Neurobiological models of obsessive compulsive disorder (OCD) suggest that there are structural and functional abnormalities in frontal-striatal-thalamic-cortical circuits. These cortical and subcortical microcircuits are physically and functionally connected through the white matter. Therefore, the disrupted white matter microstructure may be implicated in the pathophysiology of OCD. Neuroanatomical studies have reported various regional white matter abnormalities in patients with OCD. In this case, we present subcortical white matter lesions or hyperintensities in a patient with treatment resist ant OCD.