Sequential deep brain stimulation of anterior and centromedian nuclei in juvenile myoclonic epilepsy: illustrative case.

backgroundNeuromodulation, including deep brain stimulation (DBS), is a treatment option for patients with medically intractable epilepsy who do not meet candidacy for resective surgery. DBS of the anterior nucleus of the thalamus (ANT) has been FDA approved for focal epilepsy, and stimulation of the centromedian nucleus of the thalamus (CMT) is increasingly being explored for generalized epilepsies including juvenile myoclonic epilepsy (JME). The authors present the case of a patient with JME and medically intractable seizures who underwent bilateral, sequential ANT and CMT DBS implantation. OBSERVATIONS: This case describes a 51-year-old woman with medically intractable JME in whom adequate trials of more than 10 antiseizure medications and prior vagus nerve stimulation failed. She underwent bilateral ANT DBS, which reduced her generalized tonic-clonic seizures but did not improve her myoclonic jerks. Owing to persistent disability from daily myoclonus, she later underwent bilateral CMT DBS. Following CMT stimulation, she experienced rare myoclonic jerks. At 7 months postoperatively, her status remained Engel class IIb with sustained functional improvement. LESSONS: This case highlights the complementary roles of ANT and CMT stimulation in modulating distinct thalamocortical pathways. Although CMT DBS has been described in patients with generalized epilepsy, this case demonstrates a meaningful reduction specifically in disabling myoclonic jerks that previously resulted in falls and dropped objects. https://thejns.org/doi/10.3171/CASE251033.