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Purpose: Retroperitoneal lymph nodes are a recognized site of relapse in patients undergoing nephroureterectomy (NU) for high grade upper tract urothelial carcinoma (UC). Retrospective studies suggest that retroperitoneal lymph node dissection (RPLND) may be curative at the time of NU for high grade upper tract UC. We hypothesized that chemotherapy followed by RPLND may successfully salvage select patients with isolated retroperitoneal relapse of upper tract UC following prior NU., Materials and Methods: We identified four patients with metastatic UC isolated to the subdiaphragmatic retroperitoneal lymph nodes after NU for upper tract UC. These patients had either a stable response or a complete response to chemotherapy and subsequently underwent a complete full bilateral template RPLND. Our primary study endpoints were disease-specific survival and recurrence-free survival., Results: There was no perioperative mortality or long lasting surgery related sequelae in any patient. Two patients had no pathologic evidence of viable cancer at RPLND and are disease-free at 56 and 74 months from surgery. Two patients had evidence of active residual disease and subsequently developed distant disease at 2 months and 32 months after surgery. Both of these patients died of progressive disease at 3 months and 42 months following RPLND. The 5 year DSS and RFS rates were 50% and 50%., Conclusions: Chemotherapy followed by RPLND for isolated retroperitoneal recurrence after NU for upper tract UC urothelial carcinoma is a feasible and safe treatment that may be potentially therapeutic in select patients.