Establishing the minimum clinically important difference of the Quality of Life in Childhood Epilepsy Questionnaire.

OBJECTIVE: To estimate the minimum clinically important difference (MCID) for the parent-reported 55-item Quality of Life in Childhood Epilepsy Questionnaire (QOLCE-55) and its shortened 16-item version, QOLCE-16. METHODS: Data came from 74 children with epilepsy (CWE) (ages 4-10, mean age = 8 [SD = 1.8]) enrolled in the Making Mindfulness Matter in Epilepsy (M3-E) trial, a pilot, parallel randomized-controlled trial of a mindfulness-based intervention. Both anchor-based and distribution-based methods were used to estimate MCID values for the QOLCE-55 and QOLCE-16. For the anchor-based approach, the Patient Centered Global Ratings of Change (PCGRC) scale and linear regression analysis were used to estimate the MCID. For the distribution-based approach, .5 SD of the health-related quality of life (HRQOL) change score distribution was used to estimate the MCID. RESULTS: For the QOLCE-55, the MCID obtained using an anchor-based approach was 10 points and using a distribution-based method was 6 points. For the QOLCE-16, the MCID obtained using an anchor-based method was 13 points and using a distribution-based method was 7 points. SIGNIFICANCE: This is the first study to estimate MCID values for the QOLCE-55 and the QOLCE-16. It has been well documented that CWE are at risk of experiencing psychological, behavioral, and cognitive impairments, which can negatively impact their HRQOL. Reporting MCID values for the QOLCE-55 and QOLCE-16 is important in determining whether changes in HRQOL observed are meaningful to CWE themselves, as a key factor in shaping the nature of epilepsy care delivered.

Association of outdoor air and noise pollution with unprovoked seizures and new onset epilepsy: A systematic review and meta-analysis.

Research has indicated that certain environmental exposures may increase the risk of unprovoked seizures and new onset epilepsy. This study aimed to synthesize the literature that has estimated the associations between short- and long-term exposure to outdoor air and noise pollution and the risk of unprovoked seizures and new onset epilepsy. We searched Embase, MEDLINE, Scopus, Web of Science, BIOSIS Previews, Latin American and Caribbean Health Sciences Literature, Proquest Dissertations and Theses, conference abstracts, and the gray literature and conducted citation tracing in June 2023. Observational and ecological studies assessing the associations of air and noise pollution with unprovoked seizures or new onset epilepsy were eligible. One reviewer extracted summary data. Using fixed and random effects models, we calculated the pooled risk ratios (RRs) for the studies assessing the associations between short-term exposure to air pollution and unprovoked seizures. Seventeen studies were included, 16 assessing the association of air pollution with seizures and one with epilepsy. Eight studies were pooled quantitatively. Ozone (O3; RR = .99, 95% confidence interval [CI] = .99-.99) and nitrogen dioxide (NO2) exposure adjusted for particulate matter (RR = 1.02, 95% CI = 1.01-1.02) on the same day, and carbon monoxide (CO) exposure 2 days prior (RR = 1.12, 95% CI = 1.02-1.22), were associated with seizure risk. A single study of air pollution and epilepsy did not report a significant association. The risk of bias and heterogeneity across studies was moderate or high. Short-term exposure to O3, NO2, and CO may affect the risk of seizures; however, the effect estimates for O3 and NO2 were minimal. Additional research should continue to explore these and the associations between outdoor air pollution and epilepsy and between noise pollution and seizures and epilepsy.

Family-centred care interventions for children with chronic conditions: A scoping review.

INTRODUCTION: Children with chronic conditions have greater health care needs than the general paediatric population but may not receive care that centres their needs and preferences as identified by their families. Clinicians and researchers are interested in developing interventions to improve family-centred care need information about the characteristics of existing interventions, their development and the domains of family-centred care that they address. We conducted a scoping review that aimed to identify and characterize recent family-centred interventions designed to improve experiences with care for children with chronic conditions. METHODS: We searched Medline, Embase, PsycInfo and Cochrane databases, and grey literature sources for relevant articles or documents published between 1 January 2019 and 11 August 2020 (databases) or 7-20 October 2020 (grey literature). Primary studies with ≥10 participants, clinical practice guidelines and theoretical articles describing family-centred interventions that aimed to improve experiences with care for children with chronic conditions were eligible. Following citation and full-text screening by two reviewers working independently, we charted data covering study characteristics and interventions from eligible reports and synthesized interventions by domains of family-centred care. RESULTS: Our search identified 2882 citations, from which 63 articles describing 61 unique interventions met the eligibility criteria and were included in this review. The most common study designs were quasiexperimental studies (n = 18), randomized controlled trials (n = 11) and qualitative and mixed-methods studies (n = 9 each). The most frequently addressed domains of family-centred care were communication and information provision (n = 45), family involvement in care (n = 37) and access to care (n = 30). CONCLUSION: This review, which identified 61 unique interventions aimed at improving family-centred care for children with chronic conditions across a range of settings, is a concrete resource for researchers, health care providers and administrators interested in improving care for this high-needs population. PATIENT OR PUBLIC CONTRIBUTION: This study was co-developed with three patient partner co-investigators, all of whom are individuals with lived experiences of rare chronic diseases as parents and/or patients and have prior experience in patient engagement in research (I. J., N. P., M. S.). These patient partner co-investigators contributed to this study at all stages, from conceptualization to dissemination.

Trajectories of parent well-being in children with drug-resistant epilepsy.

OBJECTIVE: This longitudinal cohort study aimed to identify trajectories of parent well-being over the first 2 years after their child's evaluation for candidacy for epilepsy surgery, and to identify the baseline clinical and demographic characteristics associated with these trajectories. Parent well-being was based on parent depressive and anxiety symptoms and family resources (i.e., family mastery and social support). METHODS: Parents of 259 children with drug-resistant epilepsy (105 of whom eventually had surgery) were recruited from eight epilepsy centers across Canada at the time of their evaluation for epilepsy surgery candidacy. Participants were assessed at baseline and 6-month, 1-year, and 2-year follow-up. The trajectories of parents' depressive symptoms, anxiety symptoms, and family resources were jointly estimated using multigroup latent class growth models. RESULTS: The analyses identified three trajectories: an optimal-stable group with no/minimal depressive or anxiety symptoms, and high family resources that remained stable over time; a mild-decreasing-plateau group with mild depressive and anxiety symptoms that decreased over time then plateaued, and intermediate family resources that remained stable; and a moderate-decreasing group with moderate depressive and anxiety symptoms that decreased slightly, and low family resources that remained stable over time. Parents of children with higher health-related quality of life, fathers, and parents who had higher household income were more likely to have better trajectories of well-being. Treatment type was not associated with the trajectory groups, but parents whose children were seizure-free at the time of the last follow-up were more likely to have better trajectories (optimal-stable or mild-decreasing-plateau trajectories). SIGNIFICANCE: This study documented distinct trajectories of parent well-being, from the time of the child's evaluation for epilepsy surgery. Parents who present with anxiety and depressive symptoms and low family resources do not do well over time. They should be identified and offered supportive services early in their child's epilepsy treatment history.

A longitudinal cohort study of mediators of health-related quality of life after pediatric epilepsy surgery or medical treatment.

OBJECTIVES: The purpose of this longitudinal cohort study was to examine the variables that influence health-related quality of life (HRQOL) after epilepsy surgery in children. We examined whether treatment type (surgical vs medical therapy) and seizure control are related to other variables that have been shown to influence HRQOL, namely depressive symptoms in children with epilepsy or their parents, and the availability of family resources. METHODS: In total, 265 children with drug-resistant epilepsy were recruited from eight epilepsy centers across Canada at the time of their evaluation for candidacy for epilepsy surgery and were assessed at baseline, 6-month, 1-year, and 2-year follow-up. Parents completed the Quality of Life in Childhood Epilepsy Questionnaire (QOLCE-55) and measures of family resources and depression; children completed depression inventories. Causal mediation analyses using natural effect models were used to evaluate the extent to which the relationship between treatment and HRQOL was explained by seizure control, child and parent depressive symptoms, and family resources. RESULTS: Overall, 111 children underwent surgery and 154 were treated with medical therapy only. The HRQOL scores of surgical patients were 3.4 points higher (95% confidence interval [CI]: -0.2, 7.0) relative to medical patients at the 2-year follow-up after adjusting for baseline covariates, with 66% of the effect of surgery attributed to seizure control. Child or parent depressive symptoms and family resources had negligible mediation effects between treatment and HRQOL. The effect of seizure control on HRQOL was not mediated by child or parent depressive symptoms, or by family resources. SIGNIFICANCE: The findings demonstrate that seizure control is on the causal pathway between epilepsy surgery and improved HRQOL in children with drug-resistant epilepsy. However, child and parent depressive symptoms and family resources were not significant mediators. The results highlight the importance of achieving seizure control to improve HRQOL.

Ethical and practical considerations related to data sharing when collecting patient-reported outcomes in care-based child health research.

PURPOSE: The collection and use of patient reported outcomes (PROs) in care-based child health research raises challenging ethical and logistical questions. This paper offers an analysis of two questions related to PROs in child health research: (1) Is it ethically obligatory, desirable or preferable to share PRO data collected for research with children, families, and health care providers? And if so, (2) What are the characteristics of a model best suited to guide the collection, monitoring, and sharing of these data? METHODS: A multidisciplinary team of researchers, providers, patient and family partners, and ethicists examined the literature and identified a need for focus on PRO sharing in pediatric care-based research. We constructed and analyzed three models for managing pediatric PRO data in care-based research, drawing on ethical principles, logistics, and opportunities to engage with children and families. RESULTS: We argue that it is preferable to share pediatric PRO data with providers, but to manage expectations and balance the risks and benefits of research, this requires a justifiable data sharing model. We argue that a successful PRO data sharing model will allow children and families to have access to and control over their own PRO data and be engaged in decision-making around how PROs collected for research may be integrated into care, but require support from providers. CONCLUSION: We propose a PRO data sharing model that can be used across diverse research settings and contributes to improved transparency, communication, and patient-centered care and research.

Psychometric properties of an Arabic translation of the quality of life in childhood epilepsy questionnaire (QOLCE-55).

OBJECTIVE: To develop an Arabic translation of the Quality of Life in Children with Epilepsy-55 questionnaire (QOLCE-55), and to assess its validity and reliability to be readily used in Arabic and Egyptian cultures. SUBJECTS AND METHODS: The original English version of the QOLCE-55 was translated into Arabic using a forward-backward translation method, and then a cross-sectional survey was conducted including 100 children with epilepsy aged 4-18 years. Caregivers of children completed the Arabic version of the QOLCE-55. Assessment of psychometric properties of the translated questionnaire was conducted using test-retest reliability, internal consistency, and convergent and divergent validity. RESULTS: The translated questionnaire showed excellent test-retest reliability with the intra-class correlation coefficient for all questionnaire domains, as well as the overall questionnaire ranging from 0.91 to 0.98. Cronbach alpha exceeded 0.7 denoting good internal consistency except for the emotional functioning scale. Convergent and divergent validity assessment showed that items of all domains significantly correlated with their scale scores with r > 0.4 and these correlations were much higher than correlations with other scales' scores, consistent with good convergent and divergent validity. The mean total HRQOL score was 65.63 ±â€¯8.79 with the highest score for social functioning domain and lowest score for physical functioning domain. CONCLUSION: The Arabic version of the QOLCE-55 can be considered a suitable, reliable, and valid tool to assess the HRQOL of children with epilepsy through their caregivers' reports.

Experiences and Stressors of Parents of Trans and Gender-Diverse Youth in Clinical Care from Trans Youth CAN!

Parents of trans and gender-diverse youth can experience challenges navigating gender-affirming (GA) care such as stigma, transphobia, and lack of support. There is little information available about stressors, worries, and positive feelings of parents as they try to support their youth accessing GA care. This article presents baseline survey data on experiences and stressors of 160 parents/caregivers in the Trans Youth CAN! cohort study, which examined medical, social, and family outcomes in youth age 16 years or younger considering puberty blockers or GA hormones. Data were collected at 10 Canadian gender clinics. Authors report on participating parents' characteristics, levels of support toward youth, stressors, worries, concerns, and positive feelings related to youth's gender. Most parent participants were White (85.1 percent), female (85.1 percent), birth or adoptive parents (96.1 percent), and reported strong support for youth's gender. Participants' concerns included their youth facing rejection (81.9 percent), generalized transphobia (74.6 percent), or encountering violence (76.4 percent). Parents also reported positive feelings about seeing their youth grow more confident. Most parental worries and stressors were situated outside the family, reflecting the systemic discrimination faced by youth and their families. Social workers could address these by developing systems-focused interventions and by further taking into account intersectional health disparities.

Longitudinal changes in emotional functioning following pediatric resective epilepsy surgery: 2-Year follow-up.

OBJECTIVE: To examine longitudinal changes and predictors of depression and anxiety 2 years following resective epilepsy surgery, compared to no surgery, in children with drug-resistant epilepsy (DRE). METHOD: This multicenter cohort study involved 128 children and adolescents with DRE (48 surgical, 80 nonsurgical; 8-18 years) who completed self-report measures of depression and anxiety at baseline and follow-up (6-month, 1-year, 2-year). Child demographic (age, sex, IQ) and seizure (age at onset, duration, frequency, site and side) variables were collected. RESULTS: Linear mixed-effects models controlling for age at enrolment found a time by treatment by seizure outcome interaction for depression. A negative linear trend across time (reduction in symptoms) was found for surgical patients, irrespective of seizure outcome. In contrast, the linear trend differed depending on seizure outcome in nonsurgical patients; a negative trend was found for those with continued seizures, whereas a positive trend (increase in symptoms) was found for those who achieved seizure freedom. Only a main effect of time was found for anxiety indicating a reduction in symptoms across patient groups. Multivariate regressions failed to find baseline predictors of depression or anxiety at 2-year follow-up in surgical patients. Older age, not baseline anxiety or depression, predicted greater symptoms of anxiety and depression at 2-year follow-up in nonsurgical patients. CONCLUSION: Children with DRE reported improvement in anxiety and depression, irrespective of whether they achieve seizure control, across the 2 years following surgery. In contrast, children with DRE who did not undergo surgery, but achieved seizure freedom, reported worsening of depressive symptoms, which may indicate difficulty adjusting to life without seizures and highlight the potential need for ongoing medical and psychosocial follow-up and support.

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

OBJECTIVE: 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. METHODS: Data came from the Health-Related Quality of Life in Children with Epilepsy Study (HERQULES), a 10-year longitudinal study of children, aged 4-12 years, 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. RESULTS: 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. SIGNIFICANCE: 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.

Trajectories of quality of life 10 years following a diagnosis of epilepsy in childhood.

OBJECTIVE: This study estimated trajectories of health-related quality of life (HRQOL) over a 10-year period among children newly diagnosed with epilepsy. We also modeled the characteristics of children, parents, and families associated with each identified trajectory. METHODS: Data came from the HERQULES (Health-Related Quality of Life in Children With Epilepsy Study), a Canada-wide prospective cohort study of children (aged 4-12 years) with newly diagnosed epilepsy. Parents reported on their children's HRQOL at diagnosis, and at 0.5-, 1-, 2-, 8-, and 10-year follow-ups using the Quality of Life in Childhood Epilepsy Questionnaire-55. Trajectories of HRQOL were identified using latent class growth models. Characteristics of children, parents, and families at the time of diagnosis that were associated with each trajectory were identified using multinomial logistic regression. RESULTS: A total of 367 children were included. Four unique HRQOL trajectories were identified; 11% of the cohort was characterized by low and stable scores, 18% by intermediate and stable scores, 35% by intermediate scores that increased then plateaued, and 43% by high scores that increased then plateaued. Absence of comorbidities, less severe epilepsy, and better family environment (greater satisfaction with family relationships and fewer family demands) at the time of diagnosis were associated with better long-term HRQOL trajectories. Although the analyses used estimates for missing values and accounted for any nonrandom attrition, the proportion of children with poorer HRQOL trajectories may be underestimated. SIGNIFICANCE: Children with new onset epilepsy are heterogenous and follow unique HRQOL trajectories over the long term. Overall, HRQOL improves for the majority in the first 2 years after diagnosis, with these improvements sustained over the long term.

Validation of the Quality of Life in Childhood Epilepsy Questionnaire (QOLCE-55 and QOLCE-16) for use by parents of young adults with childhood-onset epilepsy.

OBJECTIVE: The objective of the study was to validate the parent-proxy reported Quality of Life in Childhood Epilepsy Questionnaire (QOLCE) in a sample of young adults with a history of childhood-onset epilepsy, allowing for the utilization of a consistent informant (the parent) across the youths' stages of development. The 55-item (QOLCE-55) and 16-item versions (QOLCE-16) were evaluated. METHODS: Data came from 134 young adults (aged 18.0 to 28.5 years) with childhood-onset epilepsy, recruited through community and tertiary care centers across Canada. Confirmatory factor analysis (CFA) was used to assess the higher-order factor structure of the QOLCE. Cronbach's alpha was used to evaluate internal consistency. Convergent validity was assessed by intraclass correlation coefficients (ICC) with the youth self-reported Quality of Life in Epilepsy Questionnaire (QOLIE-31-P). RESULTS: The higher-order factor structure of the QOLCE-55 and QOLCE-16 demonstrated adequate fit: QOLCE-55 comparative fit index (CFI) = 0.968, Tucker-Lewis index (TLI) = 0.966; and root mean square of approximation (RMSEA) = 0.061; QOLCE-16 CFI = 0.966, TFI = 0.959, RMSEA = 0.141. Higher-order factor loadings were strong, ranging from 0.71 to 0.90. Internal consistency was excellent for the total score (αQOLCE-55 = 0.97; αQOLCE-16 = 0.93) and good-excellent for each subscale (α > 78). Convergent validity was moderate to good for the total score (ICC > 0.72) and each subscale (ICC > 0.51). SIGNIFICANCE: These findings provide support for the use of the QOLCE-55 and QOLCE-16 among young adults with a history of childhood-onset epilepsy. Utilizing a consistent measure and informant across the stages of development is essential to reliably evaluate change over time.

Fatigue in young people with Duchenne muscular dystrophy.

AIM: To describe fatigue in Duchenne muscular dystrophy (DMD) from patients' and parents' perspectives and to explore risk factors for fatigue in children and adolescents with DMD. METHOD: A multicentre, cross-sectional study design was used. Seventy-one patients (all males; median age 12y, age range 5-17y) identified via the Canadian Neuromuscular Disease Registry, and their parents completed questionnaires. Subjective fatigue was assessed using the Pediatric Quality of Life Inventory Multidimensional Fatigue Scale by child self-report and parent proxy-report. RESULTS: Patients with DMD across ages and disease stages experienced greater fatigue compared to typically developing controls from published data. Sleep disturbance symptoms were associated with greater fatigue by child self-report (ρ=-0.42; p=0.003) and parent proxy-report (ρ=-0.51; p<0.001). Depressive symptoms were associated with greater fatigue by child self-report (ρ=-0.46; p<0.001) and parent proxy-report (ρ=-0.45; p<0.001). Lower functional ability was associated with greater fatigue by parent proxy-report (ρ=0.26; p=0.03). Physical activity level, and musculoskeletal, respiratory, and cardiac function were not associated with fatigue. INTERPRETATION: In paediatric DMD, sleep disturbance symptoms and depressive symptoms are potentially modifiable factors associated with fatigue, warranting additional investigation to facilitate the development of therapeutic strategies to reduce fatigue. WHAT THIS PAPER ADDS: Fatigue is a major issue in paediatric Duchenne muscular dystrophy (DMD) across ages and disease stages. Sleep disturbance and depressive symptoms are significantly associated with fatigue in paediatric DMD.

Seizure freedom improves health-related quality of life after epilepsy surgery in children.

AIM: To determine whether epilepsy surgery improved health-related quality of life (HRQoL) and whether seizure freedom after surgery mediated the improvement in HRQoL. METHOD: This multicenter cohort study compared HRQoL after epilepsy surgery to pharmacological management in children with drug-resistant epilepsy (DRE). HRQoL was measured using the Quality of Life in Childhood Epilepsy (QOLCE) questionnaire at baseline and 1-year follow-up. The mediator between treatment type and HRQoL was seizure freedom. RESULTS: Two hundred and thirty-seven patients were recruited (surgery group: n=147 [92 males, 45 females]; pharmacological group: n=90 [53 males, 37 females]). Mean age at seizure onset was 6 years (SD 4y 4mo) in the surgical group and 6 years 1 month (SD 4y) in the pharmacological group. The odds ratio of seizure freedom was higher for the surgery versus pharmacological group (ß=4.24 [95% confidence interval {CI}: 2.26-7.93], p<0.001). Surgery had no direct effect on total QOLCE score at 1-year (ß=0.24 [95% CI -2.04 to 2.51], p=0.839) compared to pharmacological management, but had an indirect effect on total QOLCE that was mediated by seizure freedom (ß=0.92 [95% CI 0.19-1.65], p=0.013), adjusting for baseline total QOLCE score. Surgery had a direct effect on improving social function (p=0.043), and an indirect effect on improving physical function (p=0.016), cognition (p=0.042), social function (p=0.012) and behavior (p=0.032), mediated by seizure freedom. INTERPRETATION: Greater seizure freedom achieved through epilepsy surgery mediated the improvement in HRQoL compared to pharmacological management in children with DRE. WHAT THIS PAPER ADDS: Seizure freedom is higher after pediatric epilepsy surgery compared to pharmacologically managed epilepsy. Surgery indirectly improves health-related quality of life (HRQoL) mediated by seizure freedom compared to pharmacological management. Surgery has a direct effect on improving social function relative to pharmacological management. Baseline HRQoL was an important predictor of HRQoL after treatment.

Towards the assessment of quality of life in patients with disorders of consciousness.

PURPOSE: To generate foundational knowledge in the creation of a quality-of-life instrument for patients who are clinically diagnosed as being in a vegetative or minimally conscious state but are able to communicate by modulating their brain activity (i.e., behaviourally nonresponsive and covertly aware). The study aimed to identify a short list of key domains that could be used to formulate questions for an instrument that determines their self-reported quality of life. METHODS: A novel two-pronged strategy was employed: (i) a scoping review of quality-of-life instruments created for patient populations sharing some characteristics with patients who are behaviourally nonresponsive and covertly aware was done to compile a set of potentially relevant domains of quality of life; and (ii) a three-round Delphi consensus process with a multidisciplinary panel of experts was done to determine which of the identified domains of quality of life are most important to those who are behaviourally nonresponsive and covertly aware. Five expert groups were recruited for this study including healthcare workers, neuroscientists, bioethicists, quality-of-life methodologists, and patient advocates. RESULTS: Thirty-five individuals participated in the study with an average response rate of 95% per round. Over the three rounds, experts reached consensus on 34 of 44 domains (42 domains were identified in the scoping review and two new domains were added based on suggestions by experts). 22 domains were rated as being important for inclusion in a quality-of-life instrument and 12 domains were deemed to be of less importance. Participants agreed that domains related to physical pain, communication, and personal relationships were of primary importance. Based on subgroup analyses, there was a high degree of consistency among expert groups. CONCLUSIONS: Quality of life should be a central patient-reported outcome in all patient populations regardless of patients' ability to communicate. It remains to be determined how covertly aware patients perceive their circumstances and quality of life after suffering a life-altering injury. Nonetheless, it is important that any further dialogue on what constitutes a life worth living should not occur without direct patient input.

Prevalence and trajectories of depressive symptoms among mothers of children with newly diagnosed epilepsy: A longitudinal 10-year study.

OBJECTIVE: Parental depression significantly impacts children's health and well-being. This study aimed to (1) estimate the prevalence of depressive symptoms, at six time points, among mothers over the first 10 years after their child was diagnosed with epilepsy; (2) identify trajectories of maternal depressive symptoms over time; and (3) identify baseline factors associated with each trajectory. METHODS: Data came from the Health-Related Quality of Life in Children with Epilepsy Study (HERQULES), a Canada-wide prospective cohort study. Data on child, parent, and family characteristics were collected at the time of diagnosis, and follow-ups at 0.5, 1, 2, 8, and 10 years. Maternal depressive symptoms were measured using the Center for Epidemiological Studies Depression Scale. Trajectories of depressive symptoms were evaluated using latent class growth modeling, and multinomial logistic regression was used to identify baseline factors associated with each trajectory. RESULTS: A total of 356 mothers participated in the study, of whom 57% scored in the at-risk range for major depression disorder (period-prevalence). Four unique trajectories were identified as follows: "Low-Stable" (29% of mothers), "Intermediate-Stable" (46%), "High-Stable" (20%), and "High-Decreasing" (5%). Positive family environment was consistently associated with a better trajectory of depressive symptoms over time; other significant factors included type of seizures, child cognitive comorbidity, maternal age, and maternal education. SIGNIFICANCE: A substantial proportion of mothers of children with epilepsy are at risk for depression, and this risk is stable over the long term. Family environment at the time of diagnosis has long-term and persistent effects and may be an ideal target for interventions.

Social outcomes for adults with a history of childhood-onset epilepsy: A systematic review and meta-analysis.

OBJECTIVES: This review aimed to describe social outcomes in adulthood for people with a history of childhood-onset epilepsy and identify factors associated with these outcomes; focused on educational attainment, employment, income/financial status, independence/living arrangement, romantic relationships, parenthood, and friendships. METHODS: A comprehensive search of MEDLINE, EMBASE, and PsycINFO was conducted, as well as forward and backward citation tracking. A total of 45 articles met inclusion criteria. Random effects meta-analyses were conducted, and subgroup analyses evaluated outcomes for people with epilepsy (PWE) with good prognosis (e.g., normal intelligence, 'epilepsy-only') and poor prognosis (e.g., intellectual disability, Dravet syndrome), and those who underwent epilepsy surgery in childhood. RESULTS: Among all PWE, 73% (95% confidence interval [CI]: 64-82%) completed secondary school education, 63% (95%CI: 56-70%) were employed; 74% (95%CI: 68-81%) did not receive governmental financial assistance; 32% (95%CI: 25-39%) were in romantic relationships; 34% (95%CI: 24-45%) lived independently; 21% (95%CI:12-33%) had children, and 79% (95%CI: 71-87%) had close friend(s). People with epilepsy often fared worse relative to healthy controls. Among PWE with a good prognosis, a comparable number of studies reported similar/better outcomes relative to controls as reported poorer outcomes. The most consistent predictor of poorer outcomes was the presence of cognitive problems; results of studies evaluating seizure control were equivocal. CONCLUSION: People with epilepsy with a good prognosis may show similar social outcomes as controls, though robust conclusions are difficult to make given the extant literature. Seizure control does not guarantee better outcomes. There is a need for more studies evaluating prognostic factors and studies with control groups to facilitate appropriate comparisons.

Measurement equivalence of the short-form Quality of Life in Childhood Epilepsy Questionnaire (QOLCE-16).

OBJECTIVE: The aim of this study was to assess measurement equivalence in the 16-item short-form Quality of Life in Childhood Epilepsy Questionnaire (QOLCE-16) across age, sex, and time in a sample of children with newly diagnosed epilepsy. METHODS: Data came from 373 children participating in the Health-related Quality of Life in Children with Epilepsy Study (HERQULES), a multicenter prospective cohort study. Measurement equivalence was assessed using multigroup confirmatory factor analysis methods. Comparison groups were stratified by age (4-7 years vs. 8-12 years), sex (male vs. female), and time (at diagnosis vs. 24 months postdiagnosis). RESULTS: The QOLCE-16 demonstrated measurement equivalence at the level of strict invariance for each comparison group tested - age: χ2 (3, 274) = 429.6, p < 0.001; comparative fit index (CFI) = 0.985; root mean square error of approximation (RMSEA) = 0.056 (0.046, 0.066); sex: χ2 (3, 271) = 430.5, p < 0.001; CFI = 0.984; RMSEA = 0.057 (0.047, 0.067); and time: χ2 (3, 269) = 566.4, p < 0.001; CFI = 0.985; RMSEA = 0.059 (0.052, 0.066). SIGNIFICANCE: The findings provide support for the robust psychometric profile of the QOLCE-16 as a reliable and valid measure of health-related quality of life for children with epilepsy. Demonstrating good properties and a multidimensional structure, the QOLCE-16 is an appropriate short measure for both clinicians and researchers wanting to obtain health-related quality of life information on children with epilepsy.

The evaluation and costs of transition programs for youth with epilepsy.

There is limited information about the effectiveness of transition programs for youth moving from pediatric to adult care with any chronic disease. Two Delphi studies and National Institute for Health and Care Excellence (NICE) guidelines about transition for epilepsy have suggested few critical outcome measures for transition. A single large prospective study found that the most important transition program elements were appropriate parent involvement, promotion of health self-efficacy, and meeting the adult team before transfer. Two Cochrane reviews of the value of transition for epilepsy found insufficient evidence to establish or refute the value of various programs, although evaluation of a few programs suggested a great deal of family/patient satisfaction. The cost of transition programs and their cost effectiveness have also not been established except for renal transplantation where transition programs were associated with fewer losses of the transplanted kidneys, a cost-effective outcome. Published data on the overall cost of care for children and adults with epilepsy may be helpful to establish a business plan for a transition program, and are briefly reviewed. Establishing cost effectiveness of transition programs for epilepsy would promote their establishment and viability. However, a number of studies will be needed based on the nature of the program, the healthcare system where it is carried out, and the type of epilepsy. In fee-for-service health systems, the reevaluation of patients with epilepsy prior to transfer may be sufficient to cover the costs of the transition program, whereas in single payer systems, there may be positive downstream health or societal benefits that justify the costs. A theoretical framework for comprehensive evaluation of epilepsy transition programs is needed. The Triple Aim Framework seems applicable with focus on population health, patient experiences, and cost and has the potential to assess transition interventions in the context of system-wide improvements in healthcare. Transition programs in general have not been well evaluated, and very little evaluation data exist regarding transition programs for epilepsy. We recommend more evaluative research using rigorous methodology to comprehensively assess these programs.

Web-Based Tools for Educating Caregivers About Childhood Fever: A Randomized Controlled Trial.

OBJECTIVES: Fever is a common reason for an emergency department visit and misconceptions abound. We assessed the effectiveness of an interactive Web-based module (WBM), read-only Web site (ROW), and written and verbal information (standard of care [SOC]) to educate caregivers about fever in their children. METHODS: Caregivers in the emergency department were randomized to a WBM, ROW, or SOC. Primary outcome was the gain score on a novel questionnaire testing knowledge surrounding measurement and management of fever. Secondary outcome was caregiver satisfaction with the interventions. RESULTS: There were 77, 79, and 77 participants in the WBM, ROW, and SOC groups, respectively. With a maximum of 33 points, Web-based interventions were associated with a significant mean (SD) pretest to immediate posttest gain score of 3.5 (4.2) for WBM (P < 0.001) and 3.5 (4.1) for ROW (P < 0.001) in contrast to a nonsignificant gain score of 0.1 (2.7) for SOC. Mean (SD) caregiver satisfaction scores (out of 32) for the WBM, ROW, and SOC groups were 22.6 (3.2), 20.7 (4.3), and 17 (6.2), respectively. All groups were significantly different from one another in the following rank: WBM > ROW > SOC (P < 0.001). CONCLUSIONS: Web-based interventions are associated with significant improvements in caregiver knowledge about fever and high caregiver satisfaction. These interventions should be used to educate caregivers pending the demonstration of improved patient-centered outcomes.