We aim to describe a case of a pregnant woman with drug-resistant genetic generalized epilepsy (GGE) managed to term with dual-device neuromodulation—vagus nerve stimulation (VNS) and centromedian nucleus deep brain stimulation (CM-DBS)—as the sole anti-seizure therapy.

Neuromodulation with VNS and DBS are FDA-approved treatment options for drug-resistant focal epilepsy and are also used off-label in generalized epilepsy. The centromedian nucleus (CM) is a frequent DBS target in generalized epilepsy. While each modality has demonstrated safety and efficacy, including during pregnancy when combined with antiseizure medications (ASMs), the use of dual-device neuromodulation as the exclusive anti-seizure treatment in pregnancy has not previously been reported.

A woman of childbearing age with drug-resistant GGE who previously failed multiple ASMs due to inefficacy or adverse effects underwent sequential implantation of VNS and CM-DBS for seizure control and conceived one year after CM-DBS implantation. Before neuromodulation, she experienced generalized tonic–clonic (GTC) seizures every two weeks. Device parameters were maintained unchanged throughout pregnancy. VNS reduced seizure frequency to approximately one per month, and subsequent CM-DBS further reduced seizures to once every six weeks. During pregnancy, she experienced one GTC seizure in the first trimester and remained seizure-free thereafter until three months postpartum, when one breakthrough GTC occurred. She delivered a healthy female infant at 39 weeks (birth weight 5 lbs 12 oz, small for gestational age) with normal APGAR scores and no congenital abnormalities.

This case highlights the safety of dual-device neuromodulation as primary anti-seizure therapy with VNS and CM-DBS in treating drug resistant GGE in pregnant women with epilepsy (PWWE).  Given the increasing use of neuromodulation for treating drug resistant epilepsy and concerns about fetal effects of ASMs, dual device neuromodulation presents another option and can be considered on a case-by-case basis to treat PWWE.