Breast tuberculosis is a rare disease in highly endemic countries, and it is even rarer in Western countries, where only occasionally the local population is affected. The rarity of the disease and particularly the lack of a typical clinical-radiological presentation may cause tuberculosis to be mistaken for breast cancer or a pyogenic abscess. The authors present a case of breast tuberculosis in a 27-year-old nulliparous woman, an Italian citizen of the Caucasian race, who has never resided in a tuberculosis endemic area. She presented with painful retroareolar and para-areolar swelling in the right breast associated with cutaneous hyperemia (without fistulization), resistant to antibiotic therapy. Histopathological examination revealed features of mastitis with epithelioid histiocytes and Langhans giant cells and was characterized by the presence of caseous necrosis which suggested tuberculous inflammation. Ziehl-Neelsen staining showed the presence of acid fast bacilli. In countries where tuberculosis is non-endemic, breast tuberculosis should always be included in the differential diagnosis in cases of inflammatory breast lesions resistant to the usual antibiotic therapies. Early recognition may prevent both clinical progression and surgical excision, as breast tuberculosis usually regresses as a response to appropriate anti-tuberculosis therapy.