A 59-year-old Japanese woman presented with symptoms resembling multiple system atrophy, including parkinsonism, after receiving concentrated sodium infusion therapy for hyponatremia during breast cancer treatment 20 years prior. Despite initial suspicions of multiple system atrophy, further examination revealed extrapontine myelinolysis (EPM) as the cause of her symptoms, with bilateral putamen atrophy resembling the former condition. This case highlights the importance of considering EPM in patients with parkinsonism and a history of osmotic demyelination syndromes, even in the absence of brainstem lesions.