This is a description of a clinical case of levamisole-induced leukoencephalopathy. A few hours after the single intake of the immunomodulator levamisole (150 mg), the patient developed following cerebral symptoms: febrile fever, dizziness, frequent urination, impaired color vision, divergent strabismus, unsteady gait, severe weakness, increased drowsiness, retardation, delusions. The symptoms progressed towards a full clinical picture for about 2 months. The course of the disease was of a wavy nature: left-sided hemiparesis and the left facial nerve palsy were present at the beginning, with later inversion of symptoms represented by the right-sided hemiparesis and the right facial nerve palsy combined with dysarthria. Magnetic resonance imaging (MRI) revealed multiple foci of demyelination in both hemispheres of the brain that regressed after corticosteroid therapy administration. No abnormalities of the cerebrospinal fluid were detected. No pathology of the fundus was evident. The evoked potentials of the brainstem (auditory and visual) and somatosensory evoked potentials from the hands and feet were within normal limits. Plasmapheresis (3 procedures, each 2 liter, with intravenous administration of 1 g of solu-medrol after each session) was performed in the acute phase, which benefited the decrease in severity of the neurological symptoms. Vascular and metabolic therapy was performed afterwards, with the regression of the neurologic symptoms. No specific therapy was administered after the woman was discharged from the hospital. For a 10-year survey after the acute demyelinating process outbreak there’s a complete regression of neurologic symptoms and positive dynamics of the patient’s MRI. The woman is fully adapted physically, emotionally and socially.