Primary breast lymphomas are rare, comprising 0.5 percent of all breast malignancies. Most (94 percent) are B-cell type.1 In contrast, lymphomas associated with breast implant capsules are mostly T-cell type (92 percent). Of these lymphomas associated with implants, approximately 60 cases of anaplastic large cell (T-cell) lymphoma have been identified, with only four cases of B-cell lymphoma reported.2–5 We report the first large B-cell type lymphoma associated with a breast implant capsule.
An 83-year-old woman was referred with right breast swelling associated with a 44-year-old implant placed following subtotal mastectomy for phyllodes tumor. A mammogram was read as normal. She denied fever, weight loss, or night sweats. On examination, a Baker grade IV capsular contracture of the right breast was associated with swelling and breast asymmetry (Fig. 1). She underwent removal of the intact implant and a complete capsulectomy. Periprosthetic fluid was submitted for cytologic evaluation. Histopathology results revealed large B-cell lymphoma cells within the fluid and inner lining of the capsule that was grossly calcified and layered. The implant appeared to be a smooth Arion balloon-type implant with a long-tail plugged valve (Fig. 2). The lymphoma cells stained strongly positive for CD20 and negative for anaplastic lymphoma kinase-1, CD3, CD30, kappa, lambda, and cytokeratin. These cells were moderately positive for CD45 and negative for Kaposi sarcoma–associated herpes virus. Results of bone marrow biopsy were negative. Positron emission tomography/computed tomographic scans were negative except for slight fluorodeoxyglucose activity in the left breast at the site of a biopsy performed 16 months earlier. Standard chemotherapy and radiation therapy were recommended initially; however, interdisciplinary conference review at M. D. Anderson Hospital recommended careful observation with serial positron emission tomographic/computed tomographic scans. The patient has been symptom- and disease-free for 18 months.
There have been four reported cases of B-cell lymphoma associated with breast implants; however, this case represents the first large B-cell lymphoma. The clinical presentation was very similar to the more common (but still extremely rare) anaplastic large-cell lymphoma: a T-cell lymphoma. The four other B-cell types of lymphomas associated with breast implants reported are (1) nodal marginal zone B-cell lymphoma, (2) extranodal follicular mixed lymphoma, (3) primary effusion lymphoma, and (4) lymphoplasmacytic lymphoma.2–5 These B-cell lymphomas were associated with silicone gel breast implants and, in all cases except for the patient with primary effusion lymphoma, were associated with ruptured implants and palpable lymph nodes. The primary effusion lymphoma case presented with swelling of the affected breast and was found to be positive for Kaposi sarcoma–associated herpes virus. Lymphomatous effusions in body cavities not associated with implants have been Kaposi sarcoma–associated herpes virus–positive.5 Except for the saline implant and Kaposi sarcoma–associated herpes virus negativity, our patient’s presentation was similar to that of the patient with primary effusion lymphoma.
This case renews awareness of the clinical significance of late periprosthetic seromas. As late seromas occur in slightly less than 1 percent6 and breast implant–related lymphomas occur in one in 500,000, there will be many negative aspiration results in following the current recommendations by the U.S. Food and Drug Administration and the algorithm published by Tebbetts7 for management of breast implant seromas. Still, until there is a better diagnostic test, late periprosthetic seromas (>6 months after surgery) without a history of trauma should be collected and submitted for cytology and culture. Ultrasound-guided aspiration or a blunt-tip cannula specifically designed for this purpose (SeromaCath; Greer Medical, Inc., Santa Barbara, Calif.) may be used. Suspicious cytology results warrant complete capsulectomy and implant removal.7 If implant removal without replacement is desired, seroma collection and capsulectomy can be performed at the time of surgery. If implant replacement is desired, intraoperative cytology results are not reliable enough to support immediate implant replacement. Seroma aspiration preoperatively will allow cytology, cluster of differentiation testing, and anaplastic lymphoma kinase testing. Anaplastic large-cell lymphoma stains strongly positive for CD30 and is anaplastic lymphoma kinase–negative. Documented lymphomas should be reported to the U.S. Food and Drug Administration registry (U.S. Food and Drug Administration MedWatch, 1-800-332-1088). Referral to medical oncologists familiar with this rare entity should be obtained to avoid unnecessary chemotherapy and radiation therapy. Although there is no consensus on the medical treatment, the vast majority of breast implant–associated lymphomas (whether B or T cell) are well localized and indolent such that capsulectomy with implant removal alone is satisfactory treatment.8 The cause of breast implant–related lymphoma may be similar to biofilm-protected bacteria, as Helicobacter pylori is related to mucosa-associated lymphoid tissue in the stomach. In both clinical situations, eradication or removal of the bacteria usually eliminates the lymphoproliferative disorder. Implant replacement following lymphoma diagnosis is extremely controversial but may become an option in certain patients after a disease-free interval. Further study is necessary to clarify the cause and treatment guidelines for this difficult and unusual condition.
The authors have no financial interest to declare in relation to the content of this article.
Bruce K. Smith, M.D.
Department of Plastic Surgery
St. Joseph Medical Center, and
The Methodist Hospital Plastic Surgery Residency Program
Sylvia S. Gray, M.D.
Division of Plastic Surgery
University of Texas Medical Branch
1. Surov A, Holzhausen HJ, Wienke A, et al. Primary and secondary breast lymphoma: Prevalence, clinical signs and radiological features. Br J Radiol. 2012;85:e195–e205
2. Nichter LS, Mueller MA, Burns RG, Stallman JM. First report of nodal marginal zone B-cell lymphoma associated with breast implants. Plast Reconstr Surg. 2012;129:576e–578e
3. Cook PD, Osborne BM, Connor RL, Strauss JF. Follicular lymphoma adjacent to foreign body granulomatous inflammation and fibrosis surrounding silicone breast prosthesis. Am J Surg Pathol. 1995;19:712–717
4. Kraemer DM, Tony HP, Gattenlöhner S, Müller JG. Lymphoplasmacytic lymphoma in a patient with leaking silicone implant. Haematologica. 2004;89:ELT01
5. Said JW, Tasaka T, Takeuchi S, et al. Primary effusion lymphoma in women: Report of two cases of Kaposi’s sarcoma herpes virus-associated effusion-based lymphoma in human immunodeficiency virus-negative women. Blood. 1996;88:3124–3128
6. Spear SL, Rottman SJ, Glicksman C, Brown M, Al-Attar A. Late seromas after breast implants: Theory and practice. Plast Reconstr Surg. 2012;130:423–435
7. Tebbetts JB. Diagnosis and management of seroma following breast augmentation: An update. Plast Reconstr Surg. 2011;128:17–25
8. Brody GS. Brief recommendations for dealing with a new case of anaplastic large T-cell lymphoma. Plast Reconstr Surg. 2012;129:871e–872e
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