Parental Experience and Decision-Making for Epilepsy Surgery: A Systematic Review of Qualitative and Quantitative Studies

Abstract, originally published in Epilepsy & Behavior

Objective: In selected children with drug-resistant epilepsy (DRE), epilepsy surgery is the most effective treatment option, but unfortunately remains highly underutilized. One of the critical obstacles to pursuing surgical therapy is parents/caregivers’ decision against surgery or to delay the surgery until no other treatment option exists. Understanding caregiver decision-making around epilepsy surgery can improve patient/caregiver experience and satisfaction while facilitating appropriate decision-making that optimizes clinical outcomes. The current review systematically explores the existing evidence on caregiver experience and the decision-making process toward epilepsy surgery.

Methods: The study was conducted as per the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines for systematic literature review. Databases (PubMed Ovid, PubMed Medline, Web of Science, CINHAL, PsycInfo) were systematically searched in February 2021 using a defined search strategy and inclusion/exclusion criteria. Total 1304 articles were screened for titles and abstracts, and 54 full-text articles were retrieved for further assessment. We included 14 articles with critical quality assessment using two different tools for qualitative and questionnaire-based studies. A qualitative content analysis was performed to characterize caregiver experience, perception, and decision-making toward favorable or unfavorable opinions of epilepsy surgery.

Results: Four concepts generated from the analysis may act as enablers or barriers to decision-making around epilepsy surgery: 1. Access to knowledge and information, 2. Communication and coordination issues, 3. Caregiver’s emotional state, and 4. Socioeconomic effects. Subsequently, we provided a narrative synthesis of practice recommendations and a conceptual framework to adopt multi-pronged interventions to overcome identified diverse barriers to effective caregiver decision-making.

Conclusion: Multiple influences impact how caregivers decide about epilepsy surgery for their children, with no single factor identified as the primary driver for or against surgery. However, limited research has explored these influences. Future studies should focus on quantitatively examining factors to identify significant variables most likely to influence caregiver decision-making, ultimately overcoming barriers that limit utilization of epilepsy surgery as a treatment tool.

ADHD and Epilepsy-Related QoL in Youth with Epilepsy

Abstract, originally published in Epilepsy & Behavior

Objective: Pediatric epilepsy and attention-deficit/hyperactivity disorder (ADHD) present a cumulative vulnerability to youth, particularly in the domains of executive function (EF) and quality of life (QoL). The aim of this study was to explore the relationships between EF and epilepsy-specific QoL in youth with epilepsy (YWE) who also have ADHD.

Methods: Youth with epilepsy aged 5-18 years and caregivers participated in a large multi-site national validation of the PedsQL™ Epilepsy Modules. YWE and their caregivers were asked to complete measures of epilepsy-specific QoL, ADHD symptoms, and EF. Previous ADHD diagnosis was reported by caregivers as well as abstracted from the medical record.

Results: Caregiver and YWE reports of current ADHD symptoms significantly predicted all epilepsy-specific QoL domains. Suboptimal QoL was reported for those YWE whose ADHD symptoms were above the clinical threshold compared to YWE whose ADHD symptoms were within the average range. Results were similar for YWE with a documented ADHD vs no ADHD diagnosis. In addition, poorer metacognition and behavioral regulation EF was associated with lower epilepsy-specific QoL for both YWE with ADHD and without ADHD diagnosis. However, in YWE with ADHD, effect sizes were consistently larger for behavioral regulation compared to metacognition across epilepsy-specific QoL domains, with behavioral regulation accounting for 50% of the variance in each of the EF and mood QoL domains.

Significance: Addressing ADHD symptoms, epilepsy-specific, quality of life, and executive function as part of routine epilepsy care may promote optimal outcomes for YWE with ADHD as well as relieve burden on the healthcare system.

‘To Reveal or to Conceal’- Disclosure Strategies in Parents of Children With Epilepsy in India

Abstract, originally published in Seizure

Purpose: Disclosure of epilepsy is a relevant but under-researched topic in epilepsy research. This study was done to assess the disclosure strategies in parents of children with epilepsy in a developing country with conservative culture. The study also assessed the influence of demographic factors and seizure characteristics on the choice of disclosure. Enablers and barriers behind disclosure and the consequences after disclosure were evaluated.

Methods: A cross-sectional analytical, self-report survey was done in 284 parents of CWE with the help of a semi-structured questionnaire over a 7-month period in the paediatric epilepsy clinic. Disclosure was considered present if epilepsy was revealed to two or more of the five target groups (extended family, school, friends, neighbours, and peers of children). Separate set of questions was given for reasons behind their choice and consequences after disclosure. For continuous variables, unpaired T test or Mann – Whitney U test between group and for categorized variables, Pearson’s Chi square test or Fisher’s exact test was used.

Results: 92.96 % of 284 subjects disclosed their child’s epilepsy while 7.04% concealed. Demographic factors and seizure characteristics did not influence the disclosure choice. Most parents revealed to the extended family followed by teachers. Type of seizure was the commonest information revealed. The main reason behind disclosure was better acceptance of the child followed by safety while main barrier was considering epilepsy as private grief. 92.8% felt their children were better accepted after disclosure.

Conclusion: Disclosure practices have improved in parents of children with epilepsy in India and well-being and safety of the child has overridden the fear of stigma and discrimination. This could be the first major step to bring epilepsy out of the shadows at national and global levels.

Post-Traumatic Stress Disorder (PTSD) Symptoms in Children with Severe Epilepsy

Abstract, published in, originally published in Epilepsy Behavior

Objectives: To assess symptoms of post-traumatic stress disorder (PTSD) in children with severe epilepsy and the associations of trauma symptoms across age, comorbid symptoms, epilepsy-specific factors, parental resources, and psychopathology.

Methods: Fifty children with severe epilepsy across three different age groups (0-5 yrs., 6-12 yrs., 13-18 yrs.) were assessed with developmental-sensitive and standardized PTSD assessment tools when hospitalized at the tertiary epilepsy center Filadelfia, Denmark. The Diagnostic Infant and Preschool Assessment (DIPA), the Darryl test, and the ITQ questionnaire were used to assess the three age groups, respectively.

Results: Twenty-two percent of the overall sample met the criteria for PTSD, with a prevalence of symptoms increasing with age (6%, 28%, and 40%). Comorbid psychiatric symptoms in preschoolers were present in 81% of the children witnessing a high level of distress in this group. Behavioral difficulties were elevated across all three age groups, and 40% of the children with trauma symptoms had a parent with concurrent psychopathology.

Conclusion: To the authors’ knowledge, this study is the first to assess trauma symptoms with standardized tests in children with more complicated epilepsies. Trauma symptoms in the group are high; however, there is a need for larger-scale studies and research into trauma symptoms in children with more severe epilepsy than those assessable with the included assessment tools. The trauma perspective in severe childhood epilepsy might further clarify the complex associations of biological and contextual variables that affect the children’s life quality and enable better preventative treatment options for this group.

Self-esteem Mediates Mental Health Outcomes in Young People with Epilepsy

Summary, published in Epilepsia

Objective: To evaluate the extent to which self-esteem mediates the impacts of epilepsy-specific and environmental factors on mental health outcomes in young people with epilepsy.

Methods: A prospective cohort of 480 young people with epilepsy and their families participated in five visits over 28 months. We collected data on clinical seizure burden, cognitive comorbidity, peer and parental support, self-esteem, and self-reported mental health symptoms. We used structural equation modeling to specify and test relationships among these constructs simultaneously. Direct, indirect, and total effects were estimated with confidence intervals constructed through bias-corrected bootstrapping.

Results: Self-esteem mediated the effects of clinical seizure burden ( ?? = 0.23, 95% confidence interval [0.05, 0.42]) and peer support ( ?? = ?0.15, 95% CI [?0.28, ?0.03]) on mental health. There were no mediating effects of parental support ( ?? = ?0.07, 95% CI [?0.14, 0.00]) or cognitive comorbidity ( ?? = ?0.01, 95% CI [?0.02, 0.01]) on mental health.

Significance: We found evidence that self-esteem mediates the impact that both clinical seizure burden and peer support have on mental health outcomes, indicating that assessment of and interventions targeting self-esteem may be appropriate for young people with epilepsy. Supporting self-esteem could mitigate negative influences on mental health, whether from resistant epilepsy or low peer support.

Intellectual Outcome from 1 to 5 years After Epilepsy Surgery in 81 Children and Adolescents: A Longitudinal Study

Abstract, published in Seizure

Objective: This longitudinal study aimed to measure the time course of intellectual changes after pediatric focal resective epilepsy surgery and to identify their predictors.

Methods: We analyzed a cohort of 81 school-aged children with focal epilepsy and intractable seizures who underwent neurosurgery (focal resection) from 2000 to 2018 in La Timone Hospital (Marseille). Neuropsychological assessments were carried out before and then 1, 2, 3, and 5 years after epilepsy surgery.

Results: Eighty-one patients with a median age at surgery of 13.74 years [4.25] were enrolled. Overall, 45 of the 81 (55%) recruited patients were improved after the surgery on at least one of the five domains of the Wechsler Intelligence Scale. Temporal lobe localization and postoperative seizure freedom were the main prognostic factors impacting intellectual outcome (improvement and decline) after epilepsy surgery. Younger patients at surgery were less likely to have a postoperative IQ decline. Intellectual improvement after epilepsy surgery could be delayed for up to 5 years after surgery and concerned all intellectual domains except the Verbal Comprehension Index (VCI). Intellectual decline after epilepsy surgery occurred mainly during the first two years after the surgery and was reflected in full-scale intelligence quotient (FSIQ) and Working Memory Index (WMI).

Conclusions: Our study points out that children and adolescents with temporal lobe epilepsy who achieved freedom from seizure after epilepsy surgery are the leading candidates for achieving postoperative intellectual improvement. This enhancement in intellectual function shows a long time course, whereas intellectual decline is evidenced earlier.

Impaired Social Attention Detected Through Eye Movements in Children with Early-onset Epilepsy

Abstract, posted in Epilepsia

Objectives: Children with early-onset epilepsy (CWEOE; epilepsy onset before 5 years) exhibit impaired social functioning, but social attention has not yet been examined. In this study we sought to explore visual attention via eye tracking as a component of social attention and examine its relationship with social functioning and Autism Spectrum Disorder (ASD) risk scores.

Methods: Forty-seven CWEOE (3-63 months) and 41 controls (3-61 months) completed two eye-tracking tasks: (1) preference for social versus nonsocial naturalistic scenes, and (2) face region preference task. ASD risk was measured via the Modified Checklist for Autism in Toddlers or Conners Early Childhood Total Score. Social functioning was assessed via the Greenspan Social-Emotional Growth Chart, or Infant-Toddler Social & Emotional Assessment Competence Scale, or Conners Early Childhood Social Functioning Scale, depending on age. Fixation preferences for social scenes and eyes were compared between groups and evaluated by age and social functioning scores.

Results: Regression analysis revealed that CWEOE viewed the social scene to a significantly less degree than controls. The greatest difference was found between the youngest CWEOE and controls. Fixation duration was independently and significantly related to social functioning scores. There were no significant differences between CWEOE and controls in the face scanning task, and there was no significant relationship between either task and ASD risk scores.

Significance: Children with early-onset epilepsy exhibit task-specific atypical social attention early in the course of the disease. This may be an early marker of impaired social development, and it suggests abnormal social brain development.

Epilepsy Surgery in Infants up to 3 months of Age: Safety, Feasibility, and Outcomes: A Multicenter, Multinational Study

Summary, published in Epilepsia

Objective: Drug-resistant epilepsy (DRE) during the first few months of life is challenging and necessitates aggressive treatment, including surgery. Because the most common causes of DRE in infancy are related to extensive developmental anomalies, surgery often entails extensive tissue resections or disconnection. The literature on “ultra-early” epilepsy surgery is sparse, with limited data concerning efficacy controlling the seizures, and safety. The current study’s goal is to review the safety and efficacy of ultra-early epilepsy surgery performed before the age of 3 months.

Methods: To achieve a large sample size and external validity, a multinational, multicenter retrospective study was performed, focusing on epilepsy surgery for infants younger than 3 months of age. Collected data included epilepsy characteristics, surgical details, epilepsy outcome, and complications.

Results: Sixty-four patients underwent 69 surgeries before the age of 3 months. The most common pathologies were cortical dysplasia (28), hemimegalencephaly (17), and tubers (5). The most common procedures were hemispheric surgeries (48 procedures). Two cases were intentionally staged, and one was unexpectedly aborted. Nearly all patients received blood products. There were no perioperative deaths and no major unexpected permanent morbidities. Twenty-five percent of patients undergoing hemispheric surgeries developed hydrocephalus. Excellent epilepsy outcome (International League Against Epilepsy [ILAE] grade I) was achieved in 66% of cases over a median follow-up of 41 months (19–104 interquartile range [IQR]). The number of antiseizure medications was significantly reduced (median 2 drugs, 1–3 IQR, p < .0001). Outcome was not significantly associated with the type of surgery (hemispheric or more limited resections).

Significance: Epilepsy surgery during the first few months of life is associated with excellent seizure control, and when performed by highly experienced teams, is not associated with more permanent morbidity than surgery in older infants. Thus surgical treatment should not be postponed to treat DRE in very young infants based on their age.

Pediatric Epilepsy and Psychoeducational Interventions: A review of the Literature

Abstract, published in Epilepsy & Behavior

For many individuals, living with epilepsy is truly a family affair throughout the life span. When it comes to childhood epilepsy, the unpredictability of seizure patterns, comorbid conditions, the risk of sudden unexpected death in epilepsy (SUDEP), and societal stigma can be emotionally taxing on children and their primary caregivers. To this end, this article proposes to review psychoeducational interventions provided to primary caregivers of children with an epilepsy diagnosis and the impact of such interventions on general parental coping skills. There were three main themes identified (1) caregivers’ knowledge and self-efficacy about seizure management; (2) parental epilepsy-related fears, anxiety, and stress; (3) parental sleep quality and SUDEP psychoeducation. Overall, considering research limitations, providing epilepsy-related psychoeducational interventions to primary caregivers of children with epilepsy seems to have promising evidence in the literature. After receiving such interventions, the studies show that caregivers’ psychosocial outcomes improved; they become more empowered to manage their children’s seizures and advocate for their children’s psychosocial needs.

A children’s epilepsy diagnosis aid: Development and early validation using a Bayesian approach

Abstract, published in Epilepsy & Behavior

Introduction: The diagnosis of epilepsy in children is difficult and misdiagnosis rates can be as much as 36%. Diagnosis in all countries is essentially clinical, based on asking a series of questions and interpreting the answers. Doctors experienced enough to do this are either scarce or absent in very many parts of the world so there is a need to develop a diagnostic aid to help less-experienced doctors or non-physician health workers (NPHWs) do this. We used a Bayesian approach to determine the most useful questions to ask based on their likelihood ratios (LR), and incorporated these into a Children’s Epilepsy Diagnosis Aid (CEDA).

Methods: Ninety-six consecutive new referrals with possible epilepsy aged under 10 years attending a pediatric neurology clinic in Khartoum were included. Initially, their caregivers were asked 65 yes/no questions by a medical officer, then seen by pediatric neurologist and the diagnosis of epilepsy (E), not epilepsy (N), or uncertain (U) was made. The LR was calculated and then we selected the variables with the highest and lowest LRs which are the most informative at differentiating epilepsy from non-epilepsy. An algorithm, (CEDA), based on the most informative questions was constructed and tested on a new sample of 47 consecutive patients with a first attendance of possible epilepsy. We calculated the sensitivity and specificity for CEDA in the diagnosis of epilepsy.

Results: Sixty-nine (79%) had epilepsy and 18 (21%) non-epilepsy giving pre-test odds of having epilepsy of 3.83. Eleven variables with the most informative LRs formed the diagnostic aid (CEDA). The pre-test odds and algorithm were used to determine the probability of epilepsy diagnosis in a subsequent sample of 47 patients. There were 36 patients with epilepsy and 11 with nonepileptic conditions. The sensitivity of CEDA was 100% with specificity of 97% and misdiagnosis 8.3%.

Conclusion: Children’s Epilepsy Diagnosis Aid has the potential to improve pediatric epilepsy diagnosis and therefore management and is particularly likely to be useful in the many situations where access to epilepsy specialists is limited. The algorithm can be presented as a smartphone application or used as a spreadsheet on a computer.