Introduction: Dermatofibrosarcoma protuberans is an extremely rare subtype of soft tissue neoplasm.Although DFSP typically manifests in the trunk and extremities, it rarely occurs in the breast, and is often misdiagnosed as benign or malignant breast lesions. Data regarding DFSP in the breast is scarce in existing literature. Only 13 cases in men have been documented. Here, we present a case diagnosed with breast DFSP in a young male patient who underwent surgical excision at our facility. In addition to presenting this unique case, we also conducted a literature review to draw attention to breast DFSP. Currently, there is a lack of reported studies exploring the cause of breast DSFP, the clinical, and radiological features, and the response to surgical treatment.