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Development of renal tumors is a main complication of acquired cystic kidney disease (ACKD). ACKD associated renal cell carcinoma (RCC) is the most common subtype of RCC occurring in ACKD and is a recently established entity. ACKD associated RCC occurs exclusively in patients with ACKD and hence the name. The tumor is characterized by cells with eosinophilic cytoplasm, cribriform/sieve-like architecture, and extensive intratumoral oxalate crystals. We report, a case of ACKD associated RCC in a 35-year‑old female with end-stage renal disease with ACKD on maintenance hemodialysis, who underwent left nephrectomy for massive hematuria and review the literature.