Prostate stromal tumour of uncertain malignant potential is a term used to describe a specialised proliferation of stromal cells within the prostate, most tend to be benign, but some can present with local invasion or progress to prostatic stromal sarcoma with distant metastasis. Fortunately, they represent less than 1% of prostate cancers and only a few cases have been described in the literature. We report a case of a 39-year-old male patient who was referred to our centre with this recent diagnosis in the context of acute urinary retention. After an interdisciplinary consideration, a radical prostatectomy was decided for treatment.