Anaplastic large cell lymphomas (ALCL) are a rare type of primary breast lymphoma. The association between breast implants and ALCL was first described in 1997. Breast implant associated (BIA)-ALCL arises from the inflammatory T cells surrounding the fibrous capsule, and most tumors are . Here we present the case of a 60-year-old woman with ALCL following bilateral silicone breast prosthesis implantation for aesthetic reason. The patient presented at our observation 7 years following the first surgery reporting a sport trauma in the right thoracic region with breast enlargement and tenderness, complaining breast pain at the palpation of the right breast. Imaging study showed a right fluid collection surrounding the affected breast implant. For this reason, the patient underwent bilateral complete capsulectomy (surgical specimen histologically analyzed and resulted negative for ALCL) and implantation of new breast silicone prosthesis. In 10 months, a progressive relapse of the symptoms with a right peri-implant fluid collection restauration was documented and bilateral surgical removal of breast prostheses with right peri-implant capsular biopsy were performed. The histological analysis of the peri-implant capsule laid for ALCL cellules and a new radical surgical excision of bilateral periprothesic capsule was performed followed by adjuvant radiotherapy for a skin relapse of the disease with a subsequent complete remission. No further relapse was observed afterwards. This clinical case raised a series of diagnostic and therapeutic issues highlighting similarities with previous BIA-ALCL reports, but also some peculiarities that deserve to be further investigated and described in order to make an early recognition of the disease for the proper management and treatment of the patient. Due to the rarity of these tumors, rapidly diagnosing a BIA-ALCL case, not neglecting other associated symptoms or signs, is essential to ensure timely and safe treatment.