Rationale: It is rare for uremia patients to have epileptic seizures after eating star fruit, only a dozen cases are reported worldwide. Such patients usually have poor prognoses. Few patients had good prognoses, all of them were treated with expensive renal replacement therapy. At present, there is no report on the addition of drug therapy to these patients based on the initial renal replacement therapy., Patient Concerns: A 67-year-old male patient with star fruit intoxication who had a history of diabetic nephropathy, hypertension, polycystic kidney, and chronic kidney disease in the uremic phase, and regular hemodialysis 3 times a week for 2 years. Initial clinical manifestations include hiccups, vomiting, speech disturbances, delayed reactions, and dizziness, which gradually progress to hearing and visual impairment, seizures, confusion, and coma., Diagnoses: This patient was diagnosed with seizures caused by star fruit intoxication. The experience of eating star fruit and the electroencephalograms can prove our diagnosis., Interventions: We performed intensive renal replacement therapy according to the recommendations in the literature. However, his symptoms did not improve significantly until he received an extra dose of levetiracetam and resumed his previous dialysis schedule., Outcomes: The patient was discharged after 21 days without neurologic sequelae. Five months after discharge, he was readmitted due to poor seizure control., Lessons: To improve the prognosis of these patients and to reduce their financial burden, the use of antiepileptic drugs should be emphasized., Competing Interests: The authors have no conflicts of interest to disclose., (Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.)