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Giant hydronephrosis is an uncommon clinical entity. Even more uncommon is the association of giant hydronephrosis with a double collecting system and ectopic ureter. Here, we report a case of giant hydronephrosis of the bilateral duplex systems associated with ureteral ectopia. The patient underwent upper pole nephrectomy and upper ureterectomy. To our knowledge, only four similar cases have been reported previously.
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