We report a patient with medically refractory mesial temporal lobe epilepsy treated by gamma knife radiosurgery. In lieu of a microsurgical procedure, an entorhinoamygdalohippocampectomy was performed with a gamma knife and low marginal doses (25 Gy). The clinical and imaging studies, including CT, MR imaging, 18F-fluorodeoxyglucose positron emission tomography (FDG-PET), and long-term follow-up MR examinations, are reported. The patient has been seizure-free since the day of treatment, with no clinical complications. MR studies accurately depicted the effect on the target structures and the transient secondary changes around them. FDG-PET scans showed decreased metabolism after gamma knife surgery throughout the anteromesial part of the epileptogenic temporal lobe. This metabolic decrease was reversible in the lateral temporal cortex. Our case suggests that gamma knife surgery is a promising tool for use as a minimally invasive approach to the treatment of epilepsy.