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In this article we report the case of a man with congenital liver disease who later developed psychotic illness and was diagnosed with schizophrenia. We illustrate how decompensation in liver function was associated with the exacerbation of psychotic symptoms. We discuss differential diagnostic challenges, and the possible overlapping neuropathology in these two conditions that may converge on glutamate/N-methyl-D-aspartate dysfunction. This patient’s case underscores the need for further research to elucidate the possible underlying mechanisms linking congenital liver disease and psychosis. [ABSTRACT FROM AUTHOR]