Abstract
Objectives:
Seizure action plans help patients and caregivers better self-manage their epilepsy. We hypothesized that providing pediatric patients and their caregivers with a seizure action plan would reduce unplanned health care utilization and decrease the impact of epilepsy.
Methods:
We developed a seizure action plan for use in pediatric epilepsy patients. A prospective cohort was randomly assigned to receive a seizure action plan in addition to standard epilepsy care or to standard epilepsy care alone. All caregivers were surveyed using the Modified Impact on Families (MIF) questionnaire at enrollment, 3 months, and 12 months. Health care utilization measures and Modified Impact on Families questionnaire scores were compared between the 2 groups.
Results:
Fifty-four patients received a seizure action plan and standard care, whereas 48 received standard care alone. The groups had similar demographics. There was a significantly higher proportion of overall clinic appointment no shows in the standard care group vs the seizure action plan group (P = .04); however, other significant differences in health care utilization were not found. Among patients with low seizure frequency (12 or fewer seizures per year), Seizure comfort scores on the Modified Impact on Families questionnaire were significantly higher at 12 months among the seizure action plan group compared to the standard care group.
Significance:
Caregivers for patients with epilepsy receiving a seizure action plan were more comfortable regarding seizure care and missed fewer appointments. However, differences in health care utilization were not present. The seizure action plan appears to have more impact in patients who experience lower seizure frequencies. Further studies evaluating the impact as well as assessing caregivers’ perceptions of the seizure action plan using a larger sample are needed.
Introduction
Seizure action plans are designed to help patients and caregivers better self-manage their epilepsy. 1 -3 Clinicians and researchers employ written action plans for other chronic illnesses, such as asthma, to encourage disease self-management and efficacy. 4 -7 Data regarding the utility of written action plans for chronic disorder treatment and management, such as for epilepsy, is indeed conflicting 6 and deserving of further research and validation. A recent randomized controlled trial of an Asthma Action Plan in adults with chronic asthma did show some benefit in patient outcomes. 7 The Epilepsy Foundation developed a written seizure action plan currently used in practice across the United States, 8 but little is known regarding the efficacy of such a written plan for use in the pediatric population.
Care and treatment of epilepsy is complex and costly to patients and caregivers. 9 Health care utilization in children with epilepsy is costly, reaching millions to billions of health care dollars. Patients with poorly controlled seizures represent a higher proportion of health care utilization. 10 -12 Breakthrough seizures in adult epilepsy patients also carry a high cost burden. 13 However, in adults, education aimed at improving self-management has been shown to have a positive impact on quality of life and self-efficacy. 1,14 In children with epilepsy, parental anxiety, which is closely tied to parental self-efficacy, was shown to impact patients’ reported quality of life. 15 -17 However, results of a recent study indicated that a seizure action plan had no impact on health care utilization in pediatric epilepsy patients. 3
We hypothesized that providing a seizure action plan to pediatric epilepsy patients and caregivers would improve self-management, decrease the impact of epilepsy on families, and lessen the burden of health care costs through decreased health care utilization. Therefore, our primary objective was to measure the impact of a seizure action plan on patient health care utilization, including emergency department visits, urgent care visits, and outpatient service attendance in pediatric patients. Secondarily, we hypothesized that providing patients with a seizure action plan would reduce strain on caregivers and improve parents’/caregivers’ perceived impact of epilepsy on family life.
Methods
The Institutional Review Board at Nationwide Children's Hospital reviewed and approved this study. Our Institutional Review Board waived participation consent and assent requirements for this study to avoid the Hawthorne effect. 18 Subsequently, these waivers served to guard against threats to external validity. Patients and their caregivers were assigned to one of 2 groups in a prospective controlled single-blinded study.
Participant Enrollment
Participants were enrolled in our study between October 2014 and March 2015. Patients met inclusion criterion if they were (1) an established epilepsy patient in our center for at least 12 months, (2) on at least 1 anticonvulsant (not including rescue medications), and (3) between 6 months and 21 years of age. Patients were excluded if they (1) were newly diagnosed with epilepsy, (2) were actively weaning anticonvulsant medications due to seizure freedom, (3) had a diagnosis of active infantile spasms or absence epilepsy, or (4) were actively engaging with a provider in an epilepsy surgery evaluation. Patients with new-onset epilepsy were excluded because of a lack of baseline utilization data for the comparison. A research coordinator prescreened potential participants for eligibility by conducting a chart review in the electronic health record.
Subsequently, the research coordinator enrolled patients and their families in the study during a routine epilepsy outpatient clinic visit. Study staff first stratified patients by seizure frequency (more than 12 seizures per year or less than 12 seizures per year) in order to evenly distribute seizure severity between the 2 intervention groups. Then patients and their caregivers were alternatingly assigned to receive standard care with or without a seizure action plan. Standard care included providing patients and families specific seizure care instructions in a nonstandardized fashion and a prescription for abortive seizure medications, as deemed appropriate by the provider. Patients and caregivers assigned to the seizure action plan group received the seizure action plan along with an explanation of the seizure action plan. Patients and caregivers in the standard care group received instructions on emergency seizure management in a nonstandard manner as part of current clinical care practices. All caregivers across both groups were asked to complete the modified impact on family (Modified Impact on Families questionnaire) survey at study enrollment. 19 Study personnel followed up with each family, either in clinic or via a phone call, to request their completion of the Modified Impact on Families questionnaire survey again at 3 and 12 months.
Nationwide Children’s Hospital Seizure Action Plan
A physician, nurse, technology analyst, health communication expert, and pharmacist team within the Pediatric Epilepsy Center at Nationwide Children’s Hospital developed a customizable seizure action plan for seizure patients and their caregivers (Appendix A). We modeled our seizure action plan after an existing pediatric asthma action plan created and validated by Nationwide Children’s Hospital’s pulmonary and primary care clinics.
Using stop light imagery, the internally built seizure action plan outlined what epilepsy care a caregiver should provide for his or her child when seizures are under control (green zone); when there are parental concerns, such as missed doses of medications or brief seizures (yellow zone); and in case of emergencies, such as prolonged seizures or seizure clusters (red zone) (Appendix A). Our team collaborated with an information technology specialist to ensure the seizure action plan was integrated into Nationwide Children’s Hospital’s electronic health record to facilitate health record extraction, clinical use, and care continuity. For patients in the group receiving the seizure action plan, clinicians printed, provided, and then explained the seizure action plan to caregivers. This allowed for a fully customizable seizure action plan within our electronic health record, such that patients’ medications and needs were incorporated into the plan. Each epilepsy clinician reviewed and provided the patients’ seizure action plan with the patient and caregiver(s) during their regularly scheduled clinic appointment.
Primary Outcomes
We extracted health care utilization measures from patients’ electronic health record 1 year pre- and postenrollment. The following inpatient and outpatient utilization measures were obtained: presence of any emergency department visits, presence of any unplanned hospitalizations, any neurology visit no-shows, presence of any overall (all visits within the hospital system) clinical appointment no-shows (collectively referred to as “unplanned utilization”), phone calls placed to the neurology clinic, completed neuro appointments, and completed total appointments attendance rates (collectively referred to as “planned utilization”).
Secondary Outcomes
Caregivers of all patients were asked to complete the Modified Impact on Families (MIF) questionnaire at enrollment and 3 and 12 months postenrollment. The survey contained the original Impact on Families scale items, 19 as well as 5 additional items written by the research study group to gather specific epilepsy-related information (Appendix B). The Modified Impact on Families questionnaire contains 6 subscales, including total impact (20 items), financial support (3 items), general impact (10 items), family/disruption of social relations (9 items), coping (ie, mastery; 4 items), and sibling impact (6 items), all with response options on a Likert-type scale.
Seizure Frequency
Patients were grouped based on baseline seizure frequency into 2 categories: high frequency was considered greater than 12 seizures per year and low frequency was defined as fewer than 12 seizures per year. This was done for analysis in order to identify if there were differences in health care utilization based on seizure frequency alone (eg, if patients with high seizure frequency are more likely to have unplanned visits in the emergency department than those with low frequency). We also grouped by seizure frequency to determine whether the impact of a seizure action plan differs based on frequency of seizures.
Statistical Analysis
Group comparisons between the seizure action plan and standard care groups were assessed using 2-sample t tests or Wilcoxon rank sum tests for continuous data and chi-square or Fisher exact tests for categorical data. Multivariable logistic regression was used to assess whether the odds of an emergency department visit, unplanned hospitalization, and neuro clinic no show is associated with seizure action plan while adjusting for age, sex, and seizure frequency. Mixed effects models were used to evaluate longitudinal Modified Impact on Families questionnaire subscale scores. Interaction terms were used to determine whether the potential impact of seizure action plan intervention on Modified Impact on Families questionnaire differs by seizure frequency or over time; where the interaction was nonsignificant, it was removed from the model. When the interaction was significant, post hoc mean comparisons stratified by time or seizure frequency were conducted using the Scheffe adjustment for multiple comparisons. All analyses were conducted using SAS 9.4 (SAS Institute, Cary, NC) with 2-sided P values <.05 considered statistically significant.
Results
Participants
Fifty-four patients were assigned to receive a seizure action plan in addition to standard epilepsy care and 51 were assigned to receive standard of care alone. One patient in the standard of care group did not complete the initial baseline Modified Impact on Families questionnaire and thus was excluded from the study. Two patients in the standard care group were not included in analysis as they were deceased prior to study end, decreasing the number of subjects in the standard of care group to 48 patients (Figure 1). Both patients who died had severe forms of refractory epilepsy (Lennox-Gastaut in one and symptomatic epilepsy secondary to lissencephaly in the other) and both expired outside of our facility; therefore, causes of death were not available. No significant differences in patient parameters, epilepsy characteristics, or preintervention health care utilization or baseline Modified Impact on Families questionnaire scores were identified between the 2 groups (Table 1), demonstrating group equivalency. There was patient attrition over the 3 time points with patients lost to follow-up.

Flow diagram depicting enrollment and study attrition.
Baseline Characteristics of Patients.a
Abbreviations: ED, emergency department; IQR, interquartile range; MIF, Modified Impact on Families questionnaire; SAP, seizure action plan.
a There was no statistically significant difference between the SAP groups for age, gender, years since epilepsy diagnosis, insurance, and seizure frequency greater than 12 seizures per year.
Primary Outcomes-Unplanned Health Care Utilization
Fifty-one percent of patients had at least 1 unplanned emergency department visit for seizures, whereas 25% of patients had an unplanned hospitalization. In univariate assessment, presence of any emergency department visits and unplanned hospitalizations did not differ by seizure action plan group (50% seizure action plan vs 52% standard care group and 24% seizure action plan vs 27% standard care, respectively). Fewer patients in the seizure action plan group had neurology clinic no-shows (22% vs 40% for nonseizure action plan), but the difference was not statistically significant (P = .06). Significantly fewer patients in the seizure action plan group had overall clinic no-shows (55% vs 75%, P = .04) (Table 2).
Univariate Group Differences on Health Care Utilization Outcomes.a
Abbreviations: ED, emergency department; IQR, interquartile range; SAP, seizure action plan; VNS, vagus nerve implant.
aThere was a significantly higher proportion of overall clinic appointment no-shows in the No SAP vs the SAP group (P = .04); otherwise, there were no significant differences in health care utilization.
After adjusting for age, sex, and seizure frequency, results were similar; the seizure action plan was nearly significantly associated with a reduction in odds of neurology clinic no-shows (OR 0.47, 95% CI 0.19-1.15), and seizure action plan had no significant impact on emergency department visits or unplanned hospitalization. The impact of the seizure action plan on unplanned health care utilization did not differ by seizure frequency (P value for all seizure action plan × seizure frequency interactions >0.2) (Table 3).
Odds of Any ED Visit (EDvisit), Unplanned Hospitalization (Unplanhosp), Neurology Clinic No Show (NeuroNoShow), and Composite EDvisit/Unplanhosp/NeuroNoShow (ED/HOSP/NEURO)a
Abbreviations: CI, confidence interval; ED, emergency department; OR, odds ratio; SAP, seizure action plan.
aSAP use was not a significant predictor of an EDvisit, Unplanhosp, NeuroNoShow, or ED/HOSP/NEURO.
Secondary Outcomes: Planned Health Care Utilization and Modified Impact on Families Questionnaire
Overall, there were no significant differences by seizure action plan group at any time point for the seizure comfort, total impact, financial support, general impact, family disruption/social relations, or sibling domains of the Modified Impact on Families questionnaire. When analyzing the total impact domain, scores at 3 months tended to be lower in the seizure action plan group versus the standard care group (42.4 [39.1-45.8] vs 47.1 [43.3-50.9], P = .07), but did not reach statistical significance. There were no differences at baseline or 12 months. Scores on the coping domain were consistently lower over time among the seizure action plan group compared to standard of care patients (P = .04) (Figure 2).

Modified Impact on Families questionnaire scores over time by SAP groups. (SAP, seizure action plan.)
The impact of seizure action plan on Modified Impact on Families questionnaire scores depended on seizure frequency for the seizure comfort and total impact subdomains (P = .049 and P = .03, respectively). Among patients with low seizure frequency (12 or fewer seizures per year), seizure comfort scores on the Modified Impact on Families questionnaire were significantly higher at 12 months among the seizure action plan group compared to the standard care group (18.8 [17.8, 19.7] vs 17.5 [16.5, 18.4], P = .05) (Figure 3). Moreover, among patients with low seizure frequency, the total impact score was lower at 3 months among patients with a seizure action plan compared to those without a seizure action plan but not statistically significant (P = .07) (Figure 4). There were no significant differences by seizure action plan group among patients with high seizure frequency.

Seizure comfort total score over time by seizure frequency groups.

Total Impact score over time by seizure frequency groups.
Discussion
The effect of a seizure action plan on follow-up in the pediatric epilepsy clinic is notable. Those given a seizure action plan were less likely to no-show their scheduled pediatric epilepsy clinic visit. However, we were unable to show a significant difference in other unplanned utilization of health care resources among those who received a seizure action plan as part of their epilepsy care and those who did not. We found a significant improvement in seizure comfort scores in the families who received a seizure action plan. These findings are consistent with a previous study by Roundy and colleagues. 3
Better clinic attendance may be evidence of a strengthened relationship between the patient, caregivers, and the provider. Considering that epilepsy is a chronic condition with acute exacerbations, proactive surveillance, care, and planning in the clinic environment is vital to decrease morbidity and mortality associated with acute seizure events. This is especially true regarding monitoring medication adherence in the clinic as nonadherence is associated with increased risk for status epilepticus and mortality. 20 An intensive educational model for children with epilepsy studied in Argentina showed improvement on numerous measures, including decreased emergency department visits illustrating the importance of education to promote self-efficacy. 21
The 2012 Institute of Medicine (IOM) report titled “Epilepsy Across the Spectrum: Promoting Health and Understanding” recognizes improving self-management for patients with epilepsy through better education as a crucial need and identifies “self-management” as involving optimal education for caregivers. 22 The report highlights the need for a “patient-centered approach to education, provide individuals and their families with knowledge and skills that promote competency in optimal self-management, and apply to managing both epilepsy and its comorbidities.” 22 This should include basic knowledge about the patient’s seizures, daily epilepsy management, and acute treatment—all information we have included in our seizure action plan.
More than just seizures, epilepsy is a disorder that impacts all facets of a child’s life. 22 Some of the impact can be explained by understanding epilepsy as a chronic disease. For example, Hamiwka et al compared social skills in children with epilepsy to children with chronic kidney disease and healthy controls. They found no significant differences in social functioning between the 2 chronic conditions, but there were clear differences in children with epilepsy with respect to cooperation, assertiveness, and self-control as compared to the healthy controls. 23 Our results demonstrate the seizure action plan had some impact on perceived family functioning and quality of life as measured by the Modified Impact on Families questionnaire, especially in the lower seizure frequency group. Improved self-efficacy has previously been shown to have a positive impact by other authors. 1,15,16
Our study results are similar to a previous study on seizure action plans that also did not show decrease in unplanned health care utilization for those receiving a seizure action plan, 3 despite utilizing a prospective controlled and single-blinded methodology. However, the number of unplanned emergency department visits and hospitalizations was low in all patients; the range was 0 to 3 in the patients with more than 12 seizures per year and 0 to 2 in patients with fewer than 12 seizures per year. Our epilepsy center has made great efforts including numerous quality improvement initiatives to decrease unplanned health care utilization in our epilepsy population, so it is possible this is obscuring the true effect of the seizure action plan. 24 It is also possible the seizure action plan may be more beneficial in centers with higher unplanned utilization rates.
Seizure action plans are also a potentially valuable tool for schools and school nurses, who are often provided limited resources to guide management of acute seizure events. A quality improvement initiative that utilized seizure action plans in Minnesota aimed at educating school nurses on seizure management found this intervention to be “very useful” by nurses who participated. 25 In the 2012 IOM report, the importance of improving seizure recognition and first aid education given to school personnel and community members is highlighted. 22 Seizure action plans can play a key role in that education.
Limitations
Some limitations to the current study are noteworthy and provide insight into developing further related studies and/or creating a more interactive seizure action plan. First, while patients were blinded to the intervention, providers were not blinded, which may have led to treatment bias. A randomized controlled trial, with a larger sample size across multiple sites, is necessary to detect utilization differences as a result of the treatment. With regard to the creation of the seizure action plan itself, we did not directly address parental and patient literacy prior to providing the seizure action plan and acknowledge that families with low literacy may not benefit from a written document concerning medical care and education. 26 -28 We recommended families display the seizure action plan in a visible place in their home such as on the refrigerator; however, we did not assess compliance with this recommendation. Along similar lines, if the seizure action plan was discarded or lost, it will be of no benefit. Moreover, we did not assess patient and caregiver satisfaction with the seizure action plan. All patients completed the Modified Impact on Families questionnaire at the onset of the study; however, many patients failed to complete subsequent Modified Impact on Families questionnaires despite several attempts to reach them. Therefore analyses were hampered by participant attrition.
Additionally, patients in both cohorts had low rates of emergency department visits and hospitalizations indicating other possible confounding factors that keep our epilepsy patients out of the hospital and make detecting differences difficult. Patients in the group that did not receive the seizure action plan did receive some education on emergency seizure care as part of standard care which may have confounded our ability to detect differences. Future work targeting populations of high utilizers may show more benefit in relations to health care utilization reductions.
It is likely there is a specific subset of patients who would benefit greatly from having a seizure action plan. Certain patients and families, such as those who have low literacy or for whom English is a second language would likely derive minimal benefit. Our study evaluated the effect of a seizure action plan in patients with established epilepsy. It is possible that the seizure action plan may have a different impact for patients and families with newly diagnosed epilepsy, particularly in specific Modified Impact on Families questionnaire domains. Finally, our findings may not be representative of different clinical settings in other geographic locations. A multicenter study could provide insight as to the generalizability of these findings.
Conclusions
Results of our study demonstrate improved clinic appointment attendance for patients receiving a seizure action plan and improved caregiver seizure comfort. A lack of statistically significant differences in emergency department utilization and unplanned hospitalizations between those that received a seizure action plan as part of their epilepsy care was noted.
Studies aimed at identifying measures to predict which patients would benefit most from the seizure action plan are warranted. This study provides support that seizure action plans can be a beneficial component of comprehensive care in children with epilepsy and should be recommended for most pediatric epilepsy patients. The tool may be of particular use in patients with low seizure frequency. Further work addressing patient and caregiver perceptions, the role of health literacy and socioeconomic factors, and potential impact on health care utilization is still needed.
Supplemental Material
Supplemental Material, Appendix_A_SAP - Seizure Action Plans for Pediatric Patients With Epilepsy: A Randomized Controlled Trial
Supplemental Material, Appendix_A_SAP for Seizure Action Plans for Pediatric Patients With Epilepsy: A Randomized Controlled Trial by Dara V. F. Albert, Jennifer J. Moreland, Ann Salvator, Melissa Moore-Clingenpeel, Babitha Haridas, Justin W. Cole, Peter Glynn, Marci Fults, M. Zachary Dawson, Patty Moreland and Anup D. Patel in Journal of Child Neurology
Supplemental Material
Supplemental Material, Appendix_B_MIF - Seizure Action Plans for Pediatric Patients With Epilepsy: A Randomized Controlled Trial
Supplemental Material, Appendix_B_MIF for Seizure Action Plans for Pediatric Patients With Epilepsy: A Randomized Controlled Trial by Dara V. F. Albert, Jennifer J. Moreland, Ann Salvator, Melissa Moore-Clingenpeel, Babitha Haridas, Justin W. Cole, Peter Glynn, Marci Fults, M. Zachary Dawson, Patty Moreland and Anup D. Patel in Journal of Child Neurology
Footnotes
Author Contributions
DVFA helped to conceive the idea, design the study, participated in data collection, drafting the manuscript as well as editing and approval of the final draft. JJM participated in the analysis of the data as well as review and edit of the manuscript and approval of the final draft. MMC participated in the analysis of the data as well as review and edit of the manuscript and approval of the final draft. AS participated in the analysis of the data as well as review and edit of the manuscript and approval of the final draft. BH participated in the participant enrollment, collection of the data as well as review and editing of the manuscript and approval of the final draft. PG participated in the participant enrollment, collection of the data as well as review and editing of the manuscript and approval of the final draft. MF participated in the participant enrollment, collection of the data as well as review and editing of the manuscript and approval of the final draft. MZD participated in the participant enrollment, collection of the data as well as review and editing of the manuscript and approval of the final draft. PM participating in the initial design of the Seizure Action Plan as well as review and editing of the manuscript and approval of the final draft. ADP helped to conceive the idea, design the study, participated in data collection, drafting the manuscript as well as editing and approval of the final draft. JWC helped to conceive the idea, design the study, participated in editing the manuscript and approval of the final draft.
Declaration of Conflicting Interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.
Supplemental Material
Supplemental material for this article is available online.
Ethical Approval
The Institutional Review Board at Nationwide Children’s Hospital reviewed and approved this study.
References
Supplementary Material
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