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 WMHs in a patient with treatment resistant OCD. A 35-year-old female, who was married and has a child, diagnosed with obsessive compulsive disorder. Firstly, she washed her hands excessively due to the fear of contamination. Later, different types of obsessions and compulsions were triggered by psychological stress factors related to marital status. She started spending lots of time every day in the bathroom, counting objects, and constantly checking the computer, television, stove, and iron. The patient had taken sertraline 200 mg/day, paroxetine 40 mg/day clomipramine 225 mg/day and atypical antipsychotic augmentation including risperidone, aripiprazole, paliperidone, also cognitive behavioral techniques have been used. Nevertheless, the patient could not control these obsessions and compulsions. Lastly, she was admitted to our outpatient clinic with her husband for the complaints of increased frequency and severity of obsessions-compulsive symptoms, serious functional impairment, fatigue, anhedonia, and low self esteem. She didn’t want to take medicine and want to take electroconvulsive therapy (ECT). Total seven session ECT were performed but her symptoms didn’t significantly decreased. The Yale-Brown Obsessive Compulsive scale score reduced from 36 to 30 (%15 reduction), the Hamilton Depression Rating Scale score reduced from 24 to 15 (%40 reduction). Because of resistance to treatment Magnetic Resonance Imaging (MRI) examination were performed. MRI revealed white matter hyperintensities in left posterior frontal and right parietal regions. This case may represent an evidence of impaired connectivity due to white matter abnormalities in patients with treatment resistant OCD, and thus may serve to further our understanding of white matter deficits in OCD. It may be suggested that disruption in brain networks was associated with pathogenesis of the disorder.