Anosognosia or lack of illness awareness is a clinical manifestation of both schizophrenia and right hemispheric lesions associated with stroke, neurodegeneration, or traumatic brain injury. It is thought to result from right hemispheric dysfunction or interhemispheric disequilibrium, which provides a neuroanatomical model for illness unawareness in schizophrenia. Lack of insight contributes to medication nonadherence and poor treatment outcomes and is often refractory to pharmacological and psychological interventions. We present the first report of transient illness awareness (<8 hours) after individual bilateral electroconvulsive therapy treatments in the case of a 39-year-old man with antipsychotic refractory schizophrenia. Electroencephalography demonstrated frontal slow wave activity with shifting frontotemporal predominance, which was concurrent with the patient's transient level of insight. A systematic review of the literature on electroconvulsive therapy-induced illness awareness in schizophrenia and psychotic disorders produced zero relevant results. Future research should focus on the prospective role of focal interventions, such as transcranial magnetic stimulation, in the development of a neurophysiological model for anosognosia reversal in schizophrenia that may, in turn, contribute to novel therapeutic developments targeting lack of illness awareness.