We describe the case of a 76-year-old male patient who developed acute manifestations of pseudobulbar palsy without a history of stroke or vascular risk factors as the first manifestation of intravascular lymphomatosis. Neurological symptoms of acute pseudobulbar palsy appeared on the second postoperative day after transurethral prostatectomy for the treatment of prostatic hypertrophy. Intravascular lymphomatosis was diagnosed from microscopic findings and immunohistochemical staining of the surgical specimen. Chemotherapy was started, but the patient died due to sepsis secondary to bone marrow aplasia. Intravascular lymphomatosis should be considered a rare potential cause of acute pseudobulbar palsy.