Squamous cell carcinoma (SCC) is a rare variant of prostate cancer. We report a case of a patient who was diagnosed with metastatic adenocarcinoma of the prostate, treated with leuprorelin and subsequently found to have SCC 18 months later. We have found one case in the literature with a similar scenario of possible transformation of adenocarcinoma to SCC secondary to luteinizing hormone-releasing hormone (LHRH) treatment. We found interesting similarities between the two cases, which raise the possibility of the transformation of tumour type and highlights the importance of the clinical picture in the follow-up, even with low prostate specific antigen (PSA) value.