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Adenoid cystic carcinoma (ACC) of the breast is an infrequent neoplasm, and the occurrence in males is rare. Therefore, diagnostic and therapeutic challenges are inevitable.Herein, we present a case of a 44-year-old man with a tumor on his right breast that he had known about for 6 years.The patient underwent a lumpectomy, and the histological examination confirmed a diagnosis of ACC.Modified radical mastectomy was subsequently conducted in the patient. No positive lymph nodes were observed in the postoperative pathological examination. Following the surgery, the patient received adjuvant chemotherapy.The patient remained recurrence-free at 26 months.Compared to female breast ACC, male breast ACC may behave differently biologically and have a different prognosis. Our case will provide more diagnostic and treatment experience to deal with this disease.