A 78-year-old Chinese man with a history of end stage renal disease (ESRD) presented with fever of one day duration. He was treated for catheter related sepsis with intravenous piperacillin and tazobactam, which was later switched to vancomycin and ceftazidime secondary to persistent fever with negative cultures. One the fifth day of treatment with vancomycin and ceftazidime, he developed new onset upper limb myoclonus which progressed to bilateral upper limb ataxia. A provisional diagnosis of myoclonus and ataxia secondary to neurotoxicity related to ceftazidime was made and the ceftazidime was ceased. His symptoms resolved over three days and he returned to his baseline neurological status by Day 5 following cessation.