Several case reports have described stories of schizophrenia patients reporting no discomfort in response to several medical conditions which normally elicit pain. Comparatively, experimental studies performed on pain perception in schizophrenia have not documented hypoalgesic responses that are as frank as those reported in these clinical cases. Here, we report the case of a female patient with schizoaffective disorder, who displayed markedly reduced pain perception during an experimental heat pain paradigm. Compared to a large group of healthy volunteers that we tested in 3 previous studies using the same psychophysical procedure, the experimental temperature required to induce moderate pain was radically increased in this patient (z-score = 3.6). The patient had mild psychiatric symptoms and had insight into her symptoms. She had drug-induced dyskinetic symptoms. This case report illustrates that it is possible to observe marked reductions in pain perception in schizophrenia patients tested in experimental settings but that the phenomenon is relatively rare. Regardless of the exact nature of pain indifference in schizophrenia, it can delay diagnosis and treatment of medical problems in these patients. Future studies in the field will need to pay attention to drug-induced extrapyramidal symptoms.