Cefazolin-induced neurotoxicity with the documented toxic concentration has not been reported in uremic patients on continuous ambulatory peritoneal dialysis (CAPD). We described an elderly female on CAPD for years presented with newly-onset status epilepticus. Her body weight was 60 kg. And she had received intraperitoneal ceftazidime and cefazolin 1.5 g once daily for her CAPD peritonitis 5 days earlier. She was disoriented but afebrile with normal blood pressure. Laboratory data showed WBC 18,480/uL, pH 6.93, HCO3 - 8.5 mmol/L, free Ca2+ 3.5 mmol/L, and albumin 2.8 g/dL. Although antiepileptic drugs and hypocalcemia correction ceased the seizure, her consciousness remained semi-coma. Image studies of brain were unremarkable. Despite undetectable serum ceftazidime, her cefazolin trough level was 149.5 μg/mL. Emergent hemodialysis rapidly resolved her neurological features accompanied by a markedly declined serum cefazolin concentration (28.6 μg/mL). Higher intraperitoneal cefazolin dosing in patients on CAPD may cause drug-induced neurotoxicity with status epilepticus which could be rapidly corrected by hemodialysis.