Neuroimaging studies of obsessive-compulsive disorder (OCD) often focus on the cortico-striato-thalamo-cortical (CSTC) circuitry, but recent studies have found abnormal spontaneous brain activity in regions outside the CSTC circuitry in patients with OCD using resting-state functional magnetic resonance imaging. Researchers have not clearly determined whether changes in spontaneous brain activity within and beyond the CSTC circuitry coexist in medication-free patients with OCD. To address this question, we recruited 64 medication-free patients with OCD and 60 matched healthy controls (HCs) to investigate their spontaneous brain activity by measuring the fractional amplitude of low-frequency fluctuation (fALFF) and resting-state functional connectivity. Patients with OCD showed increased fALFF values in the left dorsolateral prefrontal cortex (DLPFC) and decreased fALFF values in the right rolandic operculum compared with HCs. Furthermore, patients with OCD exhibited significantly increased functional connectivity between the left DLPFC and the left cerebellum and reduced negative functional connectivity between the right rolandic operculum and the left precuneus. These findings provided new insights into the pathophysiological model of OCD, which may include CSTC circuitry and regions outside this circuitry.