Cochrane Review: Care delivery and self-management strategies for children with epilepsy

Abstract

Background

Epilepsy care for children has been criticised for its lack of impact. Various service models and strategies have been developed in response to perceived inadequacies in care provision for children and their families.

Objectives

We set out to compare the effectiveness of specialist or dedicated teams or individuals in the care of children with epilepsy with usual care services.

Search methods

We searched the Cochrane Central Register of Controlled Trials (CENTRAL) (The Cochrane Library February issue, 2010), MEDLINE (1950 to March 2010), EMBASE (1988 to May 2006*), PsycINFO (1806 to March 2010) and CINAHL (1982 to March 2010).

*Please note that as we currently do not have access to EMBASE, we have been unable to update this aspect of our searching.

Selection criteria

We included randomised controlled trials, controlled or matched trials, cohort studies or other prospective studies with a control group, or time series studies.

Data collection and analysis

Each review author independently selected studies, extracted data and assessed the quality of included studies.

Main results

Four trials and five reports are included in this review. They report on four different education and counselling programmes for children, children and parents, or teenagers and parents. Each programme showed some benefits for the well-being of children with epilepsy, but each trial had methodological flaws and no single programme was evaluated by more than one study.

Authors' conclusions

While each of the programmes in this review showed some benefit to children with epilepsy their impacts were extremely variable. No programme showed benefits across the full range of outcomes. No study appears to have demonstrated any detrimental effects but the evidence in favour of any single programme is insufficient to make it possible to recommend one programme rather than another. More trials, carried out by independent research teams, are needed.

Plain Language Summary

Care delivery and self-management strategies for children with epilepsy

Evidence for the best ways to care for children with epilepsy is still unclear.

This review compared four education- or counselling-based interventions for children with epilepsy. One intervention was aimed solely at children, two were aimed at children and their parents, the final intervention was aimed at teenagers and their parents. Each of the interventions appeared to improve some of the outcomes studied, but no intervention improved all of the outcomes that were measured. The studies also had problems with their methods, which makes their results less reliable. While none of the interventions caused any harm, their impact was limited and we cannot recommend any single intervention as being the best one for children with epilepsy.

Background

Description of the condition

In the developed world, epilepsy is the most common serious neurological condition after stroke, with approximately one in 200 people having epilepsy at any one point in time. For any individual there is a two to three per cent lifetime risk of an epilepsy diagnosis.

Description of the intervention

Epilepsy care has been criticised as having limited impact by not fully addressing all the health needs of children with epilepsy. It has also been suggested that standard epilepsy care leads to reduced compliance with agreed care plans (Betts 1992). Various models of service provision have been developed in response to perceived deficiencies in the quality of care, for example, educational initiatives to improve self-management, improved liaison between primary (GP) and secondary/tertiary (hospital-based) care including input from social care or the voluntary sector and specialist services for epilepsy in hospital. These services may include specialist clinics for specific sub-groups such as teenagers or combine elements from some or all models in multi-disciplinary clinical networks.

How the intervention might work

These models of care may have advantages over the management of children in general care. Education for self-management has been shown to be effective for other long-term conditions. Access to professionals with specialist training in epilepsy is limited, so improved liaison between services may improve the quality of care, promote more systematic multi-disciplinary follow-up of individual children and enhance communication between professionals, children, parents and other services. On the other hand, children and families may benefit from non-specialist services in terms of shorter waiting times and improved continuity of care as a small number of professionals are involved.  When available, specialist hospital-based clinics may allow expertise to be concentrated in one service making access to multi-disciplinary assessment and treatment easier to obtain.

Why it is important to do this review

The aim of this work is to systematically review the evidence from studies investigating the effectiveness of these service models compared to non-specialist services for children with epilepsy and their families. Effective care of children with epilepsy can reduce the impact of the disorder (for example, by reducing seizure frequency), improve social wellbeing and enable greater independence for these children and their families.

Objectives

To compare the effectiveness of any specialised or dedicated intervention for the care of children with epilepsy and their families to the effectiveness of usual care.

Methods

Criteria for considering studies for this review

Types of studies

We included several study types in the review as the interventions considered were highly variable and complex. The inclusion criteria for studies were based on those used by The Cochrane Effective Practice and Organisation of Care (EPOC) Group. We included all randomised controlled, controlled or matched trials, cohort or other prospective studies with a control group or time series studies.

Types of participants

Anyone with any diagnosis of new or recurrent epilepsy under 18 years of age. We included studies incorporating epilepsy with other long term conditions if results were reported for each condition separately.

Types of interventions

Any specialised or dedicated team or individual for the care of children with epilepsy whether based:

• (a)
  in hospital e.g. a specialist epilepsy clinic;
• (b)
  in the community e.g. a specialist pharmacist;
• (c)
  in general practice e.g. a specialist epilepsy nurse;
• (d)
  elsewhere e.g. social worker, the voluntary sector;
• (e)
  as a care network combining any of these elements;
• (f)
  on education or counselling for improved self-management.

Types of outcome measures

The outcome measures to be considered are:

• (a)
  seizure frequency and severity;
• (b)
  appropriateness and volume of medication prescribed (including evidence of drug toxicity);
• (c)
  child or family's reported knowledge of information and advice received from professionals;
• (d)
  child or family's reports of health and quality of life (including side-effects of medication);
• (e)
  objective measures of general health status;
• (f)
  objective measures of social or psychological functioning (including the number of days spent on sick leave/absence from school and employment status);
• (g)
  costs of care or treatment.

Particular emphasis is given to studies that report on outcomes measured at regular intervals over a long time period (at least every six months and for at least two years). All outcome measures are assessed for reliability and validity. If measures were misused (e.g. adults scales used on children) we investigated their effect on study results by a sensitivity analysis.

Search methods for identification of studies

We searched the Cochrane Epilepsy Group Specialized Register (15 March 2010) and the following databases.

1. Cochrane Central Register of Controlled Trials (CENTRAL) (The Cochrane Library February issue, 2010). See Appendix 1 for details of search strategy.

2. MEDLINE (Ovid) (1950 to March week 1, 2010). See Appendix 2 for details of search strategy.

3. EMBASE (1988 to May 2006). See Appendix 3 for details of search strategy. (Please note that as we currently do not have access to EMBASE, we have been unable to update this aspect of our searching.)

4. PsycINFO (1806 to 15 March 2010). See Appendix 4 for details of search strategy.

5. CINAHL (1982 to 15 March 2010). See Appendix 5 for details of search strategy.

Finally we contacted experts in the field to seek information on unpublished and ongoing studies, checked the websites of epilepsy organisations and checked the reference lists of included studies.

Data collection and analysis

Selection of papers

Two review authors, one with specialist knowledge of epilepsy (BL) and one without (PB), independently selected articles. We compared results and resolved disagreements by discussion.

Data extraction and quality assessment

The same review authors extracted data and assessed quality of included studies based on explicit criteria used by The Cochrane Effective Practice and Organisation of Care (EPOC) Group. We resolved any disagreements by discussion. If reports provided inadequate information, we contacted authors for further information.

Data analysis

We assessed clinical heterogeneity between studies by reviewing the differences across trials. There was considerable clinical heterogeneity in the trials so a meta-analysis was not considered appropriate. If we had decided to combine the results of any trials in a meta-analysis, we would have investigated heterogeneity with an I² test. If the results had shown heterogeneity the cause would have been investigated.

If studies had been of a suitable quality and sufficiently homogeneous to combine in a meta-analysis, we would have used (standardised) weighted mean differences for continuous variables and relative risks (including Mantel Haenzsel analysis) for dichotomous variables using either a random-effects or fixed-effect model.

Results

Description of studies

See: Characteristics of included studies; Characteristics of excluded studies; Characteristics of studies awaiting classification.

Initial searches identified over 4000 papers, including duplicates. On first assessment 164 were assessed as potentially relevant to this review. Of these, we rejected 135 based on information from the title and abstract. Of the remaining 29 papers, seven were based on studies of children. We excluded three papers because they did not meet the criteria for inclusion in this review.

The remaining papers, four in total, are included in this review. A fifth paper (Lewis 1990) was identified from the reference list of Lewis 1991: both of these papers report on the same study.

All of the included studies investigate interventions for improved self-management: these interventions were identified by the review authors as either education, counselling or training. No included study investigated specialist teams of health or social care professionals either in hospital or community settings. One study included children only (Tieffenberg 2000), two included children and parents (Lewis 1990 and Lewis 1991; Rau 2006 ) and a single study involved teenagers and parents (Glueckauf 2002).

Strategies for children

Tieffenberg 2000 reported on the effects of ACINDES, a model for self-management training based on play techniques. The model was developed by the researchers specifically for Spanish-speaking children aged 6 to15 years. The study undertook a randomised controlled trial of 355 children in Buenos Aires, Argentina.

ACINDES was designed to train children in self-management of chronic conditions and is not epilepsy-specific. It is delivered by specially selected and trained teachers to small groups of children. The programme consists of five 2-hour meetings, held weekly, plus a "reinforcement meeting" held 2 to 6 months afterwards. Groups of children are arranged according to age (6 to 8 years, 9 to 12 years, 13 to 15 years) with no more than 10 children per teacher. Parent groups are not arranged according to the ages of the children, and are co-ordinated by one or two teachers.

Strategies for children and parents

Lewis 1990 evaluated the use of the Children's Epilepsy Program (CEP) with a group of children in Santiago, Chile. The study recruited 252 children aged 7 to 14 years, selected from 1000 families belonging to the Liga Contra Epilepsia. The sampling process was not described. Families were randomly allocated in groups of 20 to the experimental and control groups (the randomisation process was not described). The experimental group (n = 123) undertook the CEP, described below. The control group (n = 113) attended three 2-hour sessions consisting of lecturers and question and answer discussions. These sessions were delivered by a physician and presented the same information as was delivered in the CEP. It is not clear who delivered the CEP itself.

The CEP was developed and piloted with 40 children with epilepsy (aged 7-12 years) at the Medical Center of the University of California in Los Angeles (UCLA). For unreported reasons the researchers could not recruit a suitable sample from the UCLA Medical Center and so the main study was transferred to Chile. The CEP was translated into Spanish for the trial.

The CEP consists of four sessions, each lasting 1.5 hours and delivered at weekly intervals. Children and parents were taught separately, meeting to share experiences at the end of each session. Each session had a specific theme:

Session 1: Understanding body messages

This used electronic toys and cartoon drawings to teach children about seizures and to help them identify seizure-related emotions and feelings.

Session 2: Controlling seizures with medication

This focused on seizure-related information, using a card-sorting exercise to separate facts and fictions about seizures. It also taught seizure management and decision-making skills.

Session 3: Telling others matter of factly

In this session the children were encouraged to share personal experiences, especially experiences with friends or peers, whether related to epilepsy or not. Children learned how to tell others about their epilepsy.

Session 4: Coping and adapting to balance my life

Various exercises were used to develop coping skills, including ways of dealing with bullying or taunting or with negative attitudes.

The children were pre-tested immediately prior to the first session and post-tested five months after the end of the CEP. Over 97% of experimental group children and 90% of controls completed both tests. However, only 78.6% of experimental children and 52% of controls attended all the required sessions.

Lewis 1991 reported on the impact of the CEP on parents. The parental arm of the CEP followed the same basic structure as the child-focused arm but was reported as being based on a Rogerian model of counselling as well as enabling parents to review the children's sessions as described above.

Families were randomly assigned to experimental or control groups. The intervention group received four sessions as described below, the control group received three 2-hour sessions, delivered by a physician and consisting of a lecture followed by a question and answer session. This control was described as "passive learning" in contrast to the "active learning" of the intervention. The experimental group consisted of 185 parents (120 mothers, 65 fathers), the control group of 180 parents (109 mothers, 71 fathers).

The parental sessions for the intervention group were as follows (the paper does not report on who delivered these sessions):

Session 1: Telling a story

Parents introduced themselves to other group members and told of their experiences with their child with epilepsy. They undertook a card-sorting exercise to dispel false perceptions or myths.

Session 2: Making decisions

A decision-making process was used to develop decision-making skills.

Session 3: Working as a family system

The group developed their understanding of how a child's epilepsy can impact on family life and discussed their parenting styles.

Session 4: Coping and adapting

In this final session parents discussed how to be more open about their child's epilepsy and how to acknowledge the pain and grief which may arise when parenting a child with a chronic condition.

Rau 2006 evaluated FAMOSES, the modular educational program for children with epilepsy and their parents, by means of a prospective, controlled, multi-centre study. Children with epilepsy, aged from 7.2 to 15.9 years, and parents were allocated to experimental or control groups. FAMOSES aims to improve knowledge, coping, treatment outcomes and adaptation to epilepsy through a series of educational modules which are undertaken by participants. While the content of the sessions for parents and children was similar, focusing on topics such as basic knowledge, diagnosis, treatment and living with epilepsy, each group was taught separately. The children's content was presented as a journey by sea, sailing from the harbour to a series of 'islands', each of which represented a separate content area.

Strategies for teenagers and parents

Glueckauf 2002 undertook a 3-arm randomised controlled trial of a counselling programme based on video-conferencing for teenagers and their families based in the rural Midwest of the United States of America. Twenty-two teenagers with seizure disorders (14 male, 8 female, mean age 15.4 years (SD 2.5 years)) completed step 1 of the project along with 36 parents (21 mothers and 15 fathers). All participants were Caucasian. A total of 39 families were recruited but 12 dropped out before counselling and 5 more dropped out prior to 6 month follow-up. Teenagers were randomly assigned, with their parents, to one of three groups: group 1 were to receive counselling via videophone (VFC), group 2 received office-based counselling (OFC) and group 3 were placed on a waiting list. However, it was established that VFC was not possible for every family allocated to receive it because of a lack of digital services and as a result 4 of the 9 families allocated to VFC were instead given counselling by speakerphone (SFC).

Risk of bias in included studies

For children

Reporting of methodological details was variable in this randomised controlled trial (Tieffenberg 2000).  No details of randomisation, allocation concealment, power calculations or required sample size were reported. No details were provided of families lost to follow-up, but reasons for non-attendance were provided.  No details were given of how drop-outs were accounted for.  Clinicians were not blinded.  The main trial compared the results of a self-management educational programme for children with either epilepsy or asthma.  However, a sub-analysis compared the effect of a self-management educational programme for children with epilepsy.  For those children with epilepsy 13.6% of participants were lost to follow up in the experimental group and 29.7% in the control group.  No intention-to-treat analysis was reported.  The details of randomisation including the ‘clustering techniques’ used were not reported.  There was a possibility of contamination in both trials, as randomisation was not conducted by an independent centre.  There was no discussion about how baseline potential differences might affect the result of the study before the data was analysed, but no differences were reported. Participant outcomes were recorded before the programme and at six and twelve months after its completion. The trial authors used statistical methods appropriate to its design.

For children and parents

Lewis 1990, Lewis 1991

Reporting of methodological details was variable in this randomised controlled trial with two reports (Lewis 1990 and Lewis 1991).  No details of randomisation, allocation concealment, power calculations or required sample size were reported.  Ninety per cent of children in the control group and 97% in the knowledge group completed knowledge pre- and post-assessment questionnaires.  No details were provided of participants who withdrew or who were lost to follow-up.  No intention-to-treat analysis was explicitly reported, but data were reported for each individual in the final analysis despite attendance at the educational programme being incomplete. Clinicians were not blinded. There is a possibility of bias as the sample of participants was not randomly selected although participants were randomly allocated to experimental or control group.  There was no discussion about how baseline potential differences might affect the result of the study before the data were analysed.  Participant outcomes were recorded before the programme and five months after its completion.  The trial authors used statistical methods appropriate to its design but eliminated data on scholastic achievement for all children under 8 in the final analysis, because the subscale used to measure this was deemed inappropriate for this age group. Nonetheless, covariate adjustment was performed for competency scores based on pre-test values, age and sex. In Lewis 1991 while almost 100% of parents completed the pre- and post-tests, in the control group only 62% of mothers and 49% of fathers attended all three sessions and in the experimental group 73.2% of mothers and 59% of fathers attended all four sessions.

Rau 2006

Reporting of methodological details was variable in this prospective, controlled non-randomised trial (Rau 2006).  Patients were consecutively assigned to intervention and control groups.  No details of power calculations or required sample size were reported. A total of 70 children and 159 adults were recruited (some parents took part even though their children did not) but only 50 children and 103 adults completed the post-programme evaluation questionnaire and so were included in the analysis.  Information on the number of participants lost to follow up was not provided for adults by intervention group. For children the drop-out rate in the control group was 40.6% and 18.4% for the training group. Children with other conditions were significantly more common among the non-responders. The return rate of the questionnaire at the second time measurement was 76.4% of the total sample (adults: 78.6%, children: 71.4%). No details were given of how drop-outs were accounted for, but only those completing the intervention programme were included in the final analysis and numbers vary for each outcome considered.  The researchers but not the clinicians were blinded.  There was a considerable risk of contamination as this was a quasi-randomised trial. There was no discussion about how baseline potential differences might affect the result of the study before the data were analysed.  Participant outcomes were recorded at baseline and three months after the Modular education programme (FAMOSES). The control groups took part in the training after the second measurement. The statistical tests used were not fully reported.  Where they were reported they were appropriate to its design.

For teenagers and parents

Reporting of methodological details was variable in this randomised controlled trial (Glueckauf 2002).  No details of randomisation, allocation concealment, power calculations or required sample size were reported.  The randomisation failed in this trial as several families allocated to video-conference based family counselling (VFC) were unable to technically support it and were offered speakerphone family counselling as an alternative (SFC).  Details were given of how drop-outs were accounted for and it was recognised that non-responders were more likely to have other illnesses or disabilities.  Thirty-one per cent (12/39) of families did not attend any counselling and a further 13% (5/39) did not complete all sessions. No intention-to-treat analysis was reported.  There was a possibility of contamination in both trials, as randomisation was potentially done at the patient level, rather than by an independent centre.  There was limited discussion about how baseline potential differences might affect the result of the study before the data was analysed, but no differences were reported. Questionnaires were received by participants pre-evaluation, after their sixth session and at six-month follow-up.  The trial authors used statistical methods appropriate to its design.

Effects of interventions

Throughout, it should be noted that we have presented the results of the trials as reported in the published papers and that the presentation of results varied considerably between trials. Moreover, given that the outcome measures differed for each trial, we concluded that meta-analysis of the results, even within type of outcome would be inappropriate.

Tieffenberg 2000

At twelve months an evaluation of ACINDES (a child-centered model for children with chronic illness) showed no significant differences between groups in gain in parents' knowledge (P = 0.62) or reduction in fears of their child's death (P = 0.63).  

At twelve months the evaluation showed significant difference between groups in terms of some clinical outcomes:

(a) emergency visits in 12 months (intervention mean 0.90 (before) SD 0.95 to mean 0.22 SD 0.58, control mean (before) 0.83 SD 0.95  to mean 0.46 SD 0.66, P = 0.046);

(b) epileptic seizures in 12 months (intervention mean 0.80 (before) SD 1.46 to mean 0.34 SD 0.98, control mean (before) 0.49 SD 1.15  to mean 1.11 SD 2.77, P = 0.036).

The number of regular medical visits fell in each group (intervention mean 3.64 (before) SD 3.01 to mean 3.06 SD 2.57, control mean (before) 3.89 SD 4.47  to mean 2.91 SD 3.19) but the differences were reported as "not significant".

At twelve months the evaluation showed significant improvement in school absenteeism as measured by mean number of absences per 100 school days (intervention mean 10.31 (before) to mean 6.85, control mean (before) 9.32 to mean 9.21, P = 0.011).

Children's scores for health ‘locus of control’ are not reported separately for those with epilepsy and asthma.

Lewis 1990 At five months an evaluation of the Children's Epilepsy Program showed significant differences between groups in percentage of children responding correctly to the following knowledge items:

(a) inappropriate to have objects in mouth during seizure (intervention mean (before) 40.7 to mean 71.5, control mean (before) 44.3  to mean 52.2, P = 0.002;

(b) inappropriate to restrain during seizure (intervention mean (before) 34.9 to mean 79.7, control mean (before) 33.6 to mean 46.0, P = 0.001;

(c) not required to visit ER (emergency room) after seizure (intervention mean (before) 30.9 to mean 78.1, control mean (before) 29.2  to mean 52.2, P = 0.001;

(d) purpose of EEG (intervention mean (before) 62.6 to mean 82.1, control mean (before) 63.7  to mean 69.0, P = 0.02;

(e) restriction of activities should be minimal (intervention mean (before) 58.5 to mean 86.2, control mean (before) 58.4  to mean 68.1, P = 0.001.

Each group reported slightly improved scores for the following knowledge items but the differences were reported as "not significant": importance of taking medicines exactly as prescribed; knowledge that seizures start in the brain; purpose of drug blood levels to monitor dosage, positive effects of participation in sports. Both groups scored lower post-program for "loss of sleep can trigger seizures" but again differences were reported as "not significant".

At five months the evaluation showed significant differences between groups (excluding children under 8 years of age) in social competency after scores were adjusted for pre-test values, age and sex for social competency (intervention mean (before) 2.81 to mean 2.91, control mean (before) 2.77 to mean 2.76 P < 0.05).  "Non significant" changes were seen (excluding children under 8 years of age) for; scholastic competency (intervention mean (before) 2.41 to mean 2.58, control mean (before) 2.62 to mean 2.55), athletic competency (intervention mean (before) 2.69 to mean 2.80, control mean (before) 2.81 to mean 2.82), appearance competency (intervention mean (before) 2.96 to mean 3.01, control mean (before) 3.07 to mean 3.04), behaviour competency (intervention mean (before) 2.58 to mean 2.75, control mean (before) 2.74 to mean 2.68), or self-esteem competency (intervention mean (before) 2.97 to mean 2.99, control mean (before) 3.02 to mean 3.10).

At five months, children in the intervention group were more likely to report; generic gain in knowledge to the question "what were the important things that you learned" (intervention percentage mean 64% to the control percentage mean 47%, P < 0.01); to report gain in social skills (intervention percentage mean 9% to the control percentage mean 2%, P < 0.02); participation in normal activities (intervention percentage mean 11% to the control percentage mean 3.5%, P < 0.03). "Non-significant" changes were seen for children's self-care skills or children's reports of parents' behaviours or their disclosure of their epileptic status (no scores were given). Two thirds of children reported doing nothing different as a result of programme participation.

Lewis 1991

At five months an evaluation of the Children's Epilepsy Program showed significant differences between groups in percentage of parents responding correctly to the following knowledge items:

(a) loss of sleep can trigger seizures (intervention mean (before) 62.7 to mean 50.3, control mean (before) 66.3  to mean 65.2, P = 0.005);

(b) purpose of EEG (intervention mean (before) 80.0 to mean 90.3, control mean (before) 81.1  to mean 83.3, P = 0.05);

(c) purpose of drug blood levels to monitor dosage (intervention mean (before) 63.4 to mean 79.6, control mean (before) 67.2  to mean 87.8, P = 0.04.

No significant changes were seen for the following knowledge items: importance of taking medicines exactly as prescribed (intervention mean (before) 94.6 to mean 97.3, control mean (before) 97.8 to mean 99.0); inappropriate to have objects in mouth during seizure (intervention mean (before) 35.3 to mean 78.8, control mean (before) 35.6 to mean 76.1); inappropriate to restrain during seizure (intervention mean (before) 52.2 to mean 76.3, control mean (before) 56.7 to mean 81.1); not required to visit ER (emergency room) after seizure (intervention mean (before) 68.1 to mean 93.0, control mean (before) 71.1 to mean 88.3); knowledge that seizures start in the brain (intervention mean (before) 86.0 to mean 93.5, control mean (before) 86.7 to mean 90.0; restriction of activities should be minimal (intervention mean (before) 88.6 to mean 96.7, control mean (before) 93.3 to mean 97.2; positive effects of participation in sports (intervention mean (before) 80.5 to mean 95.1, control mean (before) 73.3 to mean 90.0).

At five months an evaluation of the Children's Epilepsy Program (Lewis 1991) showed significant differences between groups in parental anxiety (Taylor Manifest Anxiety Scale).  Parents in the experimental group showed greater reduction in anxiety than that of control parents (intervention mean (before) 52.5 to mean 47.9, control mean (before) 50.2 to mean 48.7, P < 0.01, although the effect was not significant for fathers of children when analysed alone.

In order to assess the impact of attending all of the educational sessions versus only some, the authors performed two-way analysis of variance on changes in parents' anxiety scores on starting and leaving the programme, but no significant results were shown.

At five months the evaluation showed significant differences between groups in terms of knowledge gain.  Intervention parents were more likely to report; generic gain in knowledge to the question "what were the important things that you learned" (intervention percentage mean 59% versus the control percentage mean 48%, P < 0.05) or recognising the importance of medicines (intervention percentage mean 19% versus the control percentage mean 9%, P < 0.01).  At five months, there was a significant difference in the proportion of parents who reported feeling less anxious after the sessions (intervention percentage mean 31% versus 10%, P < 0.001).  At five months, there was a significant difference in the proportion of parents who endorsed the programme (P < 0.001), with more parents endorsing it in the intervention group. However, only 6% of parents in the control group reported no benefit.  No significant changes were seen in answer to the question "do you deal with your child's seizure disorder differently after the sessions?" where 29% of control and 21% of intervention parents answered affirmatively.  No significant changes were seen in answer to the question "are you, as a family, doing anything different as a result of attending the programme?" where 63% of control and 53% of intervention parents answered negatively.

Rau 2006

At three months an evaluation of the modular education programme (FAMOSES) (Rau 2006) showed significant differences between groups in:

• (a)
  reduced social limitations amongst children (intervention mean (before) 83.87 standard deviation (SD) 28.38 to mean 90.32 SD 21.42, control mean (before) 94.74 SD 12.49 to mean 89.47 SD 19.41 P = 0.017);
• (b)
  increased knowledge amongst parents (intervention mean (before) 83.33 standard deviation (SD) 12.55 to mean 83.10 SD 13.36, control mean (before) 74.07 SD 16.78 to mean 89.76 SD 8.27, P = 0.000);
• (c)
  improved attitudes amongst parents (intervention mean (before) 28.86 standard deviation (SD) 15.71 to mean 32.56 SD 15.38, control mean (before) 32.99 SD 16.45 to mean 29.64 SD 14.14, P = 0.001);
• (d)
  reduced fears amongst parents (intervention mean (before) 43.02 standard deviation (SD) 21.41 to mean 45.75 SD 20.09, control mean (before) 46.14 SD 20.27 to mean 43.02 SD 18.82, P = 0.014);
• (e)
  reduced level of supervision needed and need for family resilience amongst parents (intervention mean (before) 66.15 standard deviation (SD) 26.04 to mean 65.10 SD 22.33, control mean (before) 60.20 SD 25.47 to mean 70.41 SD 23.20, P = 0.031);
• (f)
  improved behaviour during seizures amongst parents (intervention mean (before) 66.81 standard deviation (SD) 31.81 to mean 69.01 SD 28.44, control mean (before) 68.95 SD 29.25 to mean 81.37 SD 21.97, P = 0.029).

Outcome measures showing no significant differences between groups were: epilepsy frequency, epilepsy knowledge, fears, sporting limitations coping strategies, attitudes, tolerability of anti-epileptic drugs (AEDs), effectiveness of AEDs, days missed at school, quality of life as assessed by parents and children.  Outcome measures showing no significant differences for parents were: coping strategies, sporting and social limitations, restrictions because of epilepsy.

Glueckauf 2002

At six months an evaluation of video-conference based family counselling (VFC) and speakerphone family counselling (SFC) showed no differences in outcome measures between groups for: issue frequency for teenagers or parents, issue severity for teenagers or parents, pro-social behaviour scale for parents or teachers, problem behaviour scale for parents or teachers, adherence to treatment, number of missed appointments or therapeutic alliance.

However, before and after comparisons were made.  At one week post-treatment:

• Prosocial Behaviour scale is significantly improved compared to overall mean for parents, teachers (and time)

• Problem Behaviour scale is significantly improved compared to overall mean for parents and teachers

• Issue-severity is not significant for overall mean for teens or parents where overall mean is the pre-test to 6-month follow-up comparison

• Issue-frequency is not significant for overall mean for teens or parents

At 6-month follow-up:

• Issue-severity is significant for overall mean for teens and parents

• Prosocial Behaviour scale is significant for overall mean for parents and teachers

• Problem Behaviour scale is significant for overall mean for parents and teachers

• Issue frequency is not significant for overall mean for teens or parents

Discussion

Summary of main results

Four trials and five reports are included in this review. Two types of intervention were identified, both of which aimed to improve self-management: educational interventions (Lewis 1990 and Lewis 1991; Rau 2006; Tieffenberg 2000) and a counselling intervention (Glueckauf 2002). The studies were undertaken in diverse locations and investigated the use of a range of innovative interventions with children and parents.

With the exception of FAMOSES (Rau 2006) all of the interventions were designed, delivered and evaluated by the researchers who authored the reports. No intervention was evaluated on more than one occasion.

Quality of the evidence

The quality of evidence is generally poor, with all five reports containing major methodological problems. Problems include incomplete randomisation,  high drop-out rates, no intention-to-treat analysis and absence of blinding of study personnel. Data analysis was generally undertaken using appropriate methods. Although all of the studies investigated self-management improvement strategies, no individual strategy was investigated by more than one trial. Each study used a unique combination of outcome measures and no single intervention was found to be consistently effective across the full range of reported outcomes.

ACINDES (Tieffenberg 2000), when evaluated at 12 months, showed significant improvements for the intervention group in visits to the emergency room, in seizure numbers and in school absenteeism, but not in parental knowledge, parental fear of their child's death or number of routine medical consultations.

The Children's Epilepsy Programme (Lewis 1990; Lewis 1991) was evaluated five months after programme completion. The intervention group showed significant improvements over the control group in children's knowledge related to inappropriateness of putting objects into the mouth during a seizure, inappropriateness of restraining a person during a seizure, visits to the emergency room not being required after each seizure, the purpose of the EEG and the need to keep restriction of activities to a minimum. Other significant differences were reported relating to generic knowledge, social skills, participation in normal activities. Significant differences were also shown between groups in children over 8 years of age in social competency. However, there was no significant difference in scores relating to a series of other knowledge outcomes, self-care and competencies. Parents showed significant between group differences in some knowledge outcomes but not in others.

FAMOSES (Rau 2006) was evaluated at three months. Significant between groups differences were found for children in relation to reduced social limitations, reduced need for supervision and for parents in relation to increased knowledge, improved attitudes, reduced fears and improved behaviour during seizures. However, a range of other outcomes, including clinical outcomes, quality of life as measured by parents and parental restrictions showed no significant differences.

Counselling based on video conferencing (Glueckauf 2002) was evaluated at one week post treatment and again at six months. The Prosocial Behaviour Scale scores and Problem Behaviour Scale scores were significant at both time points compared to pre-intervention scores for parents and teachers. The Issue-severity score was significant at six months but not at one week. The Issue Frequency Score was not significant. Between group scores were not significant at either time point.

Authors' conclusions

Implications for practice

The evidence from this review suggests that innovative models of service delivery may improve some outcomes relating to epilepsy in children and to the impact that epilepsy can have on parents. However, no single strategy improved a comprehensive range of user outcomes and methodological deficiencies within each study mean that the results must be treated with caution.

The evaluation of each programme is based on a single evaluation and in most cases design, delivery and evaluation were undertaken by the same team of researchers. At present there is insufficient evidence in favour of any single programme and so, while no programme was shown to impact negatively on children with epilepsy or their parents, it is not possible to recommend any single programme as being more effective than any other. No programme showed consistent improvement across all of the assessed outcomes. Health professionals and families need to be aware of this when considering any of these strategies for implementation.

Implications for research

This review has identified four distinct programmes for the education or counselling of children with epilepsy and their parents, aimed at improving self-management. However, no programme was evaluated on more than one occasion, the studies show methodological flaws, were not independently assessed, and show inconsistent results. The evidence from this review suggests that innovative models of service delivery may improve some outcomes relating to epilepsy in children and to the impact that epilepsy can have on parents. However, no single strategy improved a comprehensive range of user outcomes and methodological deficiencies within each study mean that the results must be treated with caution.

As a result, further studies are needed which:

• (1)
  offer an improved quality of study design and reporting;
• (2)
  improve generalisability (e.g. include a full description of the intervention, a process evaluation, and a multi-centred assessment of the benefits for more than one population and service provider);
• (3)
  evaluate the effects of interventions for those subgroups most likely to benefit (e.g. children with newly diagnosed epilepsy, children with learning disabilities);
• (4)
  consider the cost-effectiveness of service models shown to be beneficial.

To maximise the potential of future studies for generalisability and to ensure study quality we would recommend randomised controlled trials rather than observational studies. Studies should also ensure that the interventions are adequately defined and described, and that contextual factors are taken into account in the study design. Where socially complex interventions such as these are under study sufficient service providers must be included to ensure that individual characteristics do not bias the results.

Acknowledgements

We would like to thank Laura Burfoot who helped us summarise the results from the studies.

Characteristics of studies

Characteristics of included studies [ordered by study ID]

Glueckauf 2002
Methods	Randomised controlled trial
Participants	Teenagers with epilepsy and behaviour problems, and their parents
Interventions	Issue Specific Family Counseling Model (ISFCM) delivered either a) via video-conferencing, b) via speakerphone or c) face-to-face in the counsellor's office.
Outcomes	Measures of change in severity and frequency of the behaviour problem; teenager's functional ability in school and home; adherence to intervention activities.
Notes
Risk of bias
Item	Authors' judgement	Description
Allocation concealment?	Unclear	B - Unclear

Lewis 1990
Methods	Randomised controlled trial (Santiago, Chile)
Participants	Children with epilepsy (aged 7-14 years)
Interventions	Children's Epilepsy Programme - a counselling model based on Rogerian principles
Outcomes	Knowledge about seizures, self-competency, self-perception related to knowledge, behaviour and parents' behaviour.
Notes
Risk of bias
Item	Authors' judgement	Description
Allocation concealment?	Unclear	B - Unclear

Lewis 1991
Methods	Randomised controlled trial (Santiago, Chile)
Participants	Parents of children with epilepsy
Interventions	Children's Epilepsy Programme - a counselling model based on Rogerian principles
Outcomes	Parental knowledge and anxiety: parental perception of the programme.
Notes
Risk of bias
Item	Authors' judgement	Description
Allocation concealment?	Unclear	B - Unclear

Rau 2006
Methods	Before and after study
Participants	Children with epilepsy (aged 7-16 years) and their parents
Interventions	Modular Education Programme Epilepsy for Families (FAMOSES)
Outcomes	Knowledge, coping, adaptation of children and parents. School attendance and seizure frequency in the children.
Notes
Risk of bias
Item	Authors' judgement	Description
Allocation concealment?	Unclear	D - Not used

Tieffenberg 2000
Methods	Randomised controlled trial
Participants	Children (aged 6-15 years) with asthma or epilepsy, and their parents.
Interventions	ACINDES: a child-centered training programme.
Outcomes	Knowledge, beliefs, attitudes and behaviours of the children. Prental knowledge, fear of child death. Clinical outcomes including seizure frequency, clinic attendance.
Notes
Risk of bias
Item	Authors' judgement	Description
Allocation concealment?	Unclear	B - Unclear

Characteristics of excluded studies [ordered by study ID]

Study	Reason for exclusion
Mar 2005	Audit of documentation and data recording.
Price 2004	Before and after (pre- and post-test) design. Study measured knowledge and skills of educators related to seizure management. No patient-related outcomes.
Snead 2004	Before and after (pre- and post-test) design. Small sample size (N = 7). No control group.

Characteristics of studies awaiting assessment [ordered by study ID]

Jantzen 2009
Methods
Participants
Interventions
Outcomes
Notes	To be assessed at next review update.

Shore 2008
Methods
Participants
Interventions
Outcomes
Notes	To be assessed at next review update.

Data and analyses

This review has no analyses.

Appendices

Appendix 1. CENTRAL search strategy

#1        MeSH descriptor Epilepsy explode all trees in MeSH products

#2        epilep* in All Fields in all products

#3        (#1 OR #2)

#4        MeSH descriptor Program Evaluation explode all trees in MeSH products

#5        MeSH descriptor Delivery of Health Care explode all trees in MeSH products

#6        (#4 OR #5)

#7        (#3 AND #6)

#8        MeSH descriptor Ambulatory Care explode all trees in MeSH products

#9        (#3 AND #8)

#10      epilep* NEAR/4 centre* in Title, Abstract or Keywords in all products

#11      epilep* NEAR/4 center* in Title, Abstract or Keywords in all products

#12      epilep* NEAR/3 specialist* in Title, Abstract or Keywords in all products

#13      epilep* NEAR/2 nurs* in Title, Abstract or Keywords in all products

#14      MeSH descriptor Outcome and Process Assessment (Health Care) explode all trees in MeSH products

#15      (#18 AND #3)

#16      (#7 OR #9 OR #10 OR #11 or #12 or #13 OR #15)

Appendix 2. MEDLINE search strategy

1. exp EPILEPSY/

2. epilep$.tw.

3. 1 or 2

4. exp Program Evaluation/

5. exp "Delivery of Health Care"/

6. 4 or 5

7. 3 and 6

8. exp Ambulatory Care/

9. 3 and 8

10. (epilep$ adj4 centre$).ab,ti.

11. (epilep$ adj4 center$).ab,ti.

12. (epilep$ adj3 specialist$).ab,ti.

13. (epilep$ adj2 nurs$).ab,ti.

14. exp "Outcome Assessment (Health Care)"/

15. 14 and 3

16. 7 or 9 or 10 or 11 or 12 or 13 or 15

Appendix 3. EMBASE search strategy

1 exp Epilepsy/

2 epilep$

3 1 or 2

4 exp Ambulatory Care/

5 exp Institutional Care/

6 exp Community Care/

7 exp Health Care Delivery/

8 *Outcomes Research/

9 (program$ adj2 evaluat$)

10 4 or 5 or 6 or 7 or 8 or 9

11 3 and 10

12 (center$ or centre$)

13 nurs$

14 specialist$

15 (epilep$ adj4 (centre$ or center$))

16 (epilep$ adj3 nurs$)

17 (epilep$ adj3 specialist$)

18 11 or 15 or 16 or 17

Appendix 4. PsycINFO search strategy

This search was carried out in two phases. The first search was carried out in May 2006 using the following strategy.

#10 #1 and #9

#9 #2 or #3 or #4 or #5 or #6 or #7 or #8

#8 specialist*

#7 nurs*

#6 centre* or center*

#5 treatment effectiveness evaluation

#4 treatment outcome*

#3 health care delivery

#2 ambulatory care

#1 epilep*

The second search was carried out in March 2010 using the EBSCOhost platform for PsycINFO, and the following strategy.

S12 S8 or S9 or S10 or S11

S11 S3 and S7

S10 epilep* N3 specialist*

S9 epilep* N3 nurs*

S8 epilep* N4 center* or epilep* N4 centre*

S7 S4 or S5 or S6

S6 MM "Program Evaluation"

S5 MM "Health Care Delivery"

S4 MM "Outpatient Treatment"

S3 S1 or S2

S2 epilep*

S1 MM "Epilepsy" or DE "Epileptic Seizures" or DE "Grand Mal Seizures" or DE "Petit Mal Seizures"

Appendix 5. CINAHL search strategy

This search was carried out in two phases. The first search was carried out in May 2006 using the Ovid platform for CINAHL, and the following strategy.

1. exp EPILEPSY/

2. epilep$.tw.

3. 1 or 2

4. exp Ambulatory Care/

5. exp Health Care Delivery/

6. exp Program Evaluation/

7. exp "Outcomes (Health Care)"/

8.(epilep$ adj4 (centre$ or center$)).tw.

9. (epilep$ adj3 nurs$).tw.

10. (epilep$ adj3 specialist$).tw.

11. 4 or 5 or 6 or 7

12. 3 and 11

13. 8 or 9 or 10 or 12

The second search was carried out in March 2010 using the EBSCO host platform for CINAHL, and the following strategy.

S13 S9 or S10 or S11 or S12

S12 S3 and S8

S11 epilep* N3 specialist*

S10 epilep* N3 nurs*

S9 epilep* N4 centre* or epilep* N4 center*

S8 S4 or S5 or S6 or S7

S7 (MM "Outcomes (Health Care)")

S6 (MM "Program Evaluation")

S5 (MM "Health Care Delivery")

S4 (MM "Ambulatory Care")

S3 S1 or S2

S2 epilep*

S1 (MH "Epilepsy+")

History

Protocol first published: Issue 4, 2006

Review first published: Issue 12, 2010

Contributions of authors

BL and PB developed the protocol for this review, independently reviewed papers for inclusion using Cochrane EPOC Group criteria and developed the final systematic review. PB led the analysis of included papers. BL wrote the final review.

Declarations of interest

None known.

Differences between protocol and review

PB was lead author on the protocol. Review methodology is unchanged from that included in the protocol.