Neuropsychiatric syndrome is a common and serious clinical manifestation of systemic lupus erythematosus (SLE), occurring in about half of all patients during the course of the disease. We report here a case of obsessive-compulsive symptoms as an unusual manifestation of neuropsychiatric SLE. A 17-year-old girl with SLE was admitted to a medical center with the chief complaint of recent-onset repeated doubt and repetitive checking, which subsided after treatment. She had recurrent and intrusive doubt about losing her belongings and tried to suppress the thoughts by mental acts of praying and repetitive checking. The central nervous system lupus involvement in this patient was confirmed by brain magnetic resonance imaging, which revealed widespread areas of abnormal high signal intensity over the internal capsules and basal ganglia, and focal lesions at the peripheral parenchyma of the frontal and parietal areas. Single photon emission computed tomography also showed decreased perfusion at the left temporal lobe. This supported the hypothesis that basal ganglia abnormalities could be the common pathophysiology of coexisting obsessive-compulsive symptoms and SLE. Moreover, increased awareness of hidden psychopathology, an accepting attitude, and careful probing for obsessive-compulsive symptoms are important while taking care of SLE patients.