A 40-year-old male presented to our urology department with hematospermia. He had continuous hematospermia after several sexual intercourses for the last one month. The hematospermia symptom was not accompanied by painful ejaculation, painful discomfort in the genitals, hematuria, scrotal enlargement, bleeding from other parts of the body, skin petechiae, hot flashes or night sweats. He had chronic symptoms of frequent urination and urgent urination and he was diagnosed with benign prostatic hyperplasia 8 years ago and did not take treatment. His vital signs were stable. A slightly enlarged prostate with a shallow central sulcus and a tougher texture were revealed in the rectal examination, no nodules were found, no pressure pain and the rest of the physical examination was unremarkable. He underwent a PSA and MRI examination. The results showed that TPSA was 2.97 ng/ml, FPSA was 0.249 ng/ml, F/T was 0.08. A nodule was present on the left peripheral side of the prostate, with possible bleeding at the margins of the lesion; besides, the prostate was mildly enlarged, volume 50*40*43 mm, and PI-RADS assessment category was 2, meaning the presence of clinically significant prostate cancer is unlikely (Fig. 1A-L). According to the guidelines(6), prostate biopsy is not recommended, but we learned from our previous experience and still recommended him for prostate biopsy. With the patient's consent, he underwent a combined biopsy (Fig. 2). Immunohistochemical results showed CK (-), EMA individual cells (+), CD34 (-), MyoD1 (-), CgA (-), Syn (-), NSE (-), Myogenin (-), desmin (+), SMA (+), Caldesmon (+), Vimentin (+), ki67 60% (+). The pathological results showed that the left lobe biopsy result was malignant, and combined with immunohistochemistry and histomorphology, it was consistent with leiomyosarcoma.
Based on the pathological findings and the MRI results, the diagnosis of prostate leiomyosarcoma could be confirmed.
The patient underwent a further PET/CT examination to clarify the tumor’s metastasis. The PET/CT whole-body imaging results showed a low-density mass in the left outer zone of the prostate, measuring approximately 3.0*2.7 cm, with PET showing a heterogeneous increase in radioactive uptake and a SUVmax value of 6.6, which was consistent with the characteristics of leiomyosarcoma. In addition, the punctate dense shadow was seen in the prostate. The prostate was enlarged with calcification. No abnormalities were detected in the bladder, bilateral seminal vesicles, or rectum, and no distant metastases were detected (Fig. 1M).
He was subsequently treated with robotic-assisted laparoscopic prostatectomy. After surgery, his condition was stable and he had no adverse reactions. After 7 days of observation, he felt well and was discharged from the hospital. Postoperative pathological findings were consistent with biopsy results (Fig. 3). Postoperative pathology showed no tumor involvement was found in the prostate basal and apical margins, which indicated that he might have a better prognosis. Three months after surgery, the patient's follow-up MRI results showed no recurrence or metastasis. His medical timeline is present in Fig. 4A.
From 2012 to the present, we have admitted five patients with prostate sarcoma, three with leiomyosarcoma and two with rhabdomyosarcoma (Table 1). We found that PSA were normal in all five patients. And their age distribution is wide, ranging from 26 to 80, with a median age of 40. Their duration from initial symptoms to diagnosis differ from 19 days to over 1 year. With the exception of the case we reported, other 4 prostate sarcoma patients all died of distant metastases, with overall survival range from 3 months to 7 months. We summarized their treatment process and found that most prostate sarcoma patients were missed at their first visits (Fig. 4).
Table 1
Clinical Characteristics of 5 prostate sarcoma patients at our hospital.
Patient | PT001 | PT002 | PT003 | PT004 | PT005 |
Age | 40 | 50 | 26 | 28 | 80 |
Initial symptoms | hematospermia | hematuria | dysuria and hematospermia | frequent urination, urgent urination, painful urination | hematuria |
PSA (ng/ml) | 2.97 | 2.88 | 1 | 2.31 | 3.10 |
US suggestive of a tumor when diagnosed | (-) | (+) | (+) | (+) | (+) |
MRI suggestive of a tumor when diagnosed | (-) | (+) | (+) | (+) | (+) |
How to get diagnosed | Combined Biopsy | Surgery | Surgery | TRUS | TRUS |
Diagnosis | leiomyosarcoma | leiomyosarcoma | leiomyosarcoma | rhabdomyosarcoma | rhabdomyosarcoma |
Duration from initial symptoms to diagnosis | 19 days | 14 months | 12 months | 10 months | 12 months |
Recurrence and metastasis in three months after surgery | (-) | (+) | (+) | (+) | (+) |
Overall survival | NA | 7 months | 3 months | 4 months | 3 months |
Invasion and metastasis on diagnosis |
Basement membrane | (-) | (+) | (+) | (+) | (+) |
Seminal vesicles | (-) | (-) | (+) | (-) | (-) |
Vas deferens | (-) | (-) | (-) | (-) | (+) |
Lymph Node | (-) | (-) | (+) | (+) | (-) |
Bladder | (-) | (+) | (+) | (-) | (+) |
Rectum | (-) | (-) | (+) | (+) | (-) |
Lung | (-) | (+) | (+) | (+) | (-) |
Liver | (-) | (-) | (+) | (-) | (-) |
Bone | (-) | (-) | (-) | (+) | (+) |
Patient PT001 is the patient described in the text, other 4 are previous patients at our medical center. Abbreviation: PSA, prostate-specific antigen; US, ultrasound; MRI, magnetic resonance imaging; TRUS, transrectal ultrasound-guided prostate biopsy. |