The neural correlate of working memory (WM) impairment in schizophrenia is key to the understanding of the cognitive deficits observed in this disorder. We sought to determine the clinical validity of the dual version n-back paradigm in patients with schizophrenia, and whether schizophrenia patients exhibit altered brain activation patterns compared with healthy controls in this dual version WM measure using functional magnetic resonance imaging. Patients with schizophrenia (n = 20) and healthy controls (n = 24) performed the dual n-back task that consists of both visuospatial and auditory-verbal n-back streams, in which participants were required to monitor and update the contents from these two different inputs simultaneously. Significant positive correlations were found between performance in the dual 2-back condition and another measure of WM capacity and IQ estimates. Moreover, hypoactivation was observed at the right middle frontal gyrus and the posterior parietal regions in schizophrenia participants compared with healthy controls. The right hippocampus was less deactivated in schizophrenia patients compared with healthy controls. Our results support the clinical utility of the dual n-back task in schizophrenia and may have implications for the development of specific cognitive training targeting these impaired neural substrates in relation to WM in patients with schizophrenia.