This phase 1 trial investigated a personalized neoantigen DNA vaccine (GNOS-PV01) in 9 newly diagnosed MGMT-unmethylated glioblastoma patients. The vaccine was individually designed for each patient using up to 40 tumor-specific neoantigen targets (more than most prior GBM vaccine approaches). No serious treatment-related adverse events were reported. Vaccine-induced immune responses were seen in 8 of 9 patients, and the only non-responder was on steroids. Three of the 9 patients survived beyond 2 years, and 1 patient remains recurrence-free nearly 5 years after diagnosis. The study adds to growing evidence supporting vaccine strategies for GBM and especially supporting multi-target neoantigen vaccines. The Musella Foundation continues working to support the advancement of promising investigational vaccine approaches for brain tumors, with the strong hope that more patients will gain access to these therapies in the near future.