Background This review aimed to 1) identify and assess the quality of discrete choice experiments (DCEs) examining preferences related to epilepsy treatment; 2) summarize the attributes and attribute levels measured in these studies; 3) identify how researchers selected and developed these attributes; and 4) identify which attributes are most important for epilepsy patients. Methods A systematic literature review using PubMed, Web of Science and Scopus databases from database inception to February or April 2022. We included primary discrete-choice experiments eliciting preferences for various attributes of pharmacological and surgical interventions in patients diagnosed with epilepsy or the parents/carers of children with epilepsy. We excluded non- primary studies, studies assessing preferences for nonpharmacological treatment and studies that elicit preferences using methods other than discrete choice experiments. Two authors independently selected studies, extracted data and assessed risk of bias of studies. The quality of the included studies was assessed using two validated checklists. Study characteristics and findings were summarized descriptively. Results A total of seven studies were included in the review. The majority of studies explored patients’ preferences, and two compared the preferences of patients with physicians. The majority (n = 6) compared two medications, and one compared two surgical options to continuing medication options. The studies examined 44 attributes in total, including side effects (n = 26), efficacy expressed as being seizure free or have fewer seizures (n = 8), costs (n = 3), dosing frequency (n = 3), duration of side effects (n = 2), mortality (n = 1), long-term problems after surgery (n = 1) and surgical options (n = 1). The findings indicate that people with epilepsy have strong preferences for improving seizure control, which was ranked as the top priority in all studies. Patients also have a strong preference for the reduction of adverse effects and may be willing to make trade-offs between improved seizure control and reduction of long-term side effects that may impact their quality of life. Conclusions The use of DCEs in measuring patients’ preference for epilepsy treatment is accumulating. However, inadequate reporting of methodological details may reduce decision-makers’ confidence in the findings. Suggestions for future research are provided.