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. 2024 May 30;6(1):29.
doi: 10.1186/s42466-024-00323-6.

A multicenter, matched case-control analysis comparing burden of illness among patients with tuberous sclerosis complex related epilepsy, generalized idiopathic epilepsy, and focal epilepsy in Germany

Lisa Lappe  1   2 , Christoph Hertzberg  3 , Susanne Knake  4 , Markus Knuf  5   6 , Felix von Podewils  7 , Laurent M Willems  1   2 , Stjepana Kovac  8 , Johann Philipp Zöllner  1   2 , Matthias Sauter  9 , Gerhard Kurlemann  10 , Thomas Mayer  11 , Astrid Bertsche  12 , Klaus Marquard  13 , Sascha Meyer  14 , Hannah Schäfer  15 , Charlotte Thiels  16   17 , Bianca Zukunft  18 , Susanne Schubert-Bast  1   2   19 , Jens-Peter Reese  20 , Felix Rosenow  1   2 , Adam Strzelczyk  21   22   23
Affiliations

A multicenter, matched case-control analysis comparing burden of illness among patients with tuberous sclerosis complex related epilepsy, generalized idiopathic epilepsy, and focal epilepsy in Germany

Lisa Lappe et al. Neurol Res Pract. .

Abstract

Background: Depending on the underlying etiology and epilepsy type, the burden of disease for patients with seizures can vary significantly. This analysis aimed to compare direct and indirect costs and quality of life (QoL) among adults with tuberous sclerosis complex (TSC) related with epilepsy, idiopathic generalized epilepsy (IGE), and focal epilepsy (FE) in Germany.

Methods: Questionnaire responses from 92 patients with TSC and epilepsy were matched by age and gender, with responses from 92 patients with IGE and 92 patients with FE collected in independent studies. Comparisons were made across the main QoL components, direct costs (patient visits, medication usage, medical equipment, diagnostic procedures, ancillary treatments, and transport costs), indirect costs (employment, reduced working hours, missed days), and care level costs.

Results: Across all three cohorts, mean total direct costs (TSC: €7602 [median €2620]; IGE: €1919 [median €446], P < 0.001; FE: €2598 [median €892], P < 0.001) and mean total indirect costs due to lost productivity over 3 months (TSC: €7185 [median €11,925]; IGE: €3599 [median €0], P < 0.001; FE: €5082 [median €2981], P = 0.03) were highest among patients with TSC. The proportion of patients with TSC who were unemployed (60%) was significantly larger than the proportions of patients with IGE (23%, P < 0.001) or FE (34%, P = P < 0.001) who were unemployed. Index scores for the EuroQuol Scale with 5 dimensions and 3 levels were significantly lower for patients with TSC (time-trade-off [TTO]: 0.705, visual analog scale [VAS]: 0.577) than for patients with IGE (TTO: 0.897, VAS: 0.813; P < 0.001) or FE (TTO: 0.879, VAS: 0.769; P < 0.001). Revised Epilepsy Stigma Scale scores were also significantly higher for patients with TSC (3.97) than for patients with IGE (1.48, P < 0.001) or FE (2.45, P < 0.001). Overall Quality of Life in Epilepsy Inventory-31 items scores was significantly lower among patients with TSC (57.7) and FE (57.6) than among patients with IGE (66.6, P = 0.004 in both comparisons). Significant differences between patients with TSC and IGE were also determined for Neurological Disorder Depression Inventory for Epilepsy (TSC: 13.1; IGE: 11.2, P = 0.009) and Liverpool Adverse Events Profile scores (TSC: 42.7; IGE: 37.5, P = 0.017) with higher score and worse results for TSC patients in both questionnaires.

Conclusions: This study is the first to compare patients with TSC, IGE, and FE in Germany and underlines the excessive QoL burden and both direct and indirect cost burdens experienced by patients with TSC.

Keywords: Anti-seizure medication; Anticonvulsants; Costs; Depression; Direct costs; Everolimus; Indirects costs; Quality of life; Seizure; Stigma.

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Conflict of interest statement

LL and none of the other authors report conflicts of interest related to this study.

CH reports personal fees from Desitin Arzneimittel, Eisai, GW Pharmaceuticals companies, Novartis, Shire, and Zogenix.

SKn received speaker´s honoraria from Angelini Pharma, Bial, Desitin Arzneimittel, Eisai, Epliog, GW Pharmaceuticals, Kanso, UCB Pharma, and Zogenix.

MK does not have any COI. He receives no honoraria from pharmaceutical companies.

FvP reports personal fees and grants from Bial, Desitin Arzneimittel, Eisai, GW Pharmaceutical companies, Arvelle Therapeutics, Zogenix, and UCB Pharma.

LW does not have any COI. He receives no honoraria from pharmaceutical companies.

SKo reports speakers honoraria from Eisai, UCB and Jazz Pharma, and research funding from Biogen.

JPZ does not have any COI. He receives no honoraria from pharmaceutical companies.

MS reports personal fees from Novartis and GW Pharmaceuticals companies.

GKu reports honoraria for speaking engagements from Desitin Arzneimittel, Eisai, UCB Pharma, Takeda, Shire, Zogenix, Neuraxpharm, Stada, and GW Pharmaceuticals.

TM does not have any COI. He receives no honoraria from pharmaceutical companies.

AB reports grants from UCB Pharma and honoraria for speaking engagements from Biogen, Desitin Arzneimittel, Eisai GmbH, GW Pharmaceuticals, Neuraxpharm, Shire/Takeda GmbH, UCB Pharma, and ViroPharma.

KM does not have any COI. He receives no honoraria from pharmaceutical companies.

SM reports grants from Novartis, UCB, Shire, Deutsche Forschungsgemeinschaft und Epilepsiestiftung Dr. Wolf.

HS does not have any COI. She receives no honoraria from pharmaceutical companies.

CT does not have any COI. She receives no honoraria from pharmaceutical companies.

BZ does not have any COI. She receives no honoraria from pharmaceutical companies.

SSB reports personal fees from Eisai, Desitin Pharma, GW Pharmaceuticals, Zogenix, UCB Pharma, and Marinus Pharma.

JPR does not have any COI. He receives no honoraria from pharmaceutical companies.

FR reports personal fees from Angelini Pharma, Eisai, Jazz Pharma, and UCB Pharma and grants from the Detlev-Wrobel-Fonds for Epilepsy Research, the Deutsche Forschungsgemeinschaft (DFG), the Federal Ministry of Education and Research (BMBF), the LOEWE Programme of the State of Hesse, and the European Union. FR reports that he is part of the editorial board of Neurological Research and Practice.

AS reports personal fees and grants from Angelini Pharma, Biocodex, Desitin Arzneimittel, Eisai, Jazz Pharmaceuticals, Marinus Pharma, Precisis, Takeda, UCB Pharma, and UNEEG Medical. AS reports that he is part of the editorial board of Neurological Research and Practice.

Figures

Fig. 1
Fig. 1
A. The proportions of patients reporting employment (full-time, part-time/hour reduction, vocational training) in the tuberous sclerosis complex (TSC; missing n = 7), idiopathic generalized epilepsy (IGE), and focal epilepsy (FE, missing n = 2) cohorts and general German population *P < 0.05, **P < 0.01, and ***P < 0.001 between the TSC and IGE cohorts; †P < 0.05, ††P < 0.01, and †††P < 0.001 between the TSC and FE cohorts B. Percentage of patients in each cohort (n = 92) receiving the indicated care level. Using the Chi-square test, significant differences were identified between the proportion of patients with TSC with a care grade and the proportions of patients with IGE (missing n = 1; P < 0.001) and FE (missing n = 3; P < 0.001) with a care grade C. Percentage of patients in each cohort (n = 92) with some degree of disability. Using the Chi-square test, significant differences were identified between the proportion of patients with TSC with a disability card (degree of disability ≥ 50) and the proportions of patients with IGE (P < 0.001) and FE (P < 0.001) with a disability card
Fig. 2
Fig. 2
(A) Proportion of patients taking the indicated anti-seizure medications (ASM) by disease cohort (n = 92 per cohort). * drugs intended only for patients with TSC (B) Proportion of patients from each cohort (n = 92) taking at least one medication belonging to the indicated groups of prescribed drugs. Using the Chi-square test, significant differences were identified between the proportions of patients with tuberous sclerosis complex (TSC) and the proportions of patients with idiopathic generalized epilepsy (IGE; P = 0.006) taking cardiovascular and antithrombotic drugs, between the proportions of patients with TSC and the proportions of patients with IGE (P < 0.001) and focal epilepsy (FE; P < 0.001) taking psycholeptics, and between the proportions of patients with TSC and the proportions of patients with IGE (P = 0.01) and FE (P = 0.03) taking drugs for the alimentary tract and metabolism (C) The proportions of patients from each cohort (n = 92) taking the indicated number of daily anti-seizure medications (ASM)
Fig. 3
Fig. 3
A. Mean EuroQuol-5-Dimension (EQ-5D) component scores by cohort B. Average self-rated EuroQuol Visual Analog Scale (EQ-VAS) score by age group in each disease cohort and general German population
See this image and copyright information in PMC

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