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This study described a rare case of necrotizing fasciitis presenting as neural dysfunction. The patient's serum ammonia level was significantly elevated; however, no liver function impairment was observed. Hyperammonemia is frequently related to the liver's ammonia metabolism failure. In literature reviews, infections also induce critical complications in specific circumstances. Herein, a rare case of Staphylococcus aureus necrotizing fasciitis with a hyperammonemic encephalopathy presentation was offered.