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OBJECTIVE: Epilepsy is associated with significant mortality risk. There is limited research examining how traumatic brain injury (TBI) timing affects mortality in relation to the onset of epilepsy. We aimed to assess the temporal relationship between epilepsy and TBI regarding mortality in a cohort of post-9/11 veterans. METHODS: This retrospective cohort study included veterans who received health care in the Defense Health Agency and the Veterans Health Administration between 2000 and 2019. For those diagnosed with epilepsy, the index date was the date of first antiseizure medication or first seizure; we simulated the index date for those without epilepsy. We created the study groups by the index date and first documented TBI: (1) controls (no TBI, no epilepsy), (2) TBI only, (3) epilepsy only, (4) TBI before epilepsy, (5) TBI within 6 months after epilepsy, and (6) TBI >6 months after epilepsy. Kaplan-Meier estimates of all-cause mortality were calculated, and log-rank tests were used to compare unadjusted cumulative mortality rates among groups compared to controls. Cox proportional hazard models were used to compute hazard ratios (HRs) with 95% confidence intervals (CIs). RESULTS: Among 938 890 veterans, 27 436 (2.92%) met epilepsy criteria, and 264 890 (28.22%) had a TBI diagnosis. Mortality was higher for veterans with epilepsy than controls (6.26% vs. 1.12%; p < .01). Veterans with TBI diagnosed ≤6 months after epilepsy had the highest mortality hazard (HR = 5.02, 95% CI = 4.21-5.99) compared to controls, followed by those with TBI before epilepsy (HR = 4.25, 95% CI = 3.89-4.58), epilepsy only (HR = 4.00, 95% CI = 3.67-4.36), and TBI >6 months after epilepsy (HR = 2.49, 95% CI = 2.17-2.85). These differences were significant across groups. SIGNIFICANCE: TBI timing relative to epilepsy affects time to mortality; TBI within 6 months after epilepsy or before epilepsy diagnosis was associated with earlier time to death compared to those with epilepsy only or TBI >6 months after epilepsy.
Subject(s)
Brain Injuries, Traumatic , Epilepsy , Veterans , Humans , Brain Injuries, Traumatic/mortality , Brain Injuries, Traumatic/complications , Veterans/statistics & numerical data , Male , Female , Adult , Epilepsy/mortality , Middle Aged , Retrospective Studies , United States/epidemiology , Time Factors , Cohort Studies , Aged , Proportional Hazards ModelsABSTRACT
Accurate and complete racial/ethnic data in the electronic health record are a requisite step to addressing disparities in neurologic care, and at local, regional, and national levels. The current data pertaining to the patients' race and ethnicity contained in the electronic health record are inadequate. This article outlines recommendations at the individual practice and electronic health record vendor level to improve documentation of race and ethnicity.
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BACKGROUND AND OBJECTIVES: Traumatic brain injury (TBI) is a well-established epilepsy risk factor and is common among service members. Deployment-related TBI, where combat/blast may be more common, may have different outcomes than nondeployment-related TBI. This work examined associations of all TBI exposures (not just combat), and epilepsy, while adjusting for comorbidities associated with epilepsy, among veterans by deployment status. METHODS: The cohort included post-9/11 veterans with ≥2 years of care in both Veterans Health Administration and Defense Health Agency systems. We identified epilepsy using ICD-9/10-CM codes, antiseizure medication, and service-connected disability for epilepsy. We conducted a logistic regression model with interaction terms for conditions by deployment history that adjusted for demographics and military characteristics. RESULTS: The cohort (n = 938,890) included post-9/11 veterans of whom 27,436 (2.92%) had epilepsy. Most veterans had a history of deployment (70.64%), referred to as "deployed." Epilepsy was more common among veterans who were never deployed ("nondeployed") (3.85% vs 2.54%). Deployed veterans were more likely to have had TBI, compared with the nondeployed veterans (33.94% vs 14.24%), but nondeployed veterans with moderate/severe TBI had higher odds of epilepsy compared with deployed veterans (adjusted odds ratio [aOR] 2.92, 95% CI 2.68-3.17 vs aOR 2.01, 95% CI 1.91-2.11). Penetrating TBI had higher odds of epilepsy among the deployed veterans (aOR 5.33, 95% CI 4.89-5.81), whereas the odds of epilepsy for mild TBI did not significantly differ by deployment status. Although most neurologic conditions were more prevalent among the nondeployed veterans, they were often associated with higher odds of epilepsy in the deployed veterans. DISCUSSION: Deployment history had a significant differential impact on epilepsy predictors. As expected, penetrating TBI had a greater epilepsy impact among deployed veterans perhaps due to combat/blast. Some epilepsy predictors (moderate/severe TBI, multiple sclerosis, and Parkinson disease) had a stronger association in the nondeployed veterans suggesting a potential healthy warrior effect in which such conditions preclude deployment. Other neurologic conditions (e.g., brain tumor, Alzheimer disease/frontotemporal dementia) had a greater epilepsy impact in the deployed veterans. This may be attributable to deployment-related exposures (combat injury, occupational exposures). A better understanding of deployment effects is critical to provide targeted epilepsy prevention in veterans and military service members.
Subject(s)
Brain Injuries, Traumatic , Epilepsy , Military Personnel , Veterans , Humans , Brain Injuries, Traumatic/complications , Brain Injuries, Traumatic/epidemiology , Comorbidity , Epilepsy/epidemiologyABSTRACT
OBJECTIVE: Drug-resistant epilepsy can be difficult to cure and may pose emotional challenges for epilepsy providers. Neuropalliative care (NPC) can augment quality of life (QOL) in persons with neurological diseases and may add meaningful elements to the treatment repertoire of epilepsy specialists even if seizures continue. However, NPC has not been widely implemented in epilepsy. Our study aimed to determine whether physicians of persons with drug-resistant epilepsy (PWDRE) experience distress when faced with treatment failure (Engel class ≥ 2), either failure of medications-only (PWDREmo) or of both medications and surgery (procedures with curative intent (PWDREms)). Furthermore, we evaluated physician knowledge about and referrals to NPC following treatment failures to help improve patient QOL despite ongoing seizures. METHODS: An anonymous online survey was distributed to US epilepsy physicians through the American Epilepsy Society website and personal email to assess levels of distress experienced when caring for PWDREmo and PWDREms (7-point Likert scale ["1" = "no distress", "7" = "most distress ever felt"]), and knowledge and use of NPC. RESULTS: Eighty-two physicians completed the survey. Most experienced distress when epilepsy treatments failed: 59% felt moderate distress (≥4) with PWDREmo (median "4", mean 3.74, range 1-7), 90% suffered moderate to severe distress (5, 5.17, 1-7) with PWDREms. Distress over PWDREms was significantly greater than distress over PWDREmo (p < 0.0001). Forty-three percent reported confidence in their knowledge about NPC. Only 15% were likely to refer PWDREmo to NPC, while 44% would consider it for PWDREms. CONCLUSION: Among survey responders, physician distress was high when confronted with treatment failures, especially the failure of epilepsy surgery. Fewer than half of responders were likely to refer patients to NPC. Further research is necessary to determine extent, reasons, and effects of physician distress and whether improved understanding of and patient access to NPC would help alleviate physician distress when faced with treatment failures in PWDRE.
Subject(s)
Drug Resistant Epilepsy , Epilepsy , Physicians , Humans , Quality of Life , Epilepsy/psychology , Drug Resistant Epilepsy/therapy , Seizures/therapyABSTRACT
GOALS: To define fragmentation in neurological care delivery; explain the positive and negative drivers in neurologic practice that contribute to fragmentation; illustrate situations that increase fragmentation risk; emphasize the costs and impact on both patients and providers; propose solutions that allow for more cohesive care. WORK GROUP: The Transforming Leaders Program (TLP) class of 2020 was tasked by American Academy of Neurology (AAN) leadership to identify the leading trends in inpatient and outpatient neurology and to predict their effects on future neurologic practice. METHODS: Research material included AAN data bases, PubMed searches, discussion with topic experts and AAN leadership. RESULTS: Trends in care delivery are driven by changes in the work force, shifts in health care delivery, care costs, changes in evidence-based care and patient factors. These trends can contribute to care fragmentation. Potential solutions to these problems are proposed based on care models developed in oncology and medicine. LIMITATIONS: This paper shares our opinions as there is a lack of evidence-based guidelines as to optimal neurological care delivery.
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We present the case of a young woman being treated with rituximab for rheumatoid arthritis who developed a severe enteroviral meningoencephalitis and acute flaccid myelitis (AFM). Cerebrospinal fluid (CSF) and stool reverse transcription-polymerase chain reaction (RT-PCR) testing confirmed the diagnosis and additional sequencing studies performed at the CDC further characterized the enterovirus as enterovirus A71 (EV-A71). After treatment with intravenous immunoglobulin (IVIg) and fluoxetine (based on previous reports of possible efficacy) the patient experienced a remarkable improvement over time. This case highlights the importance of considering enteroviral infection in patients treated with rituximab, depicts a possible clinical course of enteroviral meningoencephalitis and AFM, and illustrates the importance of testing multiple sites for enterovirus infection (CSF, stool, nasopharyngeal swab, blood). Here we present the case with a brief review of the literature pertaining to EV-A71.
Subject(s)
Central Nervous System Viral Diseases/diagnostic imaging , Enterovirus A, Human/isolation & purification , Enterovirus Infections/diagnostic imaging , Immunologic Factors/therapeutic use , Meningoencephalitis/diagnostic imaging , Myelitis/diagnostic imaging , Neuromuscular Diseases/diagnostic imaging , Rituximab/therapeutic use , Adult , Central Nervous System Viral Diseases/drug therapy , Central Nervous System Viral Diseases/virology , Enterovirus Infections/drug therapy , Female , Humans , Immunologic Factors/adverse effects , Meningoencephalitis/drug therapy , Meningoencephalitis/virology , Myelitis/drug therapy , Myelitis/virology , Neuromuscular Diseases/drug therapy , Neuromuscular Diseases/virology , Rituximab/adverse effectsABSTRACT
We aimed to describe perspectives of transition and transfer of adolescents and young adults with childhood-onset epilepsy from pediatric to adult care from the viewpoints of both pediatric and adult epileptologists. Telephone semi-structured interviews with pediatric (nâ¯=â¯15) and adult (nâ¯=â¯11) epileptologists at leading U.S. epilepsy centers were used to collect data about the transition process. Interviews were audio-recorded, transcribed, systematically coded using thematic analysis by two independent researchers, and subsequently checked for agreement during regular meetings. Participants were on average 46â¯years old (SDâ¯=â¯7.4), 50% male, 91% Non-Hispanic and 85% Caucasian; all had completed a formal epilepsy or clinical neurophysiology fellowship (meanâ¯=â¯11â¯years since terminal training) and were employed at a comprehensive epilepsy center. Three interrelated themes regarding epileptologists' perspectives of epilepsy transition and transfer of care were evident: (1) the process is unnatural and disruptive, (2) clinicians make the best of challenges, and (3) the epilepsy transition process includes a spectrum of broad needs some of which are unique to epilepsy care while others are common to other chronic diseases. Despite challenges, epilepsy clinicians spontaneously expressed stress, empathy, and commitment to providing the best possible care.
Subject(s)
Epilepsy , Transition to Adult Care , Adolescent , Child , Chronic Disease , Female , Humans , Male , Middle Aged , Patient Transfer , Qualitative Research , Young AdultABSTRACT
A 43-year-old woman presented with cognitive decline, focal seizures, brain MRI showing non-enhancing, bilateral hippocampal lesions, but normal cerebrospinal fluid findings, which fulfilled the Graus et al., 2016 criteria for autoimmune limbic encephalitis (ALE). Subjective improvements were observed after immunotherapy. A repeat brain MRI showed new contrast enhancement and positron emission tomography revealed left hippocampal uptake. Biopsy of the right parahippocampus yielded high-grade glioma. Five similar cases, among the 14 with unilateral hippocampal lesions on MRI, were identified in the literature whereby suspected ALE preceded the high-grade glioma diagnosis. Gliomas confined to hippocampi can have clinical features overlapping with ALE.
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OBJECTIVE: To conduct a data validation study encompassing an accuracy assessment of the data extraction process for the Axon Registry®. METHODS: Data elements were abstracted from electronic health records (EHRs) by an external auditor (IQVIA) using virtual site visits at participating sites. IQVIA independently calculated Axon Registry quality measure performance rates based on American Academy of Neurology measure specifications and logic using Axon Registry data. Agreement between Axon Registry and IQVIA data elements and measure performance rates was calculated. Discordance was investigated to elucidate underlying systemic or idiosyncratic reasons for disagreement. RESULTS: Nine sites (n = 720 patients; n = 80 patients per site) with diversity among EHR vendor, practice settings, size, locations, and data transfer method were included. There was variable concordance between the data elements in the Axon Registry and those abstracted independently by IQVIA; high match rates (≥92%) were observed for discrete elements (e.g., demographics); lower match rates (<44%) were observed for elements with free text (e.g., plan of care). Across all measures, there was a 76% patient-level measure performance agreement between Axon Registry and IQVIA (κ = 0.53, p < 0.001). CONCLUSION: There was a range of concordance between data elements and quality measures in the Axon Registry and those independently abstracted and calculated by an independent vendor. Validation of data and processes is important for the Axon Registry as a clinical quality data registry that utilizes automated data extraction methods from the EHR. Implementation of remediation strategies to improve data accuracy will support the ability of the Axon Registry to perform accurate quality reporting.
Subject(s)
Data Accuracy , Nervous System Diseases , Registries , Electronic Health Records , Humans , United StatesSubject(s)
Epilepsy/therapy , Neurology/standards , Quality Improvement/standards , Humans , Neurology/methodsABSTRACT
INTRODUCTION: A substantial proportion of young adults with childhood-onset epilepsy may require ongoing long-term epilepsy care as adults. OBJECTIVE: The objective of the study was to assess the extent to which epilepsy transition discussions occurred in adolescents with childhood-onset epilepsy prior to the age of attaining majority (prior to their 18th birthday) in a community-based cohort of individuals with childhood-onset epilepsy followed longitudinally. METHODS: The Connecticut Study of Epilepsy is a prospective, community-based study of newly diagnosed childhood-onset epilepsy with 613 children (onsetâ¯<â¯16â¯years old; year recruited: 1993-97). During the final exit interview, 308 young adults ≥18â¯years old (or parent-proxies) were asked, "Before you turned 18 years old, did your doctors or other epilepsy care providers talk with you about how your epilepsy care needs might change as you get older?" ('transition discussion'). We examined whether or not sociodemographic and clinical characteristics were associated with epilepsy transition discussions. RESULTS: For young adults with childhood-onset epilepsy (Nâ¯=â¯308; mean age: 24â¯years, SDâ¯=â¯4.0; mean age of epilepsy onset: 5.4â¯years, SDâ¯=â¯3.7), only 15% responded "Yes" to having had a "transition discussion". Of those with "active epilepsy" (Nâ¯=â¯130; seizure-freeâ¯<â¯5â¯years or on an antiseizure medication within 2â¯years of their 18th birthday) upon attaining the age of majority (18â¯years), 40/130 (~31%) young adults had "transition" discussions, compared with 7/178 (4%) of those with "inactive epilepsy" (pâ¯<â¯0.0001). Self- (Nâ¯=â¯95 active epilepsy) and proxy-reports (Nâ¯=â¯35 active epilepsy) of "transition" discussions were comparable (31%). Having a transition discussion was associated with neurodevelopmental comorbidity and type of epilepsy care provider at time of last contact (pâ¯<â¯0.05). Having a "transition" discussion was not associated with gender, race/ethnicity, high school graduation, parent insurance, epilepsy syndrome, psychiatric disorder, or intellectual disability (intelligence quotient [IQ]â¯<â¯70) for those with "active epilepsy". In assessing transfer of care, we found that only 50% of cases (who had active epilepsy at transition) were being seen by an adult or general neurologist at the time of the exit interview. CONCLUSIONS: Only one-third of young adults with active epilepsy at transition in this community-based study reported having epilepsy care transition discussions with healthcare providers before the age of 18â¯years. Identifying barriers to successful delivery of effective epilepsy transition care is critical to remediating treatment care gaps and building effective future care models.
Subject(s)
Epilepsy/diagnosis , Epilepsy/therapy , Transition to Adult Care/trends , Adolescent , Adult , Age of Onset , Child , Child, Preschool , Cohort Studies , Epilepsy/psychology , Female , Humans , Intellectual Disability/diagnosis , Intellectual Disability/psychology , Intellectual Disability/therapy , Male , Neurologists/trends , Parents/psychology , Patient Transfer/methods , Patient Transfer/trends , Prospective Studies , Young AdultABSTRACT
OBJECTIVES: To characterize and quantify diagnostic and treatment delay among children with infantile spasms, and to estimate the developmental impact of this delay. STUDY DESIGN: In this cohort study, we surveyed the parents of 100 patients with infantile spasms about their experiences with diagnosis and treatment, and ascertained medical and sociodemographic factors potentially related to care of these infants. We specifically determined the latency to first visit an "effective provider," defined as a provider who identified infantile spasms, and prescribed an appropriate first-line treatment, namely adrenocorticotropic hormone, corticosteroids, or vigabatrin. Time to the first visit to an effective provider was evaluated using Cox proportional hazards regression. RESULTS: The median time from the onset of infantile spasms to first visit with an effective provider was 24.5 days. Only 29% of patients were evaluated by an effective provider within 1 week of infantile spasms onset. The time to first effective provider visit was associated with parental language preference, but with no other sociodemographic characteristics. Parents' suspicions that "something is wrong" were often discounted by healthcare providers, and survey respondents frequently reported that pediatricians and neurologists were unfamiliar with infantile spasms. CONCLUSION: This study demonstrates that substantial delay (ie, >1 week) in appropriate care is common, and suggests that the poor awareness of infantile spasms among healthcare providers is at least partly responsible for preventable and potentially significant delays in treatment.
Subject(s)
Delayed Diagnosis/statistics & numerical data , Spasms, Infantile/diagnosis , Adrenal Cortex Hormones/therapeutic use , Adrenocorticotropic Hormone/therapeutic use , Anticonvulsants/therapeutic use , Clinical Competence , Electroencephalography , Female , Follow-Up Studies , Humans , Infant , Los Angeles , Male , Neurology , Parents , Pediatrics , Professional-Family Relations , Proportional Hazards Models , Retrospective Studies , Spasms, Infantile/drug therapy , Tertiary Care Centers , Vigabatrin/therapeutic useABSTRACT
Objective: To develop a novel software method (AR2) for reducing muscle contamination of ictal scalp electroencephalogram (EEG), and validate this method on the basis of its performance in comparison to a commercially available software method (AR1) to accurately depict seizure-onset location. Methods: A blinded investigation used 23 EEG recordings of seizures from 8 patients. Each recording was uninterpretable with digital filtering because of muscle artifact and processed using AR1 and AR2 and reviewed by 26 EEG specialists. EEG readers assessed seizure-onset time, lateralization, and region, and specified confidence for each determination. The two methods were validated on the basis of the number of readers able to render assignments, confidence, the intra-class correlation (ICC), and agreement with other clinical findings. Results: Among the 23 seizures, two-thirds of the readers were able to delineate seizure-onset time in 10 of 23 using AR1, and 15 of 23 using AR2 (p<0.01). Fewer readers could lateralize seizure-onset (p<0.05). The confidence measures of the assignments were low (probable-unlikely), but increased using AR2 (p<0.05). The ICC for identifying the time of seizure-onset was 0.15 (95% confidence interval (CI), 0.11-0.18) using AR1 and 0.26 (95% CI 0.21-0.30) using AR2. The EEG interpretations were often consistent with behavioral, neurophysiological, and neuro-radiological findings, with left sided assignments correct in 95.9% (CI 85.7-98.9%, n=4) of cases using AR2, and 91.9% (77.0-97.5%) (n=4) of cases using AR1. Conclusions: EEG artifact reduction methods for localizing seizure-onset does not result in high rates of interpretability, reader confidence, and inter-reader agreement. However, the assignments by groups of readers are often congruent with other clinical data. Utilization of the AR2 software method may improve the validity of ictal EEG artifact reduction.
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OBJECTIVE: We aimed to compare long-term social outcomes in young adults with childhood-onset epilepsy (cases) with neurologically normal sibling controls. METHODS: Long-term social outcomes were assessed at the 15-year follow-up of the Connecticut Study of Epilepsy, a community-based prospective cohort study of children with newly diagnosed epilepsy. Young adults with childhood-onset epilepsy with complicated (abnormal neurologic exam findings, abnormal brain imaging with lesion referable to epilepsy, intellectual disability (ID; IQ < 60) or informative history of neurologic insults to which the occurrence of epilepsy might be attributed), and uncomplicated epilepsy presentations were compared to healthy sibling controls. Age, gender, and matched-pair adjusted generalized linear models stratified by complicated epilepsy and 5-year seizure-free status estimated adjusted odds ratios (aORs) and 95% confidence intervals [CIs] for each outcome. RESULTS: The 15-year follow-up included 361 individuals with epilepsy (59% of initial cases; N = 291 uncomplicated and N = 70 complicated epilepsy; mean age 22 years [standard deviation, SD 3.5]; mean epilepsy onset 6.2 years [SD 3.9]) and 173 controls. Social outcomes for cases with uncomplicated epilepsy with ≥5 years terminal remission were comparable to controls; cases with uncomplicated epilepsy <5 years seizure-free were more likely to be less productive (school/employment < 20 h/week) (aOR 3.63, 95% CI 1.83-7.20) and not to have a driver's license (aOR 6.25, 95% CI 2.85-13.72). Complicated cases with epilepsy <5 years seizure-free had worse outcomes across multiple domains; including not graduating high school (aOR 24.97, 95% CI 7.49-83.30), being un- or underemployed (<20 h/week) (aOR 11.06, 95% CI 4.44-27.57), being less productively engaged (aOR 15.71, 95% CI 6.88-35.88), and not living independently (aOR 10.24, 95% CI 3.98-26.36). Complicated cases without ID (N = 36) had worse outcomes with respect to productive engagement (aOR 6.02; 95% CI 2.48-14.58) compared to controls. Cases with complicated epilepsy were less likely to be driving compared to controls, irrespective of remission status or ID. SIGNIFICANCE: In individuals with uncomplicated childhood-onset epilepsy presentations and 5-year terminal remission, young adult social outcomes are comparable to those of sibling controls. Complicated epilepsy, notable for intellectual disability, and seizure remission status are important prognostic indicators for long-term young adult social outcomes in childhood-onset epilepsy.
Subject(s)
Epilepsy/diagnosis , Epilepsy/psychology , Siblings/psychology , Activities of Daily Living/classification , Activities of Daily Living/psychology , Age of Onset , Brain/pathology , Brain Damage, Chronic/diagnosis , Brain Damage, Chronic/psychology , Case-Control Studies , Child , Cohort Studies , Connecticut , Disability Evaluation , Epilepsy/complications , Epilepsy/etiology , Female , Follow-Up Studies , Humans , Intellectual Disability/diagnosis , Intellectual Disability/psychology , Magnetic Resonance Imaging , Male , Neurologic Examination , Outcome Assessment, Health Care , Prognosis , Prospective Studies , Young AdultABSTRACT
The age-adjusted prevalence of seizure disorder in United States veterans deployed in Iraq and Afghanistan conflicts (IAV) is 6.1 per 1,000 persons (1), compared with 7.1 to 10 per 1,000 persons in the general population (2,3). Persons with epilepsy are at risk of excess mortality in part because of comorbidity (4). Although patterns of comorbidity have been associated with mortality in IAV (5), the unique contribution of epilepsy to excess mortality in IAV is unknown. A cohort study was developed using inpatient, outpatient, and pharmacy data from the U.S. Department of Veterans Affairs, Veterans Health Administration (VA) to identify epilepsy, demographic characteristics, and baseline comorbidity for IAV who received VA care in 2010 and 2011. The VA's vital status records were used to identify 5-year mortality (2011-2015). The unadjusted Kaplan-Meier estimator and adjusted proportional hazards regression models tested the hypothesis that excess mortality is associated with epilepsy. IAV with epilepsy were more likely than those without epilepsy to have mental and physical comorbidity, and significantly higher mortality, even after controlling for demographic characteristics and other comorbid conditions (adjusted hazard ratio = 2.6; 95% confidence interval [CI] 2.1-3.2). IAV with epilepsy could benefit from evidence-based chronic disease self-management programs to reduce physical and psychiatric comorbidity, and linkages to VA clinical and other community health and social service providers.
Subject(s)
Afghan Campaign 2001- , Epilepsy/epidemiology , Iraq War, 2003-2011 , Veterans/statistics & numerical data , Adolescent , Adult , Cohort Studies , Comorbidity , Female , Humans , Male , Middle Aged , Prevalence , United States/epidemiology , United States Department of Veterans Affairs , Young AdultABSTRACT
BACKGROUND: The management of drug-resistant epilepsy in children with Tuberous Sclerosis Complex (TSC) is challenging because of the multitude of treatment options, wide range of associated costs, and uncertainty of seizure outcomes. The most cost-effective approach for children whose epilepsy has failed to improve with first-line medical therapy is uncertain. METHODS: A review of MEDLINE from 1990 to 2015 was conducted. A cost-utility analysis, from a third-party payer perspective, was performed for children with drug-resistant epilepsy that had failed to improve with 2 antiseizure drugs (ASDs) and that was amenable to resective epilepsy surgery, across a time-horizon of 5years. Four strategies were included: (1) resective epilepsy surgery, (2) vagus nerve stimulator (VNS) implantation, (3) ketogenic diet, and (4) addition of a third ASD (specifically, carbamazepine). The incremental cost per quality-adjusted life year (QALY) gained was analyzed. RESULTS: Given a willingness-to-pay (WTP) of $100,000 per QALY, the addition of a third ASD ($6600 for a gain of 4.14 QALYs) was the most cost-effective treatment strategy. In a secondary analysis, if the child whose epilepsy had failed to improve with 3 ASDs, ketogenic diet, addition of a fourth ASD, and resective epilepsy surgery were incrementally cost-effective treatment strategies. Vagus nerve stimulator implantation was more expensive yet less effective than alternative strategies and should not be prioritized. CONCLUSIONS: The addition of a third ASD is a universally cost-effective treatment option in the management of children with drug-resistant epilepsy that has failed to improve with 2 ASDs. For children whose epilepsy has failed to improve with 3 ASDs, the most cost-effective treatment depends on the health-care resources available reflected by the WTP.
Subject(s)
Diet, Ketogenic/economics , Drug Resistant Epilepsy/therapy , Health Care Costs , Tuberous Sclerosis/complications , Vagus Nerve Stimulation/economics , Anticonvulsants/economics , Anticonvulsants/therapeutic use , Carbamazepine/economics , Carbamazepine/therapeutic use , Child , Cost-Benefit Analysis , Drug Resistant Epilepsy/economics , Drug Resistant Epilepsy/etiology , Drug Resistant Epilepsy/surgery , Humans , Retrospective Studies , Social Behavior , Treatment OutcomeABSTRACT
OBJECTIVE: Children with medically refractory epilepsy stand to benefit from surgery and live a life free of seizures. However, a large proportion of potentially eligible children do not receive a timely referral for a surgical evaluation. We aimed to describe experiences during the arduous time before the referral and the parent-reported facilitators that helped them move forward through this slow time. METHODS: Individual semi-structured interviews with 37 parents of children who had previously undergone epilepsy surgery at UCLA (2006-2011) were recorded, transcribed, and systematically analyzed by two independent coders using thematic analysis. Clinical data were extracted from medical records. RESULTS: Parents, 41.3years of age on average, were mostly Caucasian, English-speaking, mothers, married, and employed. The mean age at surgery for children was 8.2years with a mean time from epilepsy onset to surgery of 5.4years. Parental decision-making was facilitated when parents eventually received a presurgical referral and navigated to a multidisciplinary team that they trusted to care for their child with medically refractory epilepsy. Four themes described the experiences that parents used to feel a sense of moving forward. The first theme, processing, involved working through feelings and was mostly done alone. The second theme, navigating the complex unknowns of the health-care system, was more active and purposeful. Processing co-occurred with navigating in a fluid intersection, the third theme, which was evidenced by deliberate actions. The fourth theme, facilitators, explained helpful ways of processing and navigating; parents utilized these mechanisms to turn vulnerable times following the distress of their child's diagnosis into an experience of productivity. SIGNIFICANCE: To limit parental distress and remediate the slow and arduous journey to multidisciplinary care at a comprehensive epilepsy center for a surgical evaluation, we suggest multi-pronged interventions to modify barriers associated with parents, providers, and health-care systems. Based on the facilitators that moved parents of our sample forward, we provide practical suggestions such as increased peer support, developing the role of patient navigators and communication strategies with parents before, during, and after referral to a comprehensive epilepsy center and presurgical evaluation.
Subject(s)
Brain/surgery , Decision Making , Epilepsy/surgery , Neurosurgical Procedures , Parents , Referral and Consultation , Adolescent , Child , Child, Preschool , Communication , Female , Humans , MaleABSTRACT
BACKGROUND: Adults with childhood-onset epilepsy experience poorer adult social outcomes than their peers. The relative roles of seizures over time versus learning and psychiatric problems are unclear. METHODS: We examined independent influences of psychiatric and learning disorders and of seizure course in 241 young adults (22-35 years old) with uncomplicated epilepsy in a longitudinal community-based cohort study. Social outcomes were ascertained throughout the study. A history of psychiatric and learning problems was ascertained â¼9 years after study entry. Seizure course was: "Excellent," no seizures after the first year, in complete remission at last contact (N = 95, 39%); "Good," seizures occurred 1 to 5 years after diagnosis, in complete remission at last contact (N = 56, 23%); "Fluctuating," more complicated trajectories, but never pharmacoresistant (N = 70, 29%); "Pharmacoresistant," long-term pharmacoresistant (N = 20, 8%). Multiple logistic regression was used to identify contributors to each social outcome. RESULTS: Better seizure course predicted college completion, being either employed or pursuing a degree, and driving, but was not substantially associated with other social outcomes. Poorer seizure course was associated with a greater likelihood of having offspring, particularly in women without partners. Learning problems, psychiatric disorders, or both negatively influenced all but 2 of the social outcomes. CONCLUSIONS: In young adults with uncomplicated epilepsy, the course of seizures contributed primarily to education, employment, and driving. A history of learning problems and psychiatric disorders adversely influenced most adult outcomes. These findings identify potential reasons for vocational and social difficulties encountered by young adults with childhood epilepsy and areas to target for counseling and transition planning.