|Development of myoclonus can be manifestated as a side effect of antiepileptic drugs in subjects with preexisting epilepsy, post-traumatic brain injury, encephalopathy, and focal and multifocal brain lesions. A 69-year-old male showed newly onset severe myoclonus and confusion 2 days after taking 1200 mg of gabapentin. He had end-stage renal disease secondary to type 2 diabetes and was on hemodialysis twice a week. After increasing hemodialyis 3 times a week and discontinuing gabapentin, myoclonus spontaneously resolved. Here we first report a case of myoclonus in a Korean subject with diabetic renal failure. We recommend that administration of gabapentin for diabetic subjects with renal disease should be cautious.