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Myeloid sarcoma (MS) is an extramedullary tumor mass causing proliferation of mature or immature blast cells of one or more myeloid lineages. Involvement of the genitourinary tract is rare. We present a case of MS of the ureteral wall. A 74-year-old man was evaluated for left hydronephrosis and ipsilateral low back pain. A computed tomography scan showed a nodular formation in the pelvic ureter. Urinary cytology revealed cellular atypia, so ureteroscopy was performed showing a distal ureteral mass. The histological examination of the biopsy revealed to be malignant neoplasm. The patient underwent left laparoscopic nephroureterectomy with bladder cuff excision. Microscopic histological examination revealed a tumor compatible with MS. A postoperative positron emission tomography revealed residual hypercaptation of the bladder, pelvic muscle and iliac nodes, so the patient started chemotherapy. A multidisciplinary approach was required, taking into account the patient's age, the already poor renal function and the location of the neoplasm., Competing Interests: None declared., (Published by Oxford University Press and JSCR Publishing Ltd. © The Author(s) 2023.)