Basal cell carcinoma (BCC) is a rare nonacinar variant of prostatic carcinoma. In spite of prostatic acinar adenocarcinoma being one of the most common carcinomas in prostate, <100 prostatic BCC cases have been reported to date. Adenoid cystic/cribriform histology has been described in varying proportions to occur in prostatic BCC and is reported to be associated with aggressive behavior and high risk of metastasis. Herein, we present a case of prostatic BCC with adenoid cystic morphology, comprehensively describe its immunohistochemical and MYB/MYBL1 gene rearrangement findings, discuss its differential diagnosis, and review the literature of this rare entity.