Case Presentation: A 54-year-old male patient presenting probable multiple system atrophy with predominant parkinsonism who underwent bilateral deep brain stimulation (DBS) of the pedunculopontine nucleus (PPN) is presented. The patient had dominant freezing of gait (FOG), levodopa-resistant bradykinesia, and autonomic disturbances, but with a good cognitive condition. Methods: The patient underwent bilateral DBS of the PPN, which ended with modest benefits. Results and Conclusion: Although he had a short postoperative follow-up (6 months), his neurological status remained stable and PPN DBS provided modest improvements in the gait disorder and freezing episodes. This unusual case suggests that the mesencephalic pedunculopontine region may have a role in locomotor symptoms and the potential to provide a limited improvement in FOG.

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