A 48-year old male patient from Asia was admitted to the author’s hospital, complaining for more than two months about intermittent gross hematuria accompanied by bulky and dark red clots. The patient also suffered from hemospermia without painful ejaculation during this period and there was no special family or social-related history. A rectal examination suggested a mild enlargement of the prostate, and the central groove was accessible. An irregular and hard mass of about 4 cm in diameter was palpable on the right prostate lobe. Ultrasonographic examination indicated benign prostatic hyperplasia and a prostatic space occupying lesion (). Magnetic resonance imaging (MRI) manifested a prostatic space occupying lesion, presenting mixed signals, with a strong signal around the periphery and cluster-like low signals in the right lobe, at a diameter of about 36 mm (). The total value of prostate-specific antigen (tPSA) was 2.28 ng/mL, the value of free prostate-specific antigen (fPSA) was 0.267 ng/mL, and that of the carcinoembryonic antigen (CEA) reached 4.98 ng/mL. The values of CA-242, CA-50, and CA-199 were slightly higher than normal ones. The patient subsequently underwent a transrectal needle biopsy aimed at the low signal lesion of the prostate. The histopathological examination found no definite malignancy (). Three weeks later, this patient was hospitalized with dysuresia and transurethral plasma kinetic resection of the prostate (TUPKP) was accordingly performed to relieve the symptoms and confirm the diagnosis. It was noteworthy that urethrocystoscopy examined two cord-like neoplasms in the prostatic urethra, extending to the neck of the bladder. Both of them had pedicles that were located at the prostatic apex on the right side of the verumontanum (). The cord-like neoplasm was first removed from the pedicle, and then the right lobe of the prostate was resected. This part of the prostate tissue was surrounded by a multi-chamber cystic mass. There were clear boundaries between the cysts and prostate tissue. In the process of the resection, it was found that the surrounding prostate tissue had a tough texture and no blood supply (). For the purpose of pathological diagnosis, the surgery aimed to remove the whole tumor with clean margins. Surprisingly, postoperative pathology indicated multifocal mucinous adenocarcinoma with a Gleason score (GS) of 4 + 3 = 7 (). Further immunohistochemical staining showed sections were tested positive for PSA and prosaposin (PSAP) (), and negative for caudal type homeobox 2 (CDX-2), cytokeratin-20 (CK20), alpha-methylacyl-CoA racemase (AMACR, P504S), cytokeratin-5/6 (CK5/6), cytokeratin-7 (CK7), high molecular weight cytokeratin 34βE12, and transformation-related protein 63 (P63), and Mucin-2 (MUC2) staining revealed ∼20% positivity (). Radical prostatectomy was performed one month after it was confirmed that the bone scan and colonoscopies demonstrated no abnormality and a follow-up visit was made for the patient for three years to date. The latest examination showed the patient had no biochemical recurrence and all tumor markers remained at normal levels. The MRI indicated the signal of the anastomosis area was normal and no enlarged lymph node was detected in the pelvic cavity.