We are reporting a case of a 54-year-old transgender female with a history of breast augmentation with bilateral silicone implants. Seventeen years later, she presented with an enlarging right breast mass. Pathology confirmed breast implant-associated anaplastic large cell lymphoma (Ann Arbor Stage IIE, TNM Stage III BIA-ALCL). The patient underwent bilateral capsulectomy, sentinel lymph node biopsy with adjuvant CHOP (cyclophosphamide, doxorubicin, vincristine, and prednisone) chemotherapy, and radiotherapy to the right chest, axilla, and supraclavicular lymph nodes. BIA-ALCL is a rare entity, especially in transgender females. We report this case and a review of the literature in this report.