Breast implant-associated anaplastic large cell lymphoma is a rare tumor, affecting patients in the post-breast implant setting, first reported in 1997. We report an extraordinary case of anaplastic large cell lymphoma presenting not as a breast mass but as a spinal tumor, and thus in distant disease stage if considered breast implant-associated. A 35-year-old female, who had undergone bilateral breast augmentations over a decade ago and recent implant removal, complained of back pain with an anterior thoracic projection bilaterally. Clinical and radiological work-up revealed pleural nodules, T6 vertebral/costal infiltration, enlarged left axillary lymph nodes, and extradural spinal masses bilaterally, from which tumor samples were studied morphologically and immunohistochemically. There was diffuse infiltration by enlarged, neoplastic lymphoid cells with eosinophilic cytoplasm and large, irregular, partially embryoid, or multi-lobulated nuclei with distinctive nucleoli. The findings were interpreted as soft tissue and bone infiltration by an aggressive CD30 + , ALK − anaplastic large cell lymphoma. Since cytological or histological analysis on the initial peri-prosthetic inflammation tissue and effusion was not performed, the diagnosis of breast implant-associated anaplastic large cell lymphoma is critically difficult. If the clinical setting is not regarded coincidental, then it strongly favors the diagnosis of what could be a rare aggressive undetected form of breast implant-associated anaplastic large cell lymphoma. If so, the importance of awareness of this probable clinical entity among female patients and their surgeons as well as of the recognition and cytological study of peri-prosthetic effusions is emphasized, so as to enable early detection of a possible lymphomatous proliferation.