Case Report: Successful treatment of drug-resistant epilepsy with unilateral deep brain stimulation of the anterior nucleus of the thalamus.
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BACKGROUND: Deep brain stimulation of the anterior nucleus of the thalamus (ANT-DBS) is an established palliative treatment for drug-resistant epilepsy (DRE) and is conventionally performed bilaterally. However, the efficacy of unilateral ANT-DBS remains unclear. We report a patient with DRE who underwent unilateral ANT-DBS because of an epileptogenic network predominantly involving one side and anatomical constraints precluding electrode implantation on the contralateral side. METHODS: A 29-year-old female underwent comprehensive presurgical evaluation, including seizure semiology assessment, video-electroencephalography (EEG), computed tomography, and magnetic resonance imaging. She developed DRE following multimodal treatment for a suprasellar germ cell tumor during childhood. Despite multiple antiseizure medications and vagus nerve stimulation, she continued to experience approximately 10 habitual seizures per day. RESULTS: Seizure semiology was characterized by asymmetric tonic posturing of the limbs lasting 10-30 s, followed by an atonic phase. EEG findings indicated a widespread epileptogenic network predominantly involving the right frontal and temporal areas. Brain imaging showed severe left hemispheric atrophy, marked widening of the left subdural space, and a pre-existing shunt system, which prevented identification of the left ANT. Therefore, DBS implantation was performed in the right ANT alone. Stimulation was initiated 7 days after surgery at an intensity of 2 mA. Following initiation of right ANT stimulation, seizure frequency decreased from approximately 10 to one per day, likely due to a microlesioning effect. Thereafter, right ANT stimulation was associated with a sustained seizure reduction of approximately 60%. Postoperative EEG demonstrated a reduction in interictal epileptiform discharges. The clinical benefit was maintained at the 9-month follow-up, with no surgery- or stimulation-related adverse events were observed. CONCLUSION: Unilateral ANT-DBS can provide clinically meaningful seizure control in carefully selected patients whose predominant epileptogenic network is localized to one cerebral hemisphere. This approach may represent a feasible therapeutic option when anatomical or surgical constraints prevent conventional bilateral ANT electrode implantation. Further studies with larger patient cohorts and longer follow-up periods are required to clarify the indications, durability, and efficacy of unilateral ANT-DBS.