The thrombophlebitis of the superficial dorsal vein of the penis, called Mondor's penile disease (PMD), is a condition with a low incidence worldwide. In general, it is considered a self-limited disease that usually resolves with conservative management and very rarely requires surgical intervention. We report the case of a 41-year-old patient, who presented PMD which persists after medical treatment with nonsteroidal antiinflammatory drug and low molecular weight heparin. Surgery was decided and thrombectomy plus resection of the superficial penile vein was performed with satisfactory results. A review of the literature is presented, focusing on the limited available evidence of surgical management.