Herpes simplex virus encephalitis generally has a predilection for the frontal or temporal lobes, but isolated brainstem lesion has been rarely reported. A 32-year-old man was presented with headache, fever, dizziness and dysarthria. On admission, truncal ataxia, dysarthria and psychomotor slowing were observed. Brain MRI showed hyperintensities at pons and left thalamus in T2 weighted images. HSV PCR study was positive for HSV type 1. This patient was impro- ved during the course of anti-viral therapy. Brain lesion at follow-up MRI and neurologic deficits disappeared after 2 weeks.