Challenges in management of male breast adenomioepithelioma with malignant behavior: Case report

RATIONALE: Male adenomyoepithelioma of the breast with malignant features is a rare tumor with only one previous case reported in the literature over 25 years ago. PATIENT CONCERNS: We report the case of a 63-year-old man admitted to our Oncology Institute with a painless tumor mass of 6 cm in the left breast with no additional regional lymph nodes. Ultrasound revealed a complex cystic tumor mass of 60 mm in the left breast, with both anechoic (cystic) and echogenic (solid) components, with ill-defined margin. DIAGNOSES: Extemporaneous assessment showed a solid (invasive) papillary intracystic carcinoma. Definitive pathology examination revealed the presence of a breast malignant adenomyoepithelioma. INTERVENTIONS: Based on the extemporaneous assessment, wide tumor excision was performed. The tumor board decided to continue treatment with adjuvant anthracycline-based chemotherapy. OUTCOMES: After 6 years of follow-up, the patient is cancer-free. No chronic side effects were noted. LESSONS: Because this pathology is extremely rare, no guidelines are available for its therapeutic approach. All decisions regarding patient management should be made by a multi-disciplinary team and can only be based on clinical experience and the few cases reported in female patients.