Abstract
To systematically identify and critically appraise all published full economic evaluations assessing the cost-effectiveness of non-pharmacological interventions for patient with drug-resistant epilepsy. PICOS was used to design search strategies for the identification and selection of relevant studies. Literature search was performed using MEDLINE (via PubMed), EMBASE, INAHTA, EHS EED, and CEA Registry databases to identify articles published between January 2000 to May 2023. Web of Science was additionally used to perform forward and backward referencing. Title-, abstract- and full-text screening was performed by two independent researchers. The Consensus Health Economic Criteria (CHEC) checklist and Consolidated Health Economic Evaluation Reporting Standards (CHEERS) 2022 were applied for quality assessment. A total of 4,470 studies were identified of which 18 met our inclusion criteria. Twelve of the studies conducted model-based economic evaluation and others were trial-based. Three studies showed that epilepsy surgery was cost-effective in adults, while this remained inconclusive for children (two positive, three negative). Three studies showed negative economic outcome for ketogenic diet in children. One out of four studies showed positive results for self-management. For vagus nerve stimulation, one study showed positive results in adults and another one negative result in children. One recent study showed cost-effectiveness of RNS in adults. Finally, one study shows promising but inconclusive results for deep brain stimulation. Mean risk of bias assessment score (based on CHEC) was 95.3% and 80.9% for reporting quality (CHEERS 2022). This review identified studies that assessed the cost-effectiveness of non-pharmacological treatments in both adults and children with drug-resistant epilepsy, suggesting that in adults, epilepsy surgery, VNS and RNS are cost-effective, and that DBS and self-management appear to be promising. In children, epilepsy surgery seems to be cost-effective. Contrary, the use of ketogenic diet was shown not to be cost-effective. However, limited long-term data was available for newer interventions (i.e., ketogenic diet, DBS, and RNS).
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