Abstract
Willems LM, Hochbaum M, Frey K, Schulz J, Menzler K, Langenbruch L, Kovac S, Immisch I, von Podewils F, Hamacher M, Siebenbrodt K, Rosenow F, Reese JP, Strzelczyk A. Epilepsia. 2022;63(4):904-918. doi:10.1111/epi.17174. PMID: 35192210. This study was undertaken to quantify epilepsy-related costs of illness (COI) in Germany and identify cost-driving factors. COI were calculated among adults with epilepsy of different etiologies and severities. Multiple regression analysis was applied to determine any epilepsy-related and sociodemographic factors that serve as cost-driving factors. In total, 486 patients were included, with a mean age of 40.5 ± 15.5 years (range = 18-83 years, 58.2% women). Mean 3-month COI were estimated at €4911, €2782, and €2598 for focal, genetic generalized, and unclassified epilepsy, respectively. The mean COI for patients with drug-refractory epilepsy (DRE; €7850) were higher than those for patients with non-DRE (€4720), patients with occasional seizures (€3596), or patients with seizures in remission for >1 year (€2409). Identified cost-driving factors for total COI included relevant disability (unstandardized regression coefficient b = €2218), poorer education (b = €2114), living alone (b = €2612), DRE (b = €1831), and frequent seizures (b = €2385). Younger age groups of 18-24 years (b = −€2945) and 25-34 years (b = −€1418) were found to have lower overall expenditures. A relevant disability (b = €441), DRE (b = €1253), frequent seizures (b = €735), and the need for specialized daycare (b = €749) were associated with higher direct COI, and poorer education (b = €1969), living alone (b = €2612), the presence of a relevant disability (b = €1809), DRE (b = €1831), and frequent seizures (b = €2385) were associated with higher indirect COI. This analysis provides up-to-date COI data for use in further health economics analyses, highlighting the high economic impacts associated with disease severity, disability, and disease-related loss of productivity among adult patients with epilepsy. The identified cost drivers could be used as therapeutic and socioeconomic targets for future cost-containment strategies.Objective:
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Commentary
Given its chronic, unpredictable, and commonly debilitating nature, epilepsy takes a substantial toll on its bearers, their caregivers, and the society as whole. 1,2 Beyond its physical and emotional repercussions, epilepsy is associated with a substantial financial burden. To quantify that cost of illness (COI), a whole field of epilepsy research, that of health economics, has emerged over the past decades. By means of either willingness-to-pay methodology or, more commonly, through the human capital method, epilepsy economics focus on the direct cost (i.e., medical and nonmedical, patient- and/or family-related expenses related to disease prevention, diagnosis, treatment, and rehabilitation) and indirect cost (i.e., lost productivity at home and work), as well as the intangibles (i.e., psychosocial cost, unrelated to resource use of productivity). 3 Cost estimations are typically derived from self-reported or medical chart and billing data from patients and providers, or through hypothetical modelling. 4
The current study enriches our knowledge in the field of health economics in epilepsy. By analyzing data on adult patients with epilepsy attending 4 different epilepsy centers in Germany, this manuscript 5 aimed to characterize epilepsy-related COI and identify cost-driving factors that could be used as therapeutic and socioeconomic targets for future cost-containment strategies. This is a cross-sectional, survey-based study of 486 patients conducted over 3 months at the end of 2020. Utilizing a previously validated questionnaire, patients (or their caregivers when physically and intellectually disabled) provided detailed direct and indirect COI information, along with their sociodemographic and disease-related characteristics. Disease-related classifications were validated through medical records. Direct cost estimations were performed through nationwide anti-seizure medications (ASMs) prescription reports, Diagnosis Related Groups catalogues and valid national benchmarks. Indirect cost estimations were conducted using national retirement age standards and federal gross income statistics. Univariate and multivariate regression analysis was applied to identify drivers of the aggregate, direct, and indirect cost. It was found that epilepsy carries high economic impact, predominantly in indirect cost, particularly in older and drug-resistant patients with lower educational attainment, poorer social support, and higher daycare needs, experiencing frequent seizures and greater disability burden.
The main strength of this study is the provision of up to date, “bottom-up,” health economics data in epilepsy from multiple centers operating under a federal welfare system. The authors are experienced investigators in health economics with prolific research contributions in the field. A comprehensive, previously validated approach was deployed that allowed for stratification of prospectively recruited participants based on detailed, sociodemographic and reliably ascertained, disease-related data. A granular analysis of several aspects of direct and indirect cost was performed and determinants were sought in quest for actionable intervention targets.
On the other hand, this study was susceptible to limitations inherent to health economics research, most of which were already acknowledged by its authors. Despite its broad geographic representation, the study focused on community-dwelling participants and may have missed institutionalized patients who carry higher financial burden. On the other hand, the participants were adults who attended specialized epilepsy centers with a large portion manifesting drug-resistant epilepsy on polypharmacy; hence the results may not be fully generalizable to the overall population in Germany, to other high-income countries where the private health sector dominates, or to low/middle income countries with meager resources. Akin to any survey-based study, the results are subject to response, social desirability, and recall bias. The cross-sectional nature of the study prevented longitudinal evaluations or inference of causation, and its timing around the COVID-19 pandemic may have skewed the collected data. The impact of psychosocial comorbidities and any intangible cost related to them was not investigated. Caregiver related indirect cost was not incorporated in the overall financial impact. Despite all good intents, it is questionable how accurately the distinction between strictly epilepsy-related cost versus epilepsy culprits-related cost was performed. Although numerous potential determinants of cost were thoroughly investigated, they only accounted for a small proportion of the variance in the multiple regression models and colinearity was not explicitly accounted for. Moreover, important epilepsy severity variables, such as the type of seizures (i.e., with or without impairment of awareness and convulsions), were not reported. Absence of a control group prevented direct comparison of the financial cost of epilepsy compared to other chronic debilitating disorders, though this was admittedly beyond the scope of the study. Finally, the lack of a cost-benefit analysis between the examined diagnostic and treatment variables limits the ability to prioritize on specific interventions, despite the identified associations.
These limitations notwithstanding the current study delivers several important take-home messages: (1) epilepsy is related with substantial financial expenditure; (2) at least in adults, that burden is higher with regard to indirect costs from lost productivity than the actual direct costs related to its diagnosis and treatment; (3) severely afflicted patients with poorer support are particularly vulnerable. Allowing for temporal and contextual variations, published work from the same group extends such observations across the age spectrum 6 and highlights the incremental economic impact of the disease using similar study design and setting. 7 Within the confines of methodological heterogeneity, these results are in line with prior literature on health economics in epilepsy around the world, 8 as compared to other, prevalent, chronic, neurologic, and nonneurologic conditions. 9 The situation is direr in developing countries, where 80% of the global epilepsy burden is concentrated. 2
That inevitably begs the question: What can we do as epilepsy community to address this economic toll of epilepsy on all stakeholders? What is the most cost-efficient way to screen for epilepsy and its comorbidities, educate the public and the medical community, and secure early referrals to specialists? Factoring in the increasing familiarity with tele-medicine and the inherent transportation challenges of our patients, what is the best longitudinal care model? In the acute setting, what is the value of continuous electroencephalography monitoring to diagnose status epilepticus in all patients at risk and the relative impact of the various treatment options to treat it? What is the cost-benefit ratio of older versus newer ASMs and generic formulations, regionally and worldwide? How can we make the best use of the promising but costly, diagnostic and treatment tools (e.g., minimally invasive surgical techniques and neuromodulation modalities) that are constantly being added to our armamentarium?
In a volatile public health environment recently exposed to an unprecedented pandemic, with rapidly evolving technological advances and escalating health care expenses, there is no easy answer. It is paramount though to keep the patient, the caregiver, the provider, and the society in mind and maintain a global perspective, in order to make decisions that balance the pressures for cost containment and judicious resource allocations with service quality and protection of those in need. 10 Economic appraisal of epilepsy care is an invaluable tool to achieve these goals. Longitudinal, controlled, generalizable, cost-effectiveness, and cost-utility studies can provide evidence-based guidance to clinical epileptologists, hospital administrators, insurance carriers, research agencies, advocacy foundations, and governmental policy makers. 3 In other words, money matters in epilepsy 4 to bridge what is ethically desirable and what is economically realizable. 10
