Background: Prostate sarcoma is a malignant tumor from the prostate stroma. However, its pathogenesis is unknown. This is a rare type of prostate tumor.

Case Presentation: A rare case of prostate leiomyosarcoma has been described. A 55-year-old prostate leiomyosarcoma patient who suffered from progressive dysuria and frequent urination for half a year was examined. Pathology, immunohistochemical staining, and laparoscopic radical prostatectomy were performed using three-dimensional (3D) reconstruction to diagnose prostate leiomyosarcoma. The patient did not receive adjuvant therapy after the operation. Final pathology was used to confirm prostate leiomyosarcoma. After one year of follow-up, the patient regularly underwent a digital rectal examination and abdominal MRI. However, no tumor recurrence or metastasis was found.

Discussion: We discussed diagnosis and treatment of prostate leiomyosarcoma and reviewd literatures.

Conclusion: Prostate leiomyosarcoma is a rare special type of prostate tumor, and its diagnosis is mainly based on pathological features. Radical excision is the primary treatment for prostate leiomyosarcoma. The 3D reconstruction is essential in the surgery because it can determine the size of the tumor and its relationship to the surrounding tissue. A postoperative adjuvant therapy can also be performed.