PURPOSE: Responsive neurostimulation of the centromedian nucleus of the thalamus (CM RNS) is being investigated for treatment of drug-resistant generalized epilepsy with promising results. The aim of this study is to report outcomes of seven patients with pediatric-onset drug-resistant generalized epilepsy, including both genetic generalized epilepsy (GGE) and Lennox-Gastaut syndrome (LGS), who underwent treatment with bilateral CM RNS. METHODS: A retrospective chart review was performed for patients with drug-resistant generalized epilepsy who underwent treatment with bilateral CM RNS at Children's Hospital of Pittsburgh from 2020 to 2022. Improvement in seizure frequency was obtained through patient and/or caregiver reports on standardized patient questionnaires. The primary outcome measure was percent improvement in seizure frequency at time of last follow-up appointment compared to baseline seizure frequency. RESULTS: Five of the seven patients (71 %) had an average 50 % or greater improvement in seizure frequency among seizure types including four of the five patients (80 %) with GGE and one of the two patients (50 %) with LGS. There were no serious adverse events including post-operative infection, stroke, or device malfunction/migration. CONCLUSION: This data, along with other recent studies, suggests that CM RNS can improve seizure frequency in pediatric-onset drug-resistant generalized epilepsy, but larger systematic studies with longer follow-up times and standardized outcome measures are needed to determine long-term effectiveness and optimal patient selection for thalamic RNS.