Antiseizure medications are the primary treatment for epilepsy. However, for individuals with drug-resistant epilepsy, surgical resection is often the treatment of choice. When surgical resection is not feasible due to poorly localized seizure focus or involvement of eloquent regions, deep brain stimulation targeting the anterior nucleus of the thalamus (ANT-DBS) offers an alternative therapeutic approach. Despite its effectiveness, there are challenges in optimizing lead placement. Implantation approaches for ANT-DBS include transventricular and extraventricular trajectories. Although the transventricular approach is commonly used, some surgeons prefer extraventricular approaches to avoid the risks associated with the transventricular approach, such as hemorrhage and cerebrospinal fluid contact. Additionally, extraventricular approaches might be advantageous for patients with previous lobectomies and those requiring multiple leads. This chapter reviews the current knowledge on the clinical efficacy and safety of extraventricular ANT-DBS, providing insights into its potential and advantages over transventricular ANT-DBS.