Primary breast lymphoma represents less than 1% of all primary breast malignancies and most primary breast lymphomas are of B-Cell origin. The association of anaplastic lymphoma kinase (ALK) negative anaplastic large cell lymphoma (ALCL), a very rare form of primary breast lymphoma, with silicone-filled breast implants has been suggested and several case reports supported this proposal, especially in Western countries. Here we describe one of the first cases of primary breast ALK-negative ALCL in association with saline-filled silicone breast implants evaluated in Iran, where the rising number of breast reconstructive and aesthetic surgeries would commit both surgical pathologists and plastic surgeons to be familiar with this entity.