The adverse effects of intravesical epirubicin (EPI) instillation are usually minor. Herein, we report on the first case of a patient who received intravesical EPI instillation complicated with anaphylactic shock. A 69-year-old male patient with transitional cell carcinoma (TCC) of the urinary bladder underwent transurethral resection of a bladder tumor (TURBT) 4 times and 4 complete courses of adjuvant intravesical bacillus Calmette-Gue’rin (BCG) instillation between June 1999 and April 2000. Because of severe voiding pain during the last BCG instillation, he received intravesical epirubicin instillation twice after TURBT for recurrent TCC of the urinary bladder (grade I, TaNOMO). Skin rash, itching, shortness of breath, cold sweating, and hypotension were noted immediately after the 3rd intravesical epirubicin instillation. After adequate treatment, the patient was discharged in a stable conditio