Epilepsy-specific Health-related Quality Of Life Research Articles

The eACT study design and methods: A sequential, multiple assignment, randomized trial of A novel adherence intervention for youth with epilepsy

BackgroundEpilepsy is a common, chronic pediatric neurological condition predominately treated with anti-seizure medications (ASMs) to control or reduce seizures. Approximately 60 % of youth with epilepsy demonstrate suboptimal adherence to their ASM. This paper describes the methodology, recruitment, design, and baseline participant characteristics of a sequential, multiple assignment, randomized trial (SMART) designed to test the effectiveness of a behavioral health intervention to improve adherence in families of young children with epilepsy. MethodsUsing a two-stage SMART, youth ages 2–12 years old with newly diagnosed epilepsy and their families were enrolled. Following an 8-week run-in phase, families with ≤95 % adherence were randomized to control (education + automated digital reminders) or intervention (education + automated digital reminders + individualized feedback) arms. After three months, families in the intervention group who remained non-adherent (i.e., ≤ 95 %) were re-randomized to 1) continue with same intervention or 2) receive two telehealth problem-solving sessions with an interventionist over the next two months. Study measures were completed at baseline, 8-, 14-, and 20-months post-baseline. ResultsOf the n = 466 ethnically and racially diverse study participants, n = 268 participants were non-adherent and were randomized. The primary outcome was electronically monitored ASM adherence at post-intervention, while secondary outcomes included seizure freedom, health care utilization, and epilepsy-specific health-related quality of life. ConclusionsNovel aspects of the trial design (e.g., sequential, recruitment of racial and ethnic diverse youth), modifications to the protocol related to the COVID-19 pandemic and evolving socio-political and medical climate, as well as recruitment and retention challenges are discussed.ClinicalTrials.gov Number: NCT03817229

Key predictors of epilepsy-specific health-related quality of life (HRQOL) in youth with epilepsy

PurposeEpilepsy-specific health-related quality of life (HRQOL) is an important outcome in youth with epilepsy (YWE). The PedsQL™ Epilepsy Module is the only caregiver-proxy and youth self-report epilepsy-specific HRQOL measure that can be used with youth 2–25 years. Multiple factors affect HRQOL, including epilepsy-specific characteristics, comorbid mental and behavioral health concerns, as well as sociodemographic factors. However, we have not yet examined the cumulative impact of these factors on epilepsy-specific HRQOL in YWE using the PedsQL™ Epilepsy module. MethodYouth with epilepsy (n = 281) and their caregivers completed questionnaires focused on sociodemographic factors (e.g., youth biological sex and age), mood/anxiety and behavior symptoms (i.e., Behavioral Assessment Scale for Children – Second Edition; BASC-2, Parent Rating Scale), epilepsy characteristics [e.g., seizure frequency, number of anti-seizure medications (ASMs), ASM side effects, and years since diagnosis], and the PedsQL™ Epilepsy module (subscales: Impact, Cognitive Functioning, Executive Functioning, Sleep, and Mood/Behavior). ResultsHierarchical linear regressions were conducted to examine caregiver-proxy and youth self-reported factors that affect epilepsy-specific HRQOL. Results indicate the strongest key shared predictors of HRQOL in YWE, for both youth and caregiver informants, were mental and behavioral health symptoms. For instance, caregiver-proxy report of YWE HRQOL indicated BASC-2 Externalizing (p < 0.05), Behavioral Symptoms (p < 0.01), and Adaptive Skills (p < 0.001) explained 58 % of the variance in youth Cognitive Functioning HRQOL, while youth self-report of HRQOL indicated that BASC-2 Externalizing (p < 0.01), Behavioral Symptoms (p < 0.05), and Adaptive Skills (p < 0.001) contributed only 36 % of the variance in Cognitive Functioning HRQOL above and beyond the variance explained by sociodemographic and epilepsy-specific characteristics. Similar results were noted for Executive Functioning HRQOL domain, wherein caregiver-proxy report of YWE HRQOL indicated BASC-2 Internalizing (p < 0.01), Behavioral Symptoms (p < 0.001) and Adaptive Skills (p < 0.001) explained 65 % of variance in Executive Functioning, whereas youth self-report of Executive Functioning HRQOL indicated that caregiver-proxy BASC-2 Internalizing (p < 0.001) and Behavioral Symptoms (p < 0.01) explained 34 % of the variance in Executive Functioning HRQOL, above and beyond the variance explained by sociodemographic and epilepsy-specific characteristics. Unique mental and behavioral health predictors of YWE HRQOL were also found for both caregiver-proxy and youth self-report. ConclusionsGiven the integral role of mental and behavioral health symptoms in epilepsy-specific HRQOL, it is critical to address mental and behavioral health symptoms preventatively and proactively to provide YWE with the most optimal health plan, including good seizure control, minimal ASM side effects, and the best possible HRQOL.

Self-reported quality of life and degree of youth-parent agreement: A long-term follow-up of childhood-onset epilepsy.

To prospectively delineate self-reported health-related quality of life (HRQOL) of adolescents and young adults (AYAs) 8 and 10 years after an epilepsy diagnosis and evaluate the degree of AYA-parent agreement in ratings of AYA's HRQOL. Data came from the Health-Related Quality of Life in Children with Epilepsy Study (HERQULES), a 10-year longitudinal study of children, aged 4-12years, with newly diagnosed epilepsy. Epilepsy-specific HRQOL was self-reported by AYA 8 and 10 years after diagnosis and by parents at multiple time points throughout the 10-year follow-up. Measurers of HRQOL over time were analyzed using a linear mixed-effect model approach. AYA-parent agreement was evaluated using intraclass correlation coefficient (ICC) and Bland-Altman plots. A total of 165 AYAs participated at long-term follow-up. There was considerable heterogeneity among AYA's HRQOL, and as a group, there was no significant change in HRQOL from the 8- to 10-year follow-up. Household income at the time of diagnosis, seizure control at follow-up, and a history of emotional problems (anxiety/depression) were independent predictors of HRQOL at follow-up. AYA-parent agreement on AYA's HRQOL was moderate (ICC 0.62, 95% CI 0.51-0.71), although considerable differences were observed at the individual level. AYA-parent agreement varied with AYA's and parent's age, seizure control, and family environment. In the long-term after a diagnosis of epilepsy, AYAs report stable HRQOL over time at the group level, although notable individual differences exist. Seizure control, anxiety/depression, and family environment meaningfully impact AYA's long-term HRQOL. AYA and parent reports on HRQOL are similar at the group level, although they cannot be used interchangeably, given the large individual differences observed.

Validation of the PedsQL Epilepsy Module: A pediatric epilepsy-specific health-related quality of life measure.

To validate a brief and reliable epilepsy-specific, health-related quality of life (HRQOL) measure in children with various seizure types, treatments, and demographic characteristics. This national validation study was conducted across five epilepsy centers in the United States. Youth 5-18 years and caregivers of youth 2-18 years diagnosed with epilepsy completed the PedsQL Epilepsy Module and additional questionnaires to establish reliability and validity of the epilepsy-specific HRQOL instrument. Demographic and medical data were collected through chart reviews. Factor analysis was conducted, and internal consistency (Cronbach's alphas), test-retest reliability, and construct validity were assessed. Questionnaires were analyzed from 430 children with epilepsy (Mage = 9.9 years; range 2-18 years; 46% female; 62% white: non-Hispanic; 76% monotherapy, 54% active seizures) and their caregivers. The final PedsQL Epilepsy Module is a 29-item measure with five subscales (i.e., Impact, Cognitive, Sleep, Executive Functioning, and Mood/Behavior) with parallel child and caregiver reports. Internal consistency coefficients ranged from 0.70-0.94. Construct validity and convergence was demonstrated in several ways, including strong relationships with seizure outcomes, antiepileptic drug (AED) side effects, and well-established measures of executive, cognitive, and emotional/behavioral functioning. The PedsQL Epilepsy Module is a reliable measure of HRQOL with strong evidence of its validity across the epilepsy spectrum in both clinical and research settings.

Development of the PedsQL™ Epilepsy Module: Focus group and cognitive interviews.

Youth with epilepsy have impaired health-related quality of life (HRQOL). Existing epilepsy-specific HRQOL measures are limited by not having parallel self- and parent-proxy versions, having a restricted age range, not being inclusive of children with developmental disabilities, or being too lengthy for use in a clinical setting. Generic HRQOL measures do not adequately capture the idiosyncrasies of epilepsy. The purpose of the present study was to develop items and content validity for the PedsQL™ Epilepsy Module. An iterative qualitative process of conducting focus group interviews with families of children with epilepsy, obtaining expert input, and conducting cognitive interviews and debriefing was utilized to develop empirically derived content for the instrument. Eleven health providers with expertise in pediatric epilepsy from across the country provided feedback on the conceptual model and content, including epileptologists, nurse practitioners, social workers, and psychologists. Ten pediatric patients (age 4-16years) with a diagnosis of epilepsy and 11 parents participated in focus groups. Thirteen pediatric patients (age 5-17years) and 17 parents participated in cognitive interviews. Focus groups, expert input, and cognitive debriefing resulted in 6 final domains including restrictions, seizure management, cognitive/executive functioning, social, sleep/fatigue, and mood/behavior. Patient self-report versions ranged from 30 to 33 items and parent proxy-report versions ranged from 26 to 33 items, with the toddler and young child versions having fewer items. Standardized qualitative methodology was employed to develop the items and content for the novel PedsQL™ Epilepsy Module. The PedsQL™ Epilepsy Module has the potential to enhance clinical decision-making in pediatric epilepsy by capturing and monitoring important patient-identified contributors to HRQOL.

Predictors of trajectories of epilepsy-specific quality of life among children newly diagnosed with epilepsy.

The objective of this study was to identify two-year trajectories of epilepsy-specific health-related quality of life (HRQOL) among children newly diagnosed with epilepsy and to evaluate the predictive value of a comprehensive set of medical, psychosocial, and family factors. Ninety-four children with epilepsy (8.14 ± 2.37 years of age and 63% male) and their caregivers participated in this study. Caregivers completed the Quality of Life in Childhood Epilepsy Questionnaire (QOLCE) and measures of psychological and family functioning at one month postdiagnosis. The QOLCE was also given at eight additional time points during the subsequent two years as a part of a large observational study in children with epilepsy. Adherence data were collected via MEMS TrackCaps, and medical information was collected through chart review. Unique trajectories were identified for the overall QOLCE scale, as well as the subscales. Most trajectory models for the QOLCE subscales contained at least one at-risk trajectory for children, indicating that there is a subgroup of children experiencing poor long-term HRQOL. Health-related quality-of-life trajectories remained predominantly stable during the two-year period following treatment initiation. The number of AEDs, internalizing problems, and externalizing problems emerged as the most consistent predictors across the HRQOL domains. Medical and psychosocial interventions, such as cognitive-behavioral strategies, should target modifiable factors (e.g., internalizing symptoms, externalizing symptoms, number of AEDs trialed) shortly after diagnosis to improve HRQOL for children with epilepsy over the course of their disease.

Parent and family stress factors predict health‐related quality in pediatric patients with new‐onset epilepsy

To examine the influence of parent and family general and epilepsy-related stress on longitudinal generic and epilepsy-specific health-related quality of life (HRQOL) for children with new-onset epilepsy, while controlling for demographic characteristics, disease factors, and antiepileptic drug (AED) adherence. This prospective, longitudinal study included 124 children with new-onset epilepsy (mean age 7.2 years, standard deviation [SD] 2.9 years). Parents completed questionnaires on parenting stress, perceived stigma, fears and concerns, and HRQOL at 1, 13, and 25 months after diagnosis. Adherence to AEDs was assessed using electronic monitors. A medical chart review was conducted at each visit to obtain seizure and side effect data. Higher levels of general and epilepsy-specific parent and family stress, fears and concerns, and perceived stigma negatively affected child generic and epilepsy-specific HRQOL, above and beyond disease and demographic factors. General parenting and family stress affected child generic and epilepsy-specific HRQOL more in the first year of disease management than at 2 years after diagnosis. Higher fears and concerns predicted higher epilepsy-specific HRQOL at 13 months postdiagnosis, whereas 2 years postdiagnosis, higher fears and concerns predicted lower epilepsy-specific HRQOL. Several demographic (i.e., age) and disease-related variables (i.e., side effects and AED adherence) influenced child generic and epilepsy-specific HRQOL. Although some findings were consistent across generic and epilepsy-specific HRQOL measures, others were unique. Modifiable parent factors (i.e., general and disease-specific parent and family stress, perceived stigma) impact HRQOL for children with new-onset epilepsy differently over the first 2 years postdiagnosis. Psychosocial interventions to improve HRQOL within the first year postdiagnosis should address parenting and family stress, overall coping, and anticipatory guidance on managing epilepsy. Interventions targeting adherence, perceived stigma, and fears and concerns could improve HRQOL. Promoting parent management of stress, fears/concerns, and perceived stigma may lead to improved child HRQOL outcomes. A PowerPoint slide summarizing this article is available for download in the Supporting Information section here.

Quality of life in a cohort of men with epilepsy compared to a healthy population and those with common chronic diseases in the UK using a generic patient-reported outcome measure

Patient-reported outcome measures (PROMs) are increasingly used in epilepsy. Epilepsy-specific instruments enable clinicians to gain insight into patients' health-related quality of life (HRQoL) but do not allow comparison between conditions and do not reflect subjective well-being (SWB). Using the World Health Organization Quality-of-Life Questionnaire — Brief (WHOQOL-Bref), a short generic PROM, we compared the HRQoL in a cohort of men with epilepsy (MWE) recruited from the epilepsy clinic and via the website of a large UK epilepsy charity, with seven other groups with chronic diseases. Multiple linear regression showed that mood was the most important independent predictor of the WHOQOL-Bref score. The sample, however, rated their global quality of life as highly as the UK control group, and 38% reported life ‘very’ or ‘extremely’ meaningful, and 4% enjoyed life ‘very much’ or ‘extremely’. Because of its structure, the WHOQOL-Bref gives clinicians an indication not only of HRQoL but also of SWB, a broader construct. Our study suggests that the narrow focus of epilepsy-specific HRQoL questionnaires may give only a partial picture of a patient's quality of life. In addition, by concentrating on the negative aspects of life with epilepsy, these instruments may distract both the patient and the clinician from what is good about life, denying the patient the benefits of ‘positive psychology’ and the clinician the opportunity to build the patient's resilience.

Reliability and validity of a Spanish version of the Impact of Pediatric Epilepsy Scale in a Cuban population

The Impact of Pediatric Epilepsy Scale (IPES) is a brief, accurate, and acceptable measurement scale of the impact of pediatric epilepsy on the health-related quality of life (HRQOL) of both the child and the child's family as perceived by the child's parent(s). The aim of this study was to validate a Spanish language version of the IPES in Cuban children with epilepsy. The IPES was translated and adapted to Cuban culture and administered to 76 parents of children with epilepsy. The principal component analysis indicated that two factors accounted for 72% of the variance of the IPES (family relationships and health and social well-being). The IPES was also able to detect differences in HRQOL between subjects according to epilepsy severity. The internal consistency coefficient was 0.962, and the test–retest reliability was 0.979. The Cuban version of IPES can be used to measure a child's epilepsy-specific HRQOL in Cuba.

Reliability and validity of a Chinese version of the Impact of Pediatric Epilepsy Scale

The Impact of Pediatric Epilepsy Scale (IPES) is an accurate, acceptable, and quick tool that assesses the impact of epilepsy on the child with epilepsy and his or her family. The aim of this study was to investigate its applicability in China. After multistage translation and cultural adaptation, the final Chinese version was administered to 110 parents of children with epilepsy to evaluate its validity, reliability, and sensitivity. All items contributed significantly to the summary measure. With respect to validity, all items were substantially correlated with the criterion questionnaire subscales, and principal component analysis indicated that three factors accounted for 72% of the variance of the scale. The internal consistency coefficients of the first and second tests for the total were 0.916 and 0.930, respectively, and test–retest reliability ranged from 0.891 to 0.992. Additionally, the IPES can detect differences in health-related quality of life (HRQOL) between subjects according to epilepsy severity. In conclusion, this study indicates that the Chinese IPES has good validity, reliability, and sensitivity, and is an epilepsy-specific HRQOL questionnaire that is a brief, accurate, and valid assessment of the influence of epilepsy on the child and family.

Evaluation of the responsiveness of the Impact of Pediatric Epilepsy Scale

The Impact of Pediatric Epilepsy Scale (IPES) is an epilepsy-specific, health-related quality of life (HRQOL) questionnaire that is brief, accurate, and valid and assesses the influence of epilepsy on the child and family. This study prospectively investigates the responsiveness of the IPES, 3 years after the IPES questionnaire was initially validated. The same parents or caregivers completed the IPES. Changes in IPES scores were compared with changes in epilepsy severity. IPES Total scores were highly related to change in seizure severity (Wilks' lambda F (3, 11)=5.0, P<0.001). Those whose seizure severity decreased had decreased IPES scores (indicating improvement of HRQOL); those with increased seizure severity had increased IPES scores and reported poorer HRQOL; and those with no change in seizure severity had no change in the IPES score and no change in HRQOL. In conclusion, the IPES is useful for measurement of a child's epilepsy-specific HRQOL during treatment and is responsive to changes in epilepsy severity over time.

Health-related quality of life in childhood epilepsy: Moving beyond 'seizure control with minimal adverse effects'

Childhood epilepsy is one of the most important and prevalent neurological conditions in the developing years. Persons with childhood onset epilepsy are at a high risk for poor psychosocial outcomes, even without experiencing co-morbidities. The goal of management of children with epilepsy should be to enable the child and the family to lead a life as free as possible from the medical and psychosocial complications of epilepsy. This comprehensive care needs to go beyond simply trying to control seizures with minimal adverse drug reactions. Seizure frequency and severity is only one important outcome variable. Other factors such as social, psychological, behavioural, educational, and cultural dimensions of their lives affect children with epilepsy, their families and their close social networks.A number of epilepsy-specific health-related quality of life (HRQL) scales for children have been developed with the aim to include and measure accurately the impact and burden of epilepsy. Their target populations, details of the origin of the items, and psychometric properties vary significantly. Their strengths and weaknesses will be identified more clearly through their continued use in the clinical setting and in research studies. Only a few studies to date have utilized these or generic HRQL measures to assess the HRQL of specific populations with epilepsy.Future research needs to develop theory driven models of HRQL and identify measurable factors that have important correlations with outcomes. Since biomedical variables like seizure frequency and severity have only moderate correlations with HRQL, other independent factors including the child's resilience, co-morbid conditions, parental well-being, family factors and societal/cultural variables may play a major role. We also need to learn what encompasses comprehensive patient care, define the goals of management and evaluate the impact of different interventions. Future studies need to include the children's own perspectives of their HRQL in addition to parental reports.Finally, clinicians need to familiarize themselves with outcome measures, be able to evaluate them, and use them routinely in their day-to-day practice.

Validation of a new quality of life measure for children with epilepsy.

There is no adequate measure of health-related quality of life (HRQOL) specifically for children with epilepsy. The aim of this study was to develop an epilepsy-specific HRQOL questionnaire for children, covering five domains: physical function, emotional well-being, cognitive function, social function, and behavior. Second, we aimed to demonstrate the instrument's reliability and validity, and its sensitivity to differences in epilepsy severity. The subjects were guardians of children with refractory epilepsy, whose syndrome had been defined by using video-EEG monitoring. Each family completed the developed epilepsy-specific HRQOL scale for children and two standard, generic measures of HRQOL. The results indicated that each of the scales of the questionnaire had good internal consistency reliability. Furthermore, each scale correlated more highly with theoretically similar scales on established, generic health measures than with theoretically dissimilar scales (construct validity). The sensitivity of the questionnaire to differences in epilepsy severity also was demonstrated. As seizure severity increased, HRQOL subscale scores decreased, independent of age, gender, age of seizure onset, and IQ. Further, there was a negative relation between the number of antiepileptic medications taken and measures of memory and language performance, which was independent of age, gender, age of seizure onset, IQ, and seizure severity. This study demonstrated that the developed HRQOL instrument is a reliable and valid measure and is sensitive to differences in epilepsy. These results indicate that this new instrument may be a viable medical or surgical outcome measure for children with epilepsy.