The aim of our study was to investigate the clinical manifestations, treatment, and prognosis of urethral diverticulum carcinoma (UDC).

We retrospectively reviewed four female patients who were diagnosed with UDC between March 2023 and April 2024.

The patients, aged between 48 and 66 years, had a follow-up duration of 11.1 to 24.7 months. They initially presented with various symptoms, including hematuria, palpable anterior vaginal mass, urinary retention, and weak urine stream. Three patients were diagnosed with adenocarcinoma and one with transitional cell carcinoma based on histopathologic findings. All patients underwent radical cystourethrectomy with pelvic lymph node dissection and ileal conduit formation. Among four patients, one who received neoadjuvant chemotherapy and one who received adjuvant radiotherapy remained disease-free at the last follow-up. The remaining two patients, who underwent surgery alone, developed local recurrence at 2.7 and 2.4 months after surgery, respectively. One was treated with salvage radiotherapy, and the other with concurrent chemoradiotherapy. Both were alive with recurrent cancer at the last follow-up.

UDC is a rare and aggressive malignancy in women with nonspecific lower urinary tract symptoms. Due to its rarity and variable clinical presentation, diagnosis and treatment remain challenging. In this study, radical surgery was the main treatment. Additionally, chemotherapy and radiotherapy may also be considered to manage the disease, and a multidisciplinary approach is needed to optimize treatment.