A 39-year-old female patient underwent cosmetic bilateral breast augmentation 10 years ago. Since then, she developed a relapsing periprosthetic effusion which led to multiple implant replacement. Patient was totally asymptomatic and clinical examination was unremarkable. Her medical history was negative for neoplastic diseases. Three years ago, cytological examination of periprosthetic effusion, revealing sheets of CD30+ and CD4+ large-sized atypical cells with multiple mitosis, was consistent with the diagnosis of breast implantassociated anaplastic large cell lymphoma (BI-ALCL). Patient underwent bilateral capsulectomy and prosthetic excision; positron emission tomography (PET) and computed tomography examination excluded the presence of systemic disease dissemination. One year ago, the patient came to the Surgery Unit of our hospital for left axillary lymphadenopathy, confirmed with PET examination which highlighted tracer accumulation (Fig. 1a). Surgical excision of the largest lymph node was performed.