to provide guidance, acting like counselors. In separate groups, children and parents learn (1) about the child’s condition and to identify body signals and early warning signs; (2) recognize the elements of

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equilibrium and identify triggers; (3) understand treatment, therapeutic alternatives, and the usefulness of a direct patient–physician relationship; (4) handle specific risk situations (identify risks and learn strategies to handle them, including emergency home treatments); and (5) develop appropriate decision-making strategies. Spanish language. 7.14. Take Charge of Epilepsy [TCE] (Snead et al. [18]) Take Charge of Epilepsy contains six 1-hour weekly parallel sessions for adolescents and their parents in groups of five. The goal is to improve the quality of life of adolescents with epilepsy. Psychoeducational techniques are used to teach about adolescent issues, medical aspects, healthy lifestyle and attitudes, positive relationships with peers, family coping, stress management, and skill development. English language. Conflict of interest statement The author of this article is also the author of the Seizures & Epilepsy Education program. He had no role in SEE program outcome research cited herein aside from presenting the program for independent researchers. References [1] Austin JK. The 2007 Judith Hoyer lecture: epilepsy comorbidities: Lennox and lessons learned. Epilepsy Behav 2009;14:3–7. [2] May TW, Pfafflin M. The efficacy of an educational treatment program for patients with epilepsy (MOSES): results of a controlled, randomized study. Epilepsia 2002;43:539–49. [3] Wohlrab GC, Rinnert S, Bettendorf U, et al. Famoses: a modular educational program for children with epilepsy and their parents. Epilepsy Behav 2007;10:44–8. [4] Amdam A, Landslet B, Homseth TA. The Penguin: a teaching plan for children with epilepsy. In: Pfafflin M, Frasier RT, Specht U, Thorbeck R, editors. Comprehensive care for people with epilepsy. John Libbey Eurotext; 2001. p. 163–8. [5] Helgeson DC, Mittan R, Tan SY, Chayasirisobhon S. Sepulveda Epilepsy Education: the efficacy of a psychoeducational treatment program in treating medical and psychosocial aspects of epilepsy. Epilepsia 1990;31:75–82. [6] Shore CP, Perkins SM, Austin JK. The Seizures and Epilepsy Education (SEE) Program for families of children with epilepsy: a preliminary study. Epilepsy Behav 2008;12:157–64. [7] Jantzen S, Muller-Godeffroy E, Hallfahrt-Krisl T, et al. FLIP&FLAP: a training programme for children and adolescents with epilepsy, and their parents. Seizure 2009. doi:10.1016/j.seizure.2009.04.007. [8] Huber B, Seidel M. PEPE: a psychoeducative programme for people with epilepsy and learning disabilities. In: Pfafflin M, Frasier RT, Specht U, Thorbeck R, editors. Comprehensive care for people with epilepsy. John Libbey Eurotext; 2001. p. 155–62. [9] Lundgren T, Dahl J, Yardi N, Melin L. Commitment therapy and yoga for drugrefractory epilepsy: a randomized controlled trial. Epilepsy Behav 2008;13:102–8. [10] Lundgren T, Dahl J, Melin L, Kies B. Evaluation of acceptance and commitment therapy for drug refractory epilepsy: a randomized controlled trial in South Africa—a pilot study. Epilepsia 2006;47:2173–9. [11] Pramuka M, Hendrickson R, Zinski A, Van Cott AC. A psychosocial selfmanagement program for epilepsy: a randomized pilot study in adults. Epilepsy Behav 2007;11:533–45. [12] Austin JK, McNelis AM, Shore CP, Dunn DW, Musick B. A feasibility study of a family seizure management program: ‘‘Be Seizure Smart”. J Neurosci Nurs 2002;34:30–7. [13] Lewis MA, Salas I, de la Sota A, Chiofalo N, Leake B. Randomized trial of a program to enhance the competencies of children with epilepsy. Epilepsia 1990;31:101–9. [14] Lewis MA, Hatton CL, Salas I, Leake B, Chiofalo N. Impact of the Children’s Epilepsy Program on parents. Epilepsia 1991;32:365–74. [15] Ogata A, Kouichiro A. A psychosocial approach to epileptic patients. Epilepsia 2000;41(Suppl. 9):36–8. [16] Gillham R. Refractory epilepsy: an evaluation of psychological methods in outpatient management. Epilepsia 1990;31:427–32. [17] Tieffenberg JA, Wood EI, Alonso A, Tossutti MS, Vicente MF. A randomized field trial of ACINDES: a child-centered training model for children with chronic illnesses (asthma and epilepsy). J Urban Health 2000;77:280–97. [18] Snead K, Ackerson J, Bailey K, Schmitt MM, Madan-Swain A, Martin RC. Taking charge of epilepsy: the development of a structured psychoeducational group

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