Primary breast lymphoma is a rare disease. We report a patient who developed anaplastic large cell lymphoma in her breast adjacent to a silicone breast implant 14 years after elective breast augmentation. The patient initially presented with a periprosthetic fluid collection and metastatic work up revealed no other focus of disease. She was treated with systemic chemotherapy and then developed a recurrent periprosthetic fluid collection 4 months later prompting breast implant removal and capsulectomy. Pathology revealed recurrent lymphoma and additional chemotherapy was initiated. Review of the literature revealed 5 cases of primary breast lymphoma associated with a breast implant. All 5 patients had anaplastic large cell lymphoma, the same histological subtype as our patient. Including this case, patients presented with either a mass or a periprosthetic fluid collection an average of 8 years after either silicone gel filled or saline filled breast implant placement. Review of 6 total patients with breast implants and anaplastic large cell lymphoma suggests the possibility of an association, however, no conclusions relating silicone implants to lymphoma can be made. Clinicians should include malignancy in the differential diagnosis of periprosthetic fluid collections and periprosthetic masses.