Retrograde intrarenal surgery (RIRS) in horseshoe kidneys is technically challenging due to altered anatomy and unstable intrarenal pressure (IRP). We report a case of a 45-year-old male with complex renal stones in a horseshoe kidney managed using artificial intelligence (AI)-assisted real-time IRP monitoring during RIRS. The AI system dynamically regulated irrigation and suction to maintain IRP within a safe range (∼30 mmHg), improving intraoperative visibility and procedural control. Stone clearance was achieved without complications. This case highlights the feasibility and potential safety benefits of AI-assisted IRP monitoring in anatomically complex kidneys. © 2026 The Authors