We report a 13-year-old female with refractory frontal lobe epilepsy in whom diffusion tensor imaging was useful for exploring subtle cortical malformation. She had frequent simple partial seizures characterized by clonic movement of the right upper extremity. Conventional magnetic resonance imaging was not conclusive. A diffusion tensor imaging revealed an increased apparent diffusion coefficient and reduced anisotropy in the left frontal lobe. Positron emission tomography disclosed hypermetabolism in the adjacent area. We could identify the relatively thick cortex with an indistinct gray-white matter junction in a thin-slice T(1)-weighted image.