In this article, we report a case of a 25-year-old male patient who was under follow-up for nephrolithiasis and repeated urological interventions. His last operation was carried out 9 months ago for insertion of a double-J catheter. Pseudomonas aeruginosa, which is susceptible to only colistin treatment, was detected in the urine culture. Before the removal of the double-J catheter, colistin and ceftazidime antibiotics were started to prevent the risk of bloodstream infection. However, the treatment was stopped urgently due to signs of nephrotoxicity. His treatment was restarted with colistin 300 mg once as the initial loading dose, followed by 150 mg/day. However, this time, colistin neurotoxicity has developed and the treatment was again stopped. Meropenem 6 g/day, gentamicin 2 mg/kg and rifampicin 300 mg were prescribed. Negative urine culture was achieved on the fifth day of treatment.