Information regarding new-onset posttransplant psychotic disorders and their effect on nonadherence and posttransplant outcome is quite limited. We report a case of new-onset posttransplant schizophrenia that led to death. The patient, a woman with Wilson disease but no history of psychiatric problems or a substance use disorder had undergone liver transplantation at age 21. She married subsequently and bore children, being well able to handle her housework, child care, and full-time employment. She continued her medications as prescribed, and good graft function was maintained. At age 41, she experienced an episode of schizophrenia, then graft loss associated with nonadherence to immunosuppressive agents. Death ensued, occurring 6 months after the onset of schizophrenia. This case highlights the possibility that schizophrenia manifesting long after liver transplantation can result in graft loss and death due to medication nonadherence. Thus, awareness of the possibility of this rare clinical scenario is critical.