Background: Protease inhibitors (PIs) are a well-documented cause of nephrolithiasis. Although medications such as indinavir are known to increase risk of stone formation, the association of newer HIV medications is not as well studied. In this study, we report a case of a patient who developed atazanavir stones. Case Presentation: A 74-year-old man with HIV on antiretroviral therapy-including atazanavir, a PI-presented with right flank pain. He previously had passed two ureteral stones that were not analyzed. A CT scan showed mild right hydronephrosis without evidence of nephrolithiasis or ureteral obstruction. The patient was presumed to have passed a stone and was discharged home. He returned one day later with persistent flank pain and acute kidney injury that did not improve with intravenous fluid hydration. A right ureteral stent was placed that relieved his symptoms. Subsequent ureteroscopy demonstrated bilateral ureteral stones that were basket extracted. Stone composition was 100% atazanavir. Since being switched off of this medication, the patient has not had any further episodes of renal colic and his renal function has improved to below his baseline level on presentation. Conclusion: Patients treated with the PI atazanavir are at risk for developing nephrolithiasis and obstructive uropathy. Because these stones can be radiolucent on CT scan, a high level of suspicion is required to accurately diagnose ureteral obstruction in these patients. Alternative effective HIV treatment regimens can to be utilized when clinically indicated.