BACKGROUND Patients with end-stage renal disease (ESRD) who require dialysis can develop a variety of skin conditions, such as pruritus, xerosis, skin infections, and autoimmune reactions. Bullous pemphigoid (BP) is an autoimmune bullous disorder with an increasing incidence. It can be caused by over 90 medications, but levofloxacin-induced BP in hemodialysis patients has not yet been reported. This report is of a 27-year-old woman with ESRD on hemodialysis who developed BP after levofloxacin treatment. CASE REPORT A 27-year-old woman with hemodialysis after kidney transplantation failure was started with levofloxacin for suspected urinary tract infection 1.5 months prior to admission. Her urinary tract infection symptoms were improved after 3 weeks of levofloxacin treatment, but a serious rash developed, presenting with progressive bullous throughout the body and facial involvement. A thorough workup showed a remarkably elevated hemidesmosomal antigen, BP180 (116 RU/mL), and cutaneous indirect immunofluorescence on human salt-split skin substrate was positive for serum basement membrane zone IgG with an epidermal pattern. Skin biopsy direct immunofluorescence staining showed continuous linear C3 deposition along the basement membrane zone. Prednisone 60 mg daily was started with a taper schedule. She no longer had new skin rash during a follow-up of over 3 months. CONCLUSIONS To the best of our knowledge, this is the first case of levofloxacin-induced BP in a patient undergoing hemodialysis. This report highlights the importance of recognizing skin reactions associated with ESRD in dialysis patients, the correct diagnosis by biopsy and histopathology, and the correct and timely management.