Indolent B-cell lymphoma associated with breast implants: a case report

Introduction

Since the first silicone-gel breast implant was placed in 1962 by Cronin and Gerow, the prevalence of breast augmentation has increased significantly (1). Approximately 300,000 breast augmentation procedures are performed per year in the United States, making it the second most common cosmetic surgical procedure (1). In addition, alloplastic techniques with either saline or silicone implants remains a popular method of post-mastectomy reconstruction. As such, it is increasingly important to understand the risks of this procedure, both in the perioperative setting and over a patient’s lifespan. Approximately 30 years ago, researchers first characterized the relationship between textured silicone breast implants and the development of breast lymphoma, resulting in the recall of multiple textured implants due to heightened cancer risk (2). The majority of these cases of breast implant-associated lymphoma are of the T-cell type, with anaplastic large cell lymphoma (BIA-ALCL) being the most commonly reported. The prevalence of this varies widely in the literature, with recent meta-analyses estimating a prevalence of 1 in 913 to 1 in 9121 individuals who received textured silicone implants (3-5). To this date, there are only a limited number of cases of B-cell lymphoma associated with breast implants that have been documented in the literature. These cases show significant variation in demographics, surgical history, time from implantation to confirmed lymphoma, and immunohistochemical characteristics (2,6-20). This study presents a case of breast implant-associated indolent follicular B-cell lymphoma in a patient who had textured silicone breast implants placed 37 years prior to diagnosis, which is a longer interval between implantation to diagnosis than other studies in the literature. We also discuss our management approach to this rare diagnosis. We present this article in accordance with the CARE reporting checklist (available at https://aol.amegroups.com/article/view/10.21037/aol-26-0002/rc).

Case presentation

An 86-year-old female presented to the emergency department with a 10-day history of progressive right breast erythema, swelling, and tenderness which she described as a tearing pain. Further history was concerning for four months of slowly progressive constitutional symptoms of night sweats, an unintentional weight loss of seven pounds, and fatigue. The patient initially thought she was ill from a viral infection and anticipated that these symptoms would subside without intervention. She elected to seek care at the emergency department when her symptoms acutely worsened.

Her medical history was notable for invasive ductal carcinoma of the breast diagnosed in 1985, T1b[0.6 cm] N0[0/33]. That same year, she elected for bilateral nipple-sparing mastectomies with implant-based reconstruction utilizing saline implants with a textured silicone shell. At that time, she had a concomitant right latissimus dorsi flap due to a thermal injury of the right chest skin secondary to a heating pack she had used in an area where she developed post-mastectomy paresthesia. It is unknown what adjuvant treatments, if any, the patient received. The patient was free of associated complications from her implant-based reconstruction until the presentation of these current symptoms. The patient had no previous breast surveillance imaging in our system, although she may have had routine breast imaging in another healthcare system.

Given her history of breast implant reconstruction and the current area of concern, plastic surgery was consulted by the emergency department. Initial physical examination demonstrated an edematous right breast approximately 40% larger than the left breast with grade II capsular contracture, ptosis, and no axillary or cervical lymphadenopathy (see Figure 1). Complete blood count (CBC), metabolic panel, and lactate dehydrogenase (LDH) were unremarkable other than mild leukocytosis to 11.2×10⁹/L (see Table S1 for laboratory values on presentation). Breast magnetic resonance imaging (MRI) was ordered which showed evidence of subdermal fluid collection on the right, as well as a 2.6 cm × 2.3 cm × 2.2 cm rounded heterogeneous mass posterior to the breast implant of unknown etiology. The left breast tissue and implant appeared radiographically normal (see Figure 2).

Figure 1 Preoperative anteroposterior, oblique, and lateral views demonstrating the significant enlargement of the right breast compared to the left, as well as grade II capsular contracture and ptosis.

Figure 2 Bilateral breast MRI, axial view, demonstrating a substantial right breast periprosthetic fluid collection with a 2.6 cm × 2.3 cm × 2.2 cm rounded heterogeneous mass posterior to the implant. MRI, magnetic resonance image.

MRI imaging and her clinical presentation were initially concerning for breast implant-associated anaplastic large cell lymphoma (BIA-ALCL), although a late seroma, implant rupture, or infectious process could not be excluded from the differential. A core needle biopsy of the 2.6 cm heterogenous mass was ordered, which showed primarily fibrous tissue with reactive changes and very few lymphoid cells. Fine needle aspiration (FNA) of the periprosthetic fluid was subsequently performed under ultrasound guidance, yielding a 250-cc sample. Flow cytometric analysis identified low levels of an abnormal monotypic (kappa) B-cell population comprising 0.3% of the sample determined to be significant by laboratory (see Figure 3). The abnormal population demonstrated CD19, dim CD20, CD10, CD23, CD43, CD45, and dim surface kappa light chain expression. Immunohistochemical staining of the biopsy revealed a mixed population of CD3 and CD20-expressing lymphocytes with no significant CD30 or ALK positivity.

Figure 3 Flow cytometric analysis of peri-prosthetic fluid.(A) Flow cytometry plot demonstrating CD10 co-expression in CD20 positive regions. (B) Flow cytometry plot demonstrating monotypic kappa expression.

Due to our primary concern for lymphoma, oncology was consulted, and a positron emission tomography (PET)/computed tomography (CT) scan was obtained which did not show increased metabolic activity, although could not exclude a low-grade malignancy. Based on these results as well as the patient’s wishes for explantation without implant replacement, plastic surgery proceeded with bilateral breast implant removal and total capsulectomy with excision of excess skin for a flat, aesthetic closure. Intraoperatively, the right implant was found to be intact surrounded by a fibrous capsule and brown seroma (see Figure 4). On the table, an approximately 1 cm nodular density was palpable on the posterior aspect of the implant/capsule corresponding to the density noted on preoperative MRI imaging, in addition to a large hematoma likely secondary to core needle biopsy of the lesion preoperatively. The left implant sustained an iatrogenic dissection injury but otherwise was intact without periprosthetic fluid and consistent with preoperative imaging. The bilateral capsules were sent to pathology for analysis.

Figure 4 Intraoperative view of removed right breast implant, where the right implant was found to be intact surrounded by a fibrous capsule and brown seromatous fluid. A 2.5-cm hematoma was noted in the region of the 2.6 cm × 2.3 cm × 2.2 cm fibrous mass noted on MRI imaging which was likely secondary to biopsy preoperatively. MRI, magnetic resonance image.

Macroscopic grossing of the right breast capsule tissue identified a 0.8-cm fibrous nodule on the posterior aspect of the capsule. Given that preoperative MRI imaging measured this at approximately 2.5 cm, this likely represents an overestimation on imaging or an underestimation on macroscopic examination given the challenges of identifying the true borders of pathological tissue. Histologic sections of the nodule demonstrated an atypical nodular lymphocytic infiltrate adjacent to a large vessel. The infiltrate included abundant small mature lymphocytes, admixed plasma cells, histiocytes, and scattered eosinophils with rare follicles present. No large, atypical cells concerning for BIA-ALCL were identified. Immunohistochemical staining of the abnormal lymphoid tissue demonstrated a predominant population of CD20-expressing B-cells; areas within the CD20⁺ regions also demonstrated immunopositivity for CD10 and BCL2. Ultimately, after Hematopathology consultation with agreement, the lesion was signed out as an atypical lymphoid infiltrate; the rendered differential diagnosis included in situ follicular B-cell neoplasm vs. classic follicular lymphoma. However, classic follicular lymphoma was favored given there were no definitive germinal centers. The left breast capsule was unremarkable (Figure 5).

Figure 5 Histopathological specimens of right breast capsule. (A) Low power view of right breast capsule demonstrates atypical lymphocytic infiltrate (H&E, 2×). (B) Low-medium power view of right breast capsule which demonstrates foci of lymphoid tissue consisting of small, mature lymphocytes (H&E, 20×). (C) Low power view of right breast capsule using an immunohistochemical stain for BCL-2 showing BCL-2 positivity in the B-cell rich (CD20 positive) area (2×). (D) Low power view of right breast capsule using an immunohistochemical stain for CD20 demonstrating lymphoid aggregate with increased B-cells (2×). H&E, hematoxylin-eosin.

Her B-symptoms that drove her to initial presentation resolved promptly following surgical explantation. She had an uneventful recovery from surgery. Given the likelihood of classic follicular lymphoma, the patient was treated with close observation and has been monitored closely by plastic surgery and oncology. She has not required any additional procedures. The patient has remained clinically symptom-free at her 3-year follow-up and remains pleased with the results of her surgery.

All procedures performed in this study were in accordance with the ethical standards of the institutional review board (Walter Reed National Military Medical Center, Approval number DBS.2024.702, reference 971815. April 10, 2024) and with the Declaration of Helsinki and its subsequent amendments. Written informed consent for publication of this case report and accompanying images was not obtained from the patient or the relatives after all possible attempts were made. The article has been very sufficiently anonymized to cause no harm to the patient or her family.

Discussion

Since the first case report of breast implant-associated lymphoma was published in the late 1990s by Keech and Creech, the relationship between textured silicone implants and capsular lymphoma has been well-established and accepted in the medical community (21). The majority of these cases are anaplastic large cell lymphoma (BIA-ALCL), a type of T-cell lymphoma. To the author’s knowledge, there are only a scarce number of cases in the literature of an implant-associated B-cell lymphoma, only few of which were follicular lymphoma (see Table 1).

As such, there remains a major literature gap regarding the etiology, clinical presentation, and treatment considerations for this exceedingly rare diagnosis, thus highlighting the need for additional characterization. The etiology of implant-associated B-cell lymphoma is still debated. Although a causal relationship has not been clearly established, the literature suggests that Epstein-Barr virus (EBV) may play a role in the pathogenesis of implant-associated B-cell lymphoma, with many of the reported cases showing pleomorphic CD30⁺ EBV-infected B-cells (14). This mechanism of B-cell lymphomagenesis may differ fundamentally from BIA-ALCL which is currently understood to be driven by chronic inflammation, host genetics, and immune stimulation from the implant surface (22). Additional research is needed to delineate this relationship in implant-associated B-cell lymphoma, however this is challenging given its low incidence. We now report a rare case of breast implant-associated follicular lymphoma presenting both the oldest patient in our literature review (86 years) and the longest period between implantation and follicular lymphoma diagnosis (37 years). The number of cases of breast-implant associated lymphoma are growing in the literature, however, there will always be challenges in determining the true incidence of this in the population as there is currently no widely accepted framework for diagnosis of implant associated B-cell lymphomas as there is with BIA-ALCL where there is a worldwide recognized diagnostic and treatment framework recognized by the National Cancer Consortium Network (NCCN) and World Health Organization (WHO). Thus, the true incidence of breast implant associated B-cell lymphoma may be underreported (23).

There are numerous different treatment options for breast implant-associated lymphoma, with treatment considerations based on the patient’s diagnosis, grading, symptoms, clinical history, age, and quality of life. In general, follicular lymphoma is an indolent lymphoma characterized by a slow clinical course that is frequently incurable, although it is heterogeneous in its clinical presentation and outcomes (24). Current treatment recommendations in the literature are to treat lower-grade follicular lymphomas (1–3A) with observation unless the patient is significantly symptomatic, while grade 3B follicular lymphoma is treated in a similar fashion as diffuse large B-cell lymphoma (DLBCL) with R-CHOP chemotherapy (rituximab, cyclophosphamide, hydroxydaunorubicin, vincristine, and prednisone) with some centers also electing for maintenance with rituximab (24). As follicular lymphoma is usually an indolent cancer, observation is a common treatment for patients with low-grade follicular lymphoma given that the treatments have both short- and long-term side effects (24).

Follicular lymphoma grading varies based on the classification system utilized. The International Consensus Classification (ICC) requires the average number of centroblasts per 40× high power field (HPF) from randomly selected follicles (10-20 HPF), while the WHO5 requires absolute numbers of centroblasts/transformed cells in 10 consecutive HPFs (25,26). Both schools of thought differ from the more restrictive WHO4 revised system of counting/estimating the absolute number of centroblasts in 10 neoplastic follicles per 40× HPF with at least 10 HPFs within different follicles (27). In this patient, the atypical lymphoid infiltrate did not have sufficient neoplastic follicles for WHO4 grading, nor did it have enough follicles for ICC grading or involved regions to evaluate via the WHO5. Thus, the histologic grading in this patient was not able to be determined and represents one limitation of this study.

Regardless of the grading or treatment pursued, patients are closely followed over time to monitor for recurrence or progression (28). Additionally, some patients can have a histologic transformation, highlighting the need for close follow-up (29). In the case of this patient, we elected to treat with close observation given the absence of residual disease-causing symptoms following explantation with capsulectomy. Management of cases such as this one requires an interdisciplinary approach to care involving plastic surgery, hematology/oncology, and pathology. We would encourage other institutions to maintain this multidisciplinary approach to care given the high variety of clinical presentations and treatment considerations.

There are limitations to the generalizability of this case report. Given this patient’s original breast cancer was approximately four decades ago, we were unable to verify if the patient underwent any adjuvant radiation, chemotherapy, or hormone therapy. Our records do not indicate such, however the patient was unable to recall the details from her past treatment. Radiation is a known cause of secondary lymphomas of the breast and may have played a role in the pathogenesis of her lymphoma if the patient received adjuvant radiation therapy during her initial breast cancer treatment (30). Additionally, we were unable to verify the brand, make, and model of her textured silicone breast implants which would have been helpful for utilization in future meta-analyses trying to characterize the association between specific implant models and implant-associated B-cell lymphoma. There are a limited number of case reports in the literature characterizing the incidence of breast-implant associated B-cell lymphoma, posing challenges in understanding the true incidence in the population, independent risk factors, and optimal treatment. This emphasizes the need for continued surveillance of this population and collaborative data collections to better define this rare disease process.

Conclusions

This case highlights the exceedingly rare presentation of indolent follicular B-cell lymphoma as a complication of long-term textured silicone implantation, occurring almost four decades after the initial surgery. While BIA-ALCL remains the primary concern in patients with late-onset seromas with constitutional symptoms, clinicians must maintain a degree of suspicion for B-cell variants to ensure appropriate hematopathological workup and clinical decision making. Ultimately, an interdisciplinary approach involving surgical explantation with capsulectomy and oncological surveillance provided an excellent clinical outcome for this patient.

Acknowledgments

None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://aol.amegroups.com/article/view/10.21037/aol-26-0002/rc

Peer Review File: Available at https://aol.amegroups.com/article/view/10.21037/aol-26-0002/prf

Funding: None.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://aol.amegroups.com/article/view/10.21037/aol-26-0002/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional review board (Walter Reed National Military Medical Center, Approval number DBS.2024.702, reference 971815. April 10, 2024) and with the Declaration of Helsinki and its subsequent amendments. Written informed consent for publication of this case report and accompanying images was not obtained from the patient or the relatives after all possible attempts were made. The article has been very sufficiently anonymized to cause no harm to the patient or her family.

Disclaimer: The assertions and opinions contained herein are those of the authors and do not reflect those of the Uniformed Services University or the Department of Defense.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

References

1. Perry D, Frame JD. The history and development of breast implants. Ann R Coll Surg Engl 2020;102:478-82. [Crossref] [PubMed]
2. Chen VW, Hoang D, Clancy S. Breast Implant-Associated Bilateral B-Cell Lymphoma. Aesthet Surg J 2020;40:NP52-8. [Crossref] [PubMed]
3. Nelson JA, Dabic S, Mehrara BJ, et al. Breast Implant-associated Anaplastic Large Cell Lymphoma Incidence: Determining an Accurate Risk. Ann Surg 2020;272:403-9. [Crossref] [PubMed]
4. Santanelli di Pompeo F, Sorotos M, Clemens MW, et al. Breast Implant-Associated Anaplastic Large Cell Lymphoma (BIA-ALCL): Review of Epidemiology and Prevalence Assessment in Europe. Aesthet Surg J 2021;41:1014-25. [Crossref] [PubMed]
5. Santanelli di Pompeo F, Clemens MW, Paolini G, et al. Epidemiology of Breast Implant-Associated Anaplastic Large Cell Lymphoma in the United States: A Systematic Review. Aesthet Surg J 2023;44:NP32-40. Erratum in: Aesthet Surg J 2024 Feb 15;44(3):346. [Crossref] [PubMed]
6. Smith BK, Gray SS. Large B-cell lymphoma occurring in a breast implant capsule. Plast Reconstr Surg 2014;134:670e-1e. [Crossref] [PubMed]
7. Cook PD, Osborne BM, Connor RL, et al. Follicular lymphoma adjacent to foreign body granulomatous inflammation and fibrosis surrounding silicone breast prosthesis. Am J Surg Pathol 1995;19:712-7.
8. Nichter LS, Mueller MA, Burns RG, et al. First report of nodal marginal zone B-cell lymphoma associated with breast implants. Plast Reconstr Surg 2012;129:576e-8e. [Crossref] [PubMed]
9. Evans MG, Miranda RN, Young PA, et al. B-cell lymphomas associated with breast implants: Report of three cases and review of the literature. Ann Diagn Pathol 2020;46:151512. [Crossref] [PubMed]
10. Moling O, Piccin A, Tauber M, et al. Intravascular large B-cell lymphoma associated with silicone breast implant, HLA-DRB1*11:01, and HLA-DQB1*03:01 manifesting as macrophage activation syndrome and with severe neurological symptoms: a case report. J Med Case Rep 2016;10:254. [Crossref] [PubMed]
11. Har-Shai L, Har-Shai Y, Metanes I, et al. Composite breast implant-associated anaplastic large cell lymphoma (BIA-ALCL) and extra-nodal marginal zone lymphoma (MZL) in the capsule of a silicone breast implant. Autoimmun Rev 2019;18:556-7. [Crossref] [PubMed]
12. Geethakumari PR, Markantonis J, Shah JL, et al. Breast Implant-associated Plasmablastic Lymphoma: A Case Report and Discussion of the Literature. Clin Lymphoma Myeloma Leuk 2019;19:e568-72. [Crossref] [PubMed]
13. 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;88:3124-8. [PubMed]
14. Maerten T, Seijnhaeve E, Bernard W, et al. Breast Implant-Associated Epstein-Barr Virus-Positive Diffuse Large B-Cell Lymphoma. Case Rep Hematol 2026;2026:7488322. [Crossref] [PubMed]
15. Huang HH, Ho CL. Bilateral Breast Implant-Associated Epstein-Barr Virus-Positive Diffuse Large B-Cell Lymphoma: A Case Report and Literature Review. Ann Plast Surg 2026;96:S81-6. [Crossref] [PubMed]
16. Patarroyo LL, Vásquez FÁ, Farid Y. Epstein Barr Virus (EBV) positive large B-cell lymphoma associated with breast implants: A case report. JPRAS Open 2025;46:86-92. [Crossref] [PubMed]
17. Novis E, Parikh R, Mechera R, et al. Breast Implant-Related EBV + Diffuse Large B-Cell Lymphoma-Case Report and Review of the Literature. Int J Surg Pathol 2025;33:778-82. [Crossref] [PubMed]
18. Al-Bitar A, Zahreddin A, Shwin M, et al. Primary Breast Lymphoma in a Young Female: Non-Hodgkin's Diffuse Large B-Cell Lymphoma in the Presence of Intact Breast Prostheses. Case Rep Oncol 2025;18:271-7. [Crossref] [PubMed]
19. Brondeel S, Rogge F, De Wolf E, et al. EBV-Positive Diffuse Large B-Cell Lymphoma in Association with Polyurethane Textured Breast Implants: Case Report and Literature Overview. Aesthetic Plast Surg 2023;47:1274-8. [Crossref] [PubMed]
20. Vets J, Marcelis L, Schepers C, et al. Breast implant associated EBV-positive Diffuse Large B-cell lymphoma: an underrecognized entity?. Diagn Pathol 2023;18:52. [Crossref] [PubMed]
21. Miranda RN, Medeiros LJ, Ferrufino-Schmidt MC, et al. Pioneers of Breast Implant-Associated Anaplastic Large Cell Lymphoma: History from Case Report to Global Recognition. Plast Reconstr Surg 2019;143:7S-14S. [Crossref] [PubMed]
22. Lajevardi SS, Rastogi P, Isacson D, et al. What are the likely causes of breast implant associated anaplastic large cell lymphoma (BIA-ALCL)?. JPRAS Open 2022;32:34-42. [Crossref] [PubMed]
23. Santanelli di Pompeo F, Clemens MW, Atlan M, et al. 2022 Practice Recommendation Updates From the World Consensus Conference on BIA-ALCL. Aesthet Surg J 2022;42:1262-78. [Crossref] [PubMed]
24. Gupta G, Garg V, Mallick S, et al. Current trends in diagnosis and management of follicular lymphoma. Am J Blood Res 2022;12:105-24. [PubMed]
25. Arber D, Borowitz M, Cook J, et al. The International Consensus Classification of Myeloid and Lymphoid Neoplasms. 1st Ed. Lippincott Williams & Wilkins; 2025.
26. WHO Classification of Tumours Editorial Board. Haematolymphoid tumours. Vol 11. 5th ed. International Agency for Research on Cancer; 2024.
27. WHO Classification of Tumours Editorial Board. Haematolymphoid tumours. Vol 2. 4th ed. International Agency for Research on Cancer; 2017.
28. Ciobanu A, Stanca O, Triantafyllidis I, et al. Indolent lymphoma: diagnosis and prognosis in medical practice. Maedica (Bucur) 2013;8:338-42. [PubMed]
29. Lossos IS, Gascoyne RD. Transformation of follicular lymphoma. Best Pract Res Clin Haematol 2011;24:147-63. [Crossref] [PubMed]
30. Kim CJ, Freedman DM, Curtis RE, et al. Risk of non-Hodgkin lymphoma after radiotherapy for solid cancers. Leuk Lymphoma 2013;54:1691-7. [Crossref] [PubMed]