Prostate cancer, when metastatic, typically involves the axial skeleton. Sphenoidal metastasis is uncommon. We report a rare case of a 75-year-old man who presented with isolated unilateral exophthalmos. Digital rectal examination and serum prostate-specific antigen level were suggestive of metastatic prostate cancer. The prostate biopsy and imaging findings confirmed the source of the exophthalmos as a sphenoidal metastasis of an aggressive prostate adenocarcinoma.
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