We present serial MR perfusion and spectroscopic findings of a pathologically proved low grade glioma, which evolved into glioblastoma multiforme in 2 years in a 24-year-old man. The initial MR imaging studies, including enhanced conventional T1-weighted and perfusion imaging, were characteristic of a benign glioma with the only exception being that multi-voxel proton MR spectroscopy showed malignant features with a high choline:phosphocreatine ratio. Postoperative follow-up MR imaging revealed findings consistent with malignant glioma, with increased angiogenesis on perfusion images and heterogeneous enhancement on contrast-enhanced T1-weighted images that were further confirmed by second surgery. We suggest conducting close MR imaging follow-up of patients with glioma who have discrepant MR spectroscopic and perfusion results after treatment.