Work with individuals with lesions to specific brain regions has long been used to test or even generate theories regarding the neural systems that support specific cognitive processes. Work with individuals who have neuropsychiatric disorders that also involve neurobiological disturbances may be able to play a similar role in theory testing and building. For example, schizophrenia is a psychiatric disorder thought to involve a range of neurobiological disturbances. Further, individuals with schizophrenia are known to suffer from deficits in working memory, meaning that examining the work on the neurobiology of working memory deficits in schizophrenia may help to further our understanding of the cognitive neuroscience of working memory. This article discusses the pros and cons of extrapolating from work in schizophrenia to models of healthy working memory function, and reviews the literature on working memory function in schizophrenia in relationship to existing human and non-human primate models of the cognitive neuroscience of working memory.