Summary: The association of anaplastic large cell lymphoma (ALCL) to breast implants (breast implant-associated ALCL) has brought back the discussion on the clinical safety of the use of silicone implants. A 63-year-old woman came to our institution in early 2015, reporting a gluteal augmentation with silicone implants in 2006 and a recent increasing volume and distortion of the left buttock. Radiologic imaging showed a large amount of fluid collection around the implant. The left side implant was removed and the capsule was left intact, presupposing a future reimplantation. The fluid collected was positive for Staphylococcus aureus. Three years later, she presented again with a new seroma on the explanted side and was submitted to total capsulectomy and fluid drainage, and the material was submitted to laboratory examination. Culture results were negative. Pathologic preparation and sections of the capsule and lumps showed large cells characterized by horseshoe-shaped nuclei. Immunohistochemistry was positive for CD30/CD4 and negative for anaplastic lymphoma kinase, confirming the presence of ALCL, then associated with gluteal implant, an event not described in literature. Positron emission tomography/computed tomography and bone marrow biopsy were performed, and neither showed any other sites involved. The same disease in a new location introduces important discussions about the understanding of this abnormality and poses certain risks and safety issues to clinical scenarios to be discussed. Regardless of whether it is a breast implant-associated ALCL or a gluteal implant-associated ALCL, now we are probably facing an implant augmentation–associated disease and a new international alert should be addressed to the scientific community. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, V.