Introduction: Primary lymphoma of the breast is very rare, comprising 0.5% breast neoplasms. The majority of breast lymphomas are of B cell type, postulated to originate from intramammary lymph nodes or breast lymphatics. Moreover, a still unresolved entity known as breast implantassociated anaplastic large T cell lymphoma (BIA-ALCL) has emerged over the last 20 years, its incidence believed to be on the rise, as the prevalence of women with breast implants is increasing. Case Report: We report an extremely uncommon presentation of BIA-ALCL in a 55-year-old lady who presented 23 years postbilateral cosmetic breast implants with a locally aggressive mass in the left breast. The mass invaded the chest wall and was associated with left axillary and internal mammary lymph nodes. We review the medical imaging and histopathologic findings of this mass and briefly discuss the different presentations and recommended treatment options. Conclusion: This report reinforces the importance of understanding the possible inherent complications and variable clinical presentations associated with breast implants, in order to assist with early recognition and prompt management of this recently emerging, potentially fatal disease.