AbstractBackgroundMultiple system atrophy with parkinsonism (MSA‐P) is a progressive condition with no effective treatment.ObjectiveThe aim of this study was to describe the safety and efficacy of deep brain stimulation (DBS) of globus pallidus pars interna and externa in a cohort of patients with MSA‐P.MethodsSix patients were included. Changes in Movement Disorders Society Unified Parkinson's Disease Rating Scale Part III (MDS‐UPDRS III), Parkinson's Disease Questionnaire (PDQ‐39) scores, and levodopa equivalent daily dose were compared before and after DBS. Electrode localization and volume tissue activation were calculated.ResultsDBS surgery did not result in any major adverse events or intraoperative complications. Overall, no differences in MDS‐UPDRS III scores were demonstrated (55.2 ± 17.6 preoperatively compared with 67.3 ± 19.2 at 1 year after surgery), although transient improvement in mobility and dyskinesia was reported in some subjects.ConclusionsGlobus pallidus pars interna and externa DBS for patients with MSA‐P did not result in major complications, although it did not provide significant clinical benefit as measured by MDS‐UPDRS III. © 2023 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.