Case Presentation: A 62 year old female with a history of vitiligo, remote bilateral silicone breast implantation, and latent tuberculosis presented with 3 months of B-symptoms, weight loss, and polyarthalgias attributed to a new diagnosis of rheumatoid arthritis. Physical exam was significant for diffuse tender lymphadenopathy most notably at the supraclavicular nodes and a fixed palpable mass over the right upper sternal border of the chest wall. Imaging showed a right chest wall mass measuring 5 cm by 6 cm at the sternocostal joint and a left chest wall mass measuring 2 cm by 3 cm. Diffuse lymphadenopathy was present including bilateral internal mammary, right retropectoral, bilateral supraclavicular, and mediastinal nodes. Intact bilateral breast implants were present with no evidence of intra or extracapsular rupture on MRI. PET scan showed intensely FDG avid involvement of the enlarged lymph nodes, the chest wall mass, and the spleen concerning for malignancy.Fine needle aspiration of the chest wall mass and the supraclavicular lymph node showed clear refractile, non-polarizable foreign body material consistent with silicone particles present intracellularly and extracellularly with an adjacent inflammatory response most suggestive of autoimmune/inflammatory syndrome induced by adjuvants (ASIA) or Shoenfeld’s syndrome. Analysis of the silicone implants after surgical removal showed atypical lymphoid infiltrate of the left implant with histology most suggestive of breast-implant associated anaplastic large cell lymphoma (BI-ALCL). All AFB and fungal cultures were negative. After surgical removal of the breast implants, the patient began therapy with nivolumab and chemotherapy (EPOCH) through a clinical trial.
Discussion: Hospitalists are often involved in the work-up of diffuse adenopathy and should be aware of both of the rare diagnoses seen in this patient. During the last decade silicone breast implants have been recognized as source of silicone adjuvant disease with the potential to rarely cause a chronic inflammatory response. ASIA or Shoenfeld’s syndrome was first proposed in 2011 as a commonality of symptoms induced by various potential adjuvants and has since been associated with autoimmune rheumatic diseases (1). Patients with a history of allergies and autoimmune disease as seen in this patient with vitiligo are predisposed to the development of ASIA syndrome. Other materials used in medical therapies including mesh used for hernia repair, mineral oils, cosmetic fillers, and prosthetic materials used for arthroplasty have been associated with adjuvant effects. Additionally, patients with silicone breast implants have an increased risk to develop lymphomas, particularly anaplastic large T-cell lymphoma (2). ASIA syndrome and lymphoma can present similarly as seen in this patient who required multiple biopsies to eventually confirm both diagnoses. There is little data guiding the treatment of ASIA syndrome but removal of the foreign body results in amelioration of symptoms in most patients (3). As a newly appreciated phenomenon, BI-ALCL has no consensus treatment at this time.
Conclusions: ASIA syndrome should be considered when a patient presents with lymphadenopathy, arthalgias, and B-symptoms with a history of foreign body exposure. New lymphadenopathy in a patient with silicone breast implants should raise concern for both ASIA syndrome and BI-ALCL.