Objective To investigate the clinical features,therapeutic methods,therapeutic effect and prognosis of a case of bladder urothelial carcinoma patients in combination with trophoblast.Methods The pathological data of a patient with bladder high grade urinary epithelial carcinoma in combination with trophoblast was retrospectively analyzed and the related literatures were reviewed.It was a male patient,69 years old,presenting with total painless gross hematuria.The CT scan showed irregular nodules on the anterior wall of bladder.Results Cystoscopy showed 1.5 cm cauliflower mass on the anterior wall of bladder,and transurethral resection of bladder tumor was performed.The pathology and immunohistochemistry showed urothelial carcinoma.Lung metastases was found due to hemoptysis,and increased serum beta human chorionic gonadotropin (β-HCG) was detected simuhaneously.Pathology was double checked and bladder urothelial carcinoma with trophoblast cell differentiation was considered.Three cycles gemcitabine + cisplatin (GC regimen) and 2 cycles etoposide + methotrexate + vincristine + cyclophosphamide (modified EMA-CO regimen) therapies were given,and the disease was stable at that time.The patient died on June 10,2016,and the total survival period was 9 months.Conclusions The clinical and imaging of urothelial carcinoma with trophoblast differentiation is not special.Diagnosis must depend on the pathological and immunohistochemistry examinations.The efficacy can be evaluated by combining imaging with serumβ-HCG.Treatment should be systemic venous chemotherapy.