A novel surgical approach to treat lymphangioma of the breast and prevent seroma—a case report

Introduction

Lymphangiomas are benign lymphatic tumours which are histologically composed of networks of endothelium-lined spaces (1). They can be classified as simple (capillary) or cavernous lymphangioma (cystic hygroma). On histological assessment, simple lymphangiomas can be distinguished from capillaries only by the absence of blood cells (1). Cavernous lymphangiomas are typically found in the neck and axilla of children (2). These lesions are composed of dilated endothelial lined lymphatic spaces separated by connective tissue stroma with lymphoid aggregates (1).

Lymphangioma of the breast is extremely rare, with few cases described in the literature. Table 1 summarises previously published cases of breast lymphangioma. In previously published case reports, surgical excision has been a successful treatment modality (3-8). There is no evidence of successful management with aspiration or steroid injection. Quilting in breast surgery typically refers to the use of sutures to secure the skin flaps to the underlying pectoralis major muscle. This technique significantly reduced seroma formation in mastectomy patients (9,10). There is a paucity of evidence within the literature describing surgical techniques, in cases of lymphangioma, to reduce seroma formation and achieve acceptable cosmesis. There are no comments on patient satisfaction with cosmesis in published case reports.

This case describes a novel surgical approach to treatment of lymphangioma of the breast, focusing on the application of quilting which reduces the dead space within the cavity after surgical excision to eliminate the risk of seroma formation and maintain acceptable cosmesis. The operative approach has not previously been described. We present this article in accordance with the CARE reporting checklist (available at https://abs.amegroups.com/article/view/10.21037/abs-24-13/rc).

Case presentation

All procedures performed in this study were in accordance with the ethical standards of the institution of NHS Lothian Caldicott Office, as approved by the local Caldicott Guardian (Caldicott Application No. 2315) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

This 44-year-old lady presented with a left sided breast lump in the upper outer quadrant. She described recurrence of the lump following multiple aspirations of fluid over an 18-month period (within a different health board), the last occasion being 4 months previously. She had no significant past medical history. She had no systemic symptoms and no previous history of breast or axillary disease. There was no family history of breast or ovarian cancer.

On clinical examination there was a 10 cm mass lesion in the upper outer quadrant of the left breast. There were multiple palpable lymph nodes in both axillae and a recent history of coronavirus disease 2019 (COVID-19) vaccination.

Magnetic resonance imaging (MRI) scan revealed a 16 cm × 8 cm × 8 cm fluid collection in the left upper outer quadrant extending into the axilla to lie under pectoralis major muscle (Figure 1). Breast ultrasound scan revealed a large cystic lesion measuring 79 mm. Aspiration of 375 mL of straw colour fluid was carried out in the clinic setting using a large bore (14 Gauge) cannula with ultrasound guidance and cytology showed abundant lymphocytes and no atypical cells. Ultrasound scan of the axilla revealed multiple enlarged nodes. On a repeat ultrasound scan of the left axilla, the nodes remained enlarged, and a core biopsy showed features in keeping with a reactive lymph node. This patient initially underwent aspiration every second month, using the previously mentioned technique, with the synthetic glucocorticoid Triamcinolone being injected on two occasions. The patients primary concern was discomfort and a large unsightly mass. Repeated aspirations, with re-accumulation of the fluid, brought temporary symptomatic relief but did not provide long-term resolution.

Figure 1 MRI breasts: 16 cm × 8 cm × 8 cm fluid collection in the left upper outer quadrant extending into the axilla to lie under pectoralis major muscle. MRI, magnetic resonance imaging.

Following 8 aspiration procedures over a period of 14 months, with no decrease in size, a decision was taken to excise the lesion. Following an axillary skin crease incision, the lesion was excised intact by a combination of sharp and blunt dissection, care was required in the upper axilla where the cystic lesion was closely applied to a duplicate axillary vein. Attached to the lesion were several axillary nodes that were pigmented from nearby tattoos. Following complete excision, the large residual cavity was closed in multiple layers using a running suture technique with a barbed 2/0 Stratafix suture (Figure 2). The wound was closed with 3/0 Stratafix and Monocryl.

Figure 2 Surgical approach. (A) Large lymphocele excised intact via axillary skin crease incision. (B) Stratafix sutures placed loosely throughout the cavity in multiple layers. This image is published with the patient’s consent.

The specimen weighed 173 grams and measured 150 mm × 100 mm × 25 mm (Figure 3). On microscopy there were 2 main components. The first was varied size interweaved vascular channels, in some places large and cystically dilated, occasionally showing a papillary/villo-nodular appearance reminiscent of reactive synovium (Figure 3). They were lined by flat endothelium, lacking atypia. The channels were associated with reactive lymphoid tissue, with large germinal centres, some of which contained black pigment (thought to be tattoo pigment) and background fibrosis (Figure 3B). Their reactive nature was well supported by their normal immunohistochemical profile for selected markers. The features were consistent with a large benign lymphangioma with secondary inflammation and reactive nodal change.

Figure 3 Pathology, macroscopy (A,B) and microscopy (C,D). (C,D) The staining method is haematoxylin and eosin with the insert on 3C as stated (D2-40 immunohistochemistry), and the width of pictures C&D is equivalent to 2.2 mm. (A) Cross section of the received specimen, showing fatty and membranous areas, with occasional fleshy nodule (lymph node). These were interspersed with possible open cystic structures (the more membranous areas where you can see through it to mat). There was background fat (the yellow areas). (B) Example of specimen tissue showing cut faces of one non-pigmented (right) and one pigmented (left) node. (C) Microscopy. Low power view of histology, showing villo-nodular appearance. The villi are endothelial lined (D2-40 positive, see insert), and are associated with lymphoid aggregates containing scattered black pigment. (D) Microscopy shows interweaving vascular channels of various sizes illustrating background fibrosis.

On review 4 weeks post-operatively, there was no seroma formation or recurrence of lymphangioma. The patient was pleased with the cosmetic outcome and the discomfort associated with the lesion had disappeared (Figure 4). On review fifteen months following surgery, there has been no seroma formation, cosmesis is excellent and there is no indication for lipofilling.

Figure 4 Pre-operative clinical photographs (top) and 4 weeks post-operative clinical photographs (bottom).

Discussion

Lymphangioma of the breast is a rare entity and surgical excision is the mainstay of treatment. There is no evidence to support a conservative approach in the current literature.

Seroma formation is common following axillary and breast surgery due to the potential space that is left behind which accumulates fluid. Seromas can be complicated by infection, discomfort, can have a negative impact on patients due to multiple visits to hospital and can cause breast distortion as the seroma cavity contracts. Although more commonly described following breast surgery for malignancy, there is a need to effectively prevent seroma formation following excision of large breast lymphangiomas. Different methods described to reduce seroma formation include local steroid injection, fibrin glue, drain insertion and quilting.

Triamcinolone is a synthetic glucocorticoid which has been shown to effectively reduce donor site seroma formation after autologous latissimus dorsi reconstruction (11). In a double-blind randomised controlled trial of 52 patients, intra-cavity triamcinolone injection, following seroma aspiration, reduced the need for further aspiration, total number of aspirations, total volume aspirated and total time to dryness. Local triamcinolone has also been shown to significantly reduce seroma accumulation, and supressed levels of inflammatory markers, in a randomised controlled trial of abdominoplasty patients (12). In a randomised clinical trial of 212 women having mastectomy and sentinel lymph node biopsy, methylprednisolone into the wound cavity on the first day significantly prevented seroma formation (46% in methylprednisolone group, 67% in control group, P<0.001) with no complications (13). These patient cohorts are clearly very different from a patient with lymphangioma, with some patients receiving adjuvant chemotherapy and radiotherapy. The evidence from the patient treated here is that triamcinolone is not effective in the treatment of lymphoceles.

In a meta-analysis of 11 randomised controlled trials including 2,009 mastectomy patients, quilting significantly reduced seroma formation compared with conventional closure (P<0.001, RR: 0.367) (9). These findings were supported when a further meta-analysis of 21 studies including 3,473 patients was published in 2022, showing reduced rates of seroma, total volume of drainage, days to drain removal and length of stay in patients following mastectomy +/− axillary lymph node dissection (10). Compared to fibrin glue or triamcinolone, quilting emerged as the optimal strategy to decrease donor-site seroma following latissimus dorsi flap reconstruction (quilting group 5%, control 34.5%; fibrin, 27.6%; and triamcinolone 37.6%) in a randomised trial of 96 breast cancer patients (14). There was no difference in complications. A recent study shows that rows of Stratafix sutures as used in this patient are the most effective quilting technique (15). This is the first case to describe the use of quilting to prevent seroma formation in a case of large breast lymphocele.

An alternative to surgical excision, presented in the literature in the form of a case report, is the combination of lymphaticovenular anastomosis (LVA), and ethanol sclerotherapy which closes the dead space (16). LVA enables lymphatic fluid to flow directly from lymphatic channels to the vascular system, stopping the flow of lymphatic fluid within the cyst and maintain effective drainage. The authors describe resolution of a large cystic lymphangioma, arising from the axilla to the sub-clavicular area between the pectoral muscles, with no subsequent lymphoedema or recurrence at 18 months (16). Key benefits of this approach are the minimally invasive nature of the surgery. However, the lymphatics must be detectable, and this is not always the case if the lymphocele is large, persistent or infected. LVA is a delicate microsurgical procedure that requires subspecialist training and specialised instruments. Open surgical excision offers definitive treatment for lymphangioma in cases not amenable to LVA, and is feasible within healthcare systems where specialist microsurgery such as LVA is not routinely available.

Aesthetic outcome is a factor that must not be overlooked in surgery for benign and malignant breast disease. In a study of patients who had breast surgery and radiotherapy, poor patient reported cosmetic satisfaction was independently associated with impaired quality of life, body image, and lower emotional and social functioning (17). Seromas can adversely affect cosmesis after breast surgery. The so-called ‘ideal’ female breast is complex and is influenced by culture, race and body type. It is critical to explore each individual patients’ preferences and make shared decisions to arrive at the optimal outcome, in terms of the management of breast disease and achieving an acceptable cosmetic outcome. Reducing seroma formation by quilting in our experience improves cosmetic outcomes.

The surgical approach in this case was successful and strengths included complete excision of the large lesion through a cosmetically acceptable skin incision and the application of the quilting technique to prevent seroma formation within a large potential space. Pre-operative MRI scanning was important to delineate the anatomy of the tumour in relation to major axillary vessels. The cosmetic result was very good from the perspective of both the patient and the surgeon.

The delay between first presentation and surgery was due to a variety of factors. These include initial diagnostic uncertainty, patient factors including re-locating to a new health board, inexperience in treating this rare entity and the timing during the COVID-19 pandemic when non-urgent operations were not taking place locally. Once seen by a senior surgeon the operation was planned and executed in a timely manner with an excellent outcome. A key limitation of this article is that this is a single case report. However, the same approach has been adopted in many different scenarios. This includes removing implants with their capsules and closing the space by quilting and its use in two patients with augmentation with polyacrylamide hydrogel where we drained the gel and excised the pseudocapsule. Thus the technique of quilting as described here can be applied to any situation where, following breast surgery, there are large spaces in the breast that need to be closed. This case report thus adds to the current literature by describing a novel approach in such patients of using quilting with Stratafix to prevent seroma.

Conclusions

In this case report, surgical excision was the definitive treatment of breast lymphangioma. Quilting using a barbed suture to secure the skin flap to the underlying breast tissue or muscle was a successful technique with no seroma formation. The use of Stratafix for quilting in this setting is novel and contributed to the patients rapid recovery.

Acknowledgments

The authors would like to thank Dr Stuart Thomas for providing valuable insights into the pathological reporting of this case.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://abs.amegroups.com/article/view/10.21037/abs-24-13/rc

Peer Review File: Available at https://abs.amegroups.com/article/view/10.21037/abs-24-13/prf

Funding: None.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://abs.amegroups.com/article/view/10.21037/abs-24-13/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 NHS Lothian Caldicott Office, as approved by the local Caldicott Guardian (Caldicott Application No. 2315) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

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/.

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