Rare Presentation of Breast Implant Infection and Breast Implant Illness Caused by Penicillium Species

Questions

1. What is breast implant illness?

2. What is the proposed pathophysiology behind breast implant illness?

3. What are the most common etiologies of fungal breast implant infections?

4. What risks should patients undergoing breast augmentation be informed of?

Case Description

A 39-year-old woman with a history of augmentation mammaplasty 20 years prior presented with unilateral breast swelling, tenderness, and erythema in the context of months of fatigue, unusual body odor, brain fog, and inability to lose weight despite exercise and healthy diet. Physical exam revealed Baker-Gordon grade IV capsular contracture in the left breast with tenderness on palpation (Figure 1). The patient underwent implant removal and peri-implant capsulectomy. Upon entering the left breast capsule, 150 mL of purulent drainage was immediately encountered (Figure 2). The implant had a visible white coating on the outside with turbid green fluid with small debris inside (Figure 3) and, surprisingly, showed no evidence of rupture. Both implants were removed for symmetry purposes. Intraoperative fluid and tissue cultures grew Propionibacterium acnes (P acnes) in addition to an unspecified Penicillium species. Following surgery, the patient reported complete resolution of her mammary as well as extramammary symptoms and did not have another breast augmentation (Figure 4). This is the first case of a fungal species associated with breast implant illness and is among the minority of breast implant illness cases observed in a saline implant; therefore, this case may add important context to the growing fund of knowledge about this syndrome.

FIGURE 1.

Preoperative unilateral capsular contracture in left breast.

FIGURE 2.

Purulent fluid encountered upon entering the breast capsule.

FIGURE 3.

White fungal coating with green saline inside the intact infected left breast implant.

FIGURE 4.

Postoperative visit after removal of bilateral breast implants.

Questions

Q1. What is breast implant illness?

Breast implant illness is not a new clinical concept, but its existence remains a contentious debate that has been reignited recently due to patient advocacy and connection via social media. This syndrome includes diverse symptoms such as fatigue, brain fog, muscle fatigue, insomnia, lymphadenopathy, gastrointestinal symptoms, muscle pain, hair loss, and slow healing.¹ Additionally, these patients may exhibit symptoms mimicking autoimmune disorders such as rheumatoid arthritis and Sjogren's syndrome.² Breast implant illness is most commonly seen in patients with silicone implants whose symptoms improve with breast implant removal.¹ This finding may be secondary to the increasing prevalence of silicone implants compared with saline implants. Breast implant illness remains a highly controversial topic in the plastic surgery community but has become a more mainstream concept as patients learn about the disease, as increasing case reports are published, and because of its new recognition by the Food and Drug Administration (FDA).

Q2. What is the proposed pathophysiology behind breast implant illness?

Breast implant illness has been associated with a bacterial biofilm development around implants that can cause a subclinical infection and an inflammatory systemic response.^1,3 This process has been described as “human adjuvant disease,” which describes autoimmune connective tissue disorders arising from injection of paraffin, petroleum, silicone products, and bacterial biofilms that stimulate the immune system.⁴ This syndrome has never been associated with fungal infections. The properties of bacterial biofilms that trigger an immune response and resulting symptoms are unknown, but it is possible that the presence of fungal antigens may cause a similar response. Subclinical bacterial biofilms surrounding implants have also been associated with development of capsular contracture both with and without breast implant illness.^3,5,6 Interestingly, P acnes is the most common microbe cultured in patients with symptoms of breast implant illness,¹ which this patient had colonized in addition to the Penicillium species. It is possible that the breast implant illness symptoms were related to the P acnes colonization, although it seems unlikely to be the sole culprit given the simultaneous florid fungal infection. It is possible fungal infections are the culprit of more breast implant infections and breast implant illness symptoms than previously thought, as 25% of breast infections have negative cultures⁷ and fungal cultures are not routinely done.

Q3. What are the most common etiologies of fungal breast implant infections?

Fungal breast implant infections are exceedingly rare, with few case reports in the literature. Most fungal implant infections are Candida or Aspergillus species. Fungal infections are most often seen in saline implants and tissue expanders due to the more favorable environment to fungi of saline compared with silicone, as demonstrated in in vitro studies by Saray, Young, and Horch.^8,10 Etiology of fungal implant infections is often unknown but is commonly attributed to skin flora contamination associated with tissue expansion and augmentation. Contamination at time of augmentation was identified as a major source of infection in several case reports.^8,11 In 1 center, multiple fungal implant infections with Culvaria species were attributed to saline storage under a water-damaged ceiling.¹² Another possible etiology for fungal implant infections was theorized in a case study by Wright et al,¹³ who proposed that implant membranes may be semipermeable to fungal spores, suggesting infections of surrounding tissue or blood can disseminate and involve the implant as opposed to the predominant theory of contamination of the implant itself. In this case, the source of the patient's infection is unclear. The presented case supports Wright's hypothesis given the timeline and as there was clear evidence of significant fungal infection both inside and outside of the implant without any signs of rupture or discontinuity of implant membrane, suggesting permeability to fungal species.

Q4. What risks should be communicated to patients undergoing breast augmentation?

As with any surgery, it is important to discuss both long- and short-term risks and benefits of the procedure with the patient. Hagopian et al¹⁴ demonstrated both the variation and consensus in the field about what information should be conveyed to patients undergoing breast augmentation for both cosmetic and reconstructive purposes to allow for proper informed consent. The FDA released a new boxed warning in October 202015 for saline and silicone breast implants that recommends patients be informed of the following: the increased risk of breast implant-associated anaplastic large cell lymphoma, particularly with the use of textured implants; breast implant illness and the knowledge that breast implants are not considered lifetime devices; that the chance of developing complications increases over time; and that some complications will require more surgery. While preoperative discussions remain individualized between surgeons and their patients, it seems there is a consensus among both physicians and the FDA regarding risks patients must be informed of prior to consenting for breast augmentation surgery, which now includes generalized symptoms of breast implant illness.

Summary

This case reports a 39-year-old woman with capsular contracture in addition to generalized symptoms who was found to have Penicillium fungal species infection of a saline implant 20 years after original augmentation mammaplasty. Penicillium has not been reported in the literature previously as being associated with breast implant infections or breast implant illness. The patient's mammary and extramammary symptoms improved after bilateral breast implant removal.