Primary classical Hodgkin lymphoma of breast presenting as recurrent breast abscess in a non-lactating woman: a case report and literature review

Abstract

Introduction

Primary breast lymphoma (PBL) is an exceptionally rare entity, accounting for only 0.3–0.5% of all breast lymphomas. The majority of reported cases are diffuse large B- cell lymphoma. Clinical and imaging findings often mimic breast carcinoma, and diagnosis is usually confirmed incidentally by biopsy. Recurrent breast abscesses in non-lactating women or in men should raise suspicion of underlying malignancy.

Case presentation

We report the case of a 55-year-old non-lactating woman who presented to the surgical clinic with a non-healing wound following the incision and drainage of a recurrent breast abscess. A second excisional biopsy revealed primary classical Hodgkin lymphoma of the right breast, mixed cellularity subtype. The patient was treated with the BV-AD regimen, resulting in an excellent clinical response and complete wound healing.

Conclusion

This case highlights the importance of considering malignant etiologies, including rare lymphomas, in patients with recurrent breast abscesses. It underscores the diagnostic challenges posed by overlapping features with breast carcinoma and contributes to the limited literature on primary classical Hodgkin lymphoma of the breast.

Introduction

Breast lymphoma (BL) is a rare clinical entity that can be classified as either primary or secondary. It may present as a primary tumor originating in the breast or as extra- nodal involvement secondary to systemic disease. Most cases are primary non-Hodgkin lymphomas (NHL), whereas Hodgkin lymphoma (HL) more commonly represents secondary involvement rather than a primary breast manifestation (1, 2).

Primary breast lymphoma (PBL) represents 0.04% to 0.5% of all breast malignancies, 0.38% to 0.7% of all lymphomas, and 1.7% to 2.2% of all extranodal lymphomas. The majority of patients are female, aged between 31 and 81 years. PBL usually presents with unilateral involvement, predominantly in older women. In contrast, bilateral involvement is seen more commonly in younger women and is often associated with pregnancy (3).

The clinical features of breast lymphoma are nonspecific, difficult to differentiate from breast carcinoma, and not helpful in making the diagnosis. The most common presenting symptom is a rapidly growing, painless breast mass, followed by palpable axillary lymph nodes. Nevertheless, pain has been reported to occur in 4–25% of patients. The mass in PBL tends to be larger, and the lymph nodes are softer than in carcinoma. Signs of nipple or skin involvement are uncommon. B symptoms, such as night sweats, weight loss, and fever, are distinctly unusual in BL (2, 4).

In a very few reported cases, patients with breast malignancies present with a breast abscess. Breast abscesses are a common presentation in outpatient surgical clinics and emergency departments. These occur predominantly in lactating women and are typically bacterial in etiology (1, 5). Whenever the abscess or its cavity shows atypical morphology, signs of delayed healing after incision and drainage, or necrosis, it should be referred to secondary care for further assessment and possible biopsy.

Furthermore, a breast abscess in a non-lactating woman should warrant surgical review and biopsy (6).

In this paper, we present an interesting case of PBL with uncommon clinical manifestations in a middle-aged woman who presented with recurrent abscesses associated with delayed wound healing and necrosis. This contrasts with most cases of PBL, which present with a painless mass resembling the presentation of most breast carcinomas.

Case presentation

In March 2023, a 55-year-old married woman, gravida 4 para 4, with a history of prediabetes and prior surgeries (including two cesarean sections and a unilateral salpingectomy for a complicated ectopic pregnancy), presented to the surgical clinic. She reported a one-month history of localized breast pain and swelling in the right breast, associated with a fever >38 °C. On physical examination, there was an erythematous, tender swelling in the upper outer quadrant with prominent local inflammatory signs and a fluctuant area highly suggestive of abscess formation. No nipple or skin changes were noted. Palpable, tender ipsilateral axillary lymphadenopathy was present. The patient had a prior history of a right breast abscess at the 12 o’clock position, approximately 3 cm from the nipple–areolar complex, six months prior (September 2022). This previous abscess had been managed by incision and drainage with secondary healing and yielded a negative biopsy.

An initial wide incision and drainage were performed with secondary intention closure, and a biopsy of the cavity wall was taken, which was negative for malignancy.

However, over the subsequent two months (by May 2023), the wound failed to heal and evolved into a wide oval ulcer measuring 10 × 12 cm, with sloping edges and an infected necrotic floor producing a yellow discharge (Figure 1). Surgical debridement was performed, and a second incisional biopsy was obtained. Histology revealed B lymphoid cells with extensive necrosis, initially raising suspicion for diffuse large B-cell lymphoma, with CD30 positivity. However, due to the extensive necrosis and inconclusive findings, a third biopsy targeting viable tissue at the ulcer margins was obtained three weeks later to ensure diagnostic accuracy. The slides were re-reviewed for confirmation. Final pathology demonstrated atypical lymphoid cells and Reed– Sternberg cells, consistent with classical Hodgkin lymphoma, mixed cellularity subtype. Immunohistochemistry showed positivity for CD30, CD15, weak PAX-5, and variable weak CD20, with negativity for CD3 and CD45.

Figure 1.

Clinical photograph showing the initial presentation of the wide oval ulcer measuring 10 × 12 cm on the right breast, with sloping edges and an infected necrotic floor producing yellow discharge.

The infected ulcer was managed with regular dressings using Flaminal ointment and hydrogen peroxide solution, resulting in modest but slow healing (Figure 2).

Figure 2.

Clinical photograph demonstrating modest but slow healing of the infected ulcer following regular dressings with flaminal ointment and hydrogen peroxide solution.

A PET scan (Figure 3) demonstrated increased FDG uptake (SUVmax 17.4) in the skin ulceration of the upper outer quadrant of the right breast, extending through the breast tail up to the axilla. The ulceration was accompanied by subcutaneous fat stranding and collection-like nodules without a clear separation plane. There was no evidence of pathological lymph node involvement, as the axillary lymph nodes were deemed reactive. Additional findings included an infected sebaceous cyst in the right temporal– parietal scalp region and a left ovarian cyst, both deemed clinically insignificant.

Figure 3.

Axial FDG-PET/CT image showing intense hypermetabolic activity (SUVmax 17.4) in the right breast ulcer and extending through the breast tail up to the axilla, consistent with malignant involvement.

According to the Ann Arbor staging system, this case was classified as stage IVE, reflecting extranodal organ involvement (the breast and overlying skin ulceration) with the presence of B symptoms (unexplained fever >38 °C).

In July 2023, the patient was initiated on the BV-AD regimen (Brentuximab Vedotin, Adriamycin, and Dacarbazine), which includes Brentuximab Vedotin, Adriamycin (doxorubicin), and Dacarbazine. This regimen was chosen over the standard ABVD protocol to avoid bleomycin-induced pulmonary toxicity, particularly given the patient’s active systemic inflammatory state and the extensive necrotic breast wound. Furthermore, recent data support the efficacy of brentuximab vedotin in frontline treatment for CD30-positive Hodgkin lymphoma.

Remarkably, within one week of initiating chemotherapy, significant wound healing was observed, with increased granulation tissue, reduced odor, and decreased secretions (Figure 4).

Figure 4.

Clinical photograph taken one week after initiating the BV-AD chemotherapy regimen, showing significant wound healing with increased granulation tissue, reduced odor, and decreased secretions.

At the two-month follow-up (after two cycles), an interim PET-CT demonstrated incomplete metabolic remission. The patient subsequently completed four additional cycles of BV-AD. A repeat PET-CT in December 2023 showed complete metabolic remission. Clinically, she achieved an excellent response, with complete healing of the previously open breast wound, resolution of oozing, and full resolution of the abscess. B symptoms resolved completely, and the patient reported feeling well. As of her last follow-up in March 2024, she remains in clinical and radiological remission. Table 1 Timeline of clinical events (CARE guideline format).

Table 1.

Timeline of clinical events.

Date	Event
Sep-22	First right breast abscess, incision & drainage, biopsy negative
Mar-23	Recurrent abscess with fever, biopsy negative
May-23	Non-healing ulcer, biopsy inconclusive
Jun-23	Third biopsy confirmed classical Hodgkin lymphoma
Jul-23	BV-AD regimen (Brentuximab Vedotin, Adriamycin, Dacarbazine) started
Dec-23	PET-CT showed complete metabolic remission
Mar-24	Patient remains in clinical and radiological remission

Discussion

Primary classical Hodgkin lymphoma of the breast is an exceptionally rare entity, and its presentation as a recurrent, non-healing breast abscess in a non-lactating woman makes this case particularly unique and clinically significant. The rarity of BL can be explained by the fact that the breast contains very little lymphoid tissue compared to the lung or gut, where primary lymphomas are more common (3, 4).

The diagnostic process in this case was particularly challenging. A recurrent, non- healing abscess in a non-lactating woman warrants a high index of suspicion and necessitates a tissue biopsy. This presentation contrasts sharply with the typical presentation of PBL, which is usually a painless mass. Furthermore, the presence of extensive tissue necrosis posed a significant diagnostic pitfall. Multiple biopsies and repeated pathological evaluations were required to establish a definitive diagnosis. The predominance of necrotic tissue limited the identification of cellular markers through immunohistochemistry, rendering initial samples inconclusive. Therefore, necrotic tissue in biopsy specimens should be regarded as non-diagnostic rather than negative, and repeat sampling targeting viable tissue is strongly recommended when clinical suspicion persists (7).

A summary of reported cases of primary breast lymphoma from the literature is presented in Table 2. A structured literature search was conducted using the PubMed database to identify reported cases of breast lymphoma, employing the keywords “primary breast lymphoma” and “Hodgkin lymphoma of the breast.” The search covered the period from 2000 to 2024. Inclusion criteria were case reports, case series, and retrospective studies describing primary Hodgkin lymphoma involving the breast. Articles not written in English or lacking sufficient clinical details were excluded. A total of 17 relevant studies were identified. Analysis of these cases reveals several important patterns. First, the vast majority of patients were female, with only one reported male case. Second, most patients were over the age of 50, which is a general pattern also seen in breast carcinomas. Third, the most common clinical presentation was a painless breast mass. Fourth, the right breast was more frequently involved.

Table 2.

Summary of reported cases of primary breast lymphoma (2000-2024).

Author/year	Age/sex	Presentation	Side	Histologic subtype	Treatment	Outcome
Stanton et al. (2000)	45/F	Breast Abscess	Right	DLBCL	CHOP	Remission
Karamanak os et al. 2001	38/F	Painless mass	Left	Classical HL	ABVD + RT	Remissio n
Cox et al. 2004	62/F	Painless Mass	Right	DLBCL	R-CHOP	Remission
Hoimes et al. (2009)	29/F	Painless mass	Left	Nodular Sclerosis HL	ABVD	Remissio n
Ventham et al. (2010)	71/M	Breast abscess	Right	DLBCL	R-CHOP	Remission
Sun et al. 2011	54/F	Acute mastitis	Left	DLBCL	R-CHOP	Remission
Pandure et al. (2013) (4)	60/F	Painless mass	Right	DLBCL	CHOP	Remission
Anele et al. 2015	48/F	Axillary abscess	Right	DLBCL	R-CHOP	Remission
Zarnescu et al. (2015)	35/F	Painless mass	Left	Classical HL	ABVD	Remission
Lee et al. 2015	58/F	Painless mass	Right	DLBCL	R-CHOP + RT	Remission
Faenza et al. (2017) (6)	42/F	Painless mass	Right	Classical HL	ABVD	Remission
Nisar et al. 2017 (3)	50/F	Painless mass	Left	DLBCL	R-CHOP	Remission
Villalón- López et al. 2017	65/F	Painless mass	Right	DLBCL	R-CHOP	Remission
Orlandi et al. (2018)	55/F	Painless mass	Right	DLBCL	R-CHOP	Remission
Manimala et al. (2020)	47/F	Painless mass	Left	DLBCL	R-CHOP	Remission
Parashar et al. (2020)	68/F	Painless mass	Right	DLBCL	R-CHOP	Remission
Kazi et al. 2024 (5)	52/M	Breast abscess	Left	DLBCL	R-CHOP	Remission

DLBCL, Diffuse Large B-Cell Lymphoma; HL, Hodgkin Lymphoma; RT, Radiotherapy.

Finally, diffuse large B-cell lymphoma was the most commonly reported histological type. Our case fits the demographic patterns (female, age >50, right breast involvement) but is highly unique due to its presentation as a recurrent abscess and its histological subtype (classical Hodgkin lymphoma).

Regarding treatment, the patient was managed with the BV-AD regimen (Brentuximab Vedotin, Adriamycin, and Dacarbazine). This represents a significant modification of the standard ABVD protocol, replacing bleomycin and vinblastine with brentuximab vedotin. The rationale for this choice was multifaceted. Given the presence of an active, extensive, and infected necrotic breast wound, the use of a bleomycin- containing regimen was considered suboptimal due to the increased risk of pulmonary toxicity and treatment-related complications in the setting of active infection.

Additionally, recent clinical data have demonstrated the robust efficacy of brentuximab vedotin in the frontline treatment of CD30-positive classical Hodgkin lymphoma, providing a strong therapeutic justification for this targeted approach.

Staging of PBL is performed according to the Ann Arbor Staging System. Our case was classified as stage IVE due to the extensive extranodal involvement of the breast tissue and overlying skin ulceration, combined with the presence of B symptoms (fever). While localized stage I disease generally carries a favorable prognosis, extranodal extension and B symptoms are associated with a more aggressive clinical course. Nevertheless, outcomes remain highly dependent on treatment response, and as demonstrated in our case, targeted therapy can achieve excellent remission rates.

This report has several strengths, including the extreme rarity of the condition and the unusual clinical presentation, which provides an important clinical pearl for practicing surgeons and oncologists. However, limitations include the inherent nature of a single case report and the relatively short duration of long-term follow-up. Further studies are needed to better understand the optimal diagnostic and therapeutic approaches for this rare entity.

Conclusion

This case highlights a rare presentation of primary breast lymphoma and underscores the diagnostic challenges associated with recurrent breast abscesses. Clinicians should consider underlying breast malignancy, including lymphoma, in non-lactating patients presenting with recurrent or non-healing abscesses, particularly when clinical progression is atypical.

Funding Statement

The author(s) declared that financial support was not received for this work and/or its publication.

Data availability statement

The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.

Ethics statement

Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article. Written informed consent was obtained from the participant/patient(s) for the publication of this case report.

Author contributions

AK: Writing – original draft, Writing – review & editing. BA: Writing – original draft. IS: Data curation, Methodology, Writing – original draft. HA: Supervision, Visualization, Writing – review & editing. AA: Formal analysis, Investigation, Resources, Writing – review & editing. MI: Conceptualization, Investigation, Software, Writing – review & editing, Writing – original draft.

Conflict of interest

The author(s) declared that this work was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

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The author(s) declared that generative AI was not used in the creation of this manuscript.

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