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Primary lymphomas of the breast are rare and predominately of B-cell phenotype. Anaplastic large cell lymphoma is a T-cell lymphoma that accounts for only 3% of all non-Hodgkin lymphomas. We present a rare case of primary anaplastic large cell lymphoma of the breast in association with a silicone breast implant capsule in a healthy 40-year-old woman. The patient had bilateral breast implants placed at 21 years of age and presented with painful bilateral breast contractures and associated breast asymmetry. Histology, immunohistochemistry, and T-cell gene rearrangement studies were supportive of a CD 30-positive ALK-1 negative anaplastic large cell lymphoma. This case represents the 14th reported case of primary breast lymphoma in association with breast prosthesis. Of interest is that 11 of these cases were T-cell lymphomas with 8 specifically of the CD30-positive anaplastic large cell lymphoma type. This rare case highlights the importance of histologic examination of breast capsule specimens.