The cavum septum pellucidum (CSP), a putative marker of neurodevelopmental anomaly, has been associated with an increased risk of several psychiatric disorders. The purpose of this study was to evaluate the CSP in patients with obsessive-compulsive disorder (OCD) compared with healthy control subjects. Seventy-one patients with OCD and 71 healthy volunteers matched for age and sex were evaluated with magnetic resonance imaging. We evaluated the CSP using criteria employed in previous studies: presence of the CSP, length of the CSP, and overall size of the CSP, measured in five grades, ranging from grades 0 (no CSP) to 4 (severe CSP). We evaluated OCD symptom severity using the Yale-Brown Obsessive-Compulsive Scale (Y-BOCS). The CSP presence was significantly greater in the OCD group (60.6%) than in control subjects (29.6%), and CSP size grade was significantly larger in the OCD group (chi(2) = 15.609, P = 0.004). CSP length showed no significant group difference. Among patients with OCD, those with a CSP had higher scores on the obsession subscale of the Y-BOCS than those without a CSP (Z = -2.358, P = 0.018), while they did not show significant difference from those without a CSP in the compulsion subscale of the Y-BOCS, age, duration of illness, or age at onset. These results indicate that neurodevelopmental alterations in midline structures might contribute to the pathogenesis of OCD.