A survival case of breast necrotizing fasciitis treated with mastectomy: a case report

Introduction

Backgrounds

Necrotizing fasciitis is a rare fatal inflammatory disease caused by bacteria that invades the skin and local subcutaneous tissues, extending to the facia and causing necrosis to these areas with systemic implications leading to loss of limbs and possibly death due to septic shock; it is rare however, to see necrotizing fasciitis reach the muscle unless the facia is breached the infection then could lead to myositis (1,2). Necrotizing fasciitis is divided into polymicrobial (Type 1), monomicrobial (Type 2), marine Vibrio related (Type 3), and a very rare fungal related type that is discussed in case reports and have yet to be widely established as a fourth type (3,4). Polymicrobial type is seen more frequently and is associated with comorbidities such as diabetes, obesity, peripheral vascular diseases, and being immunocompromised. Microbes associated with Type 1 are nongroup A streptococci and Enterobacteriaceae. Type 1 is mainly seen in the abdomen and perineum. Type 2 is less common, unassociated with comorbidities, involves bacteria such as streptococci and staphylococci, and is mostly seen within extremities (3).

Common symptoms of necrotizing fasciitis are fever, tachycardia, pain out of proportion, erythema in skin, edema with grayish discharge and bullae. Necrotizing fasciitis is clinically diagnosed through clinical findings quantified in the Laboratory Risk Indicator for Necrotizing Fasciitis (LRINEC) score and imaging findings through ultrasound and computed tomography (CT) scans. Conditions with a high suspicion of necrotizing fasciitis carry a LRINEC score of 8 and above. Imaging findings characteristic of necrotizing fasciitis would be the presence of air foci in tissues (5). The regions most affected by necrotizing fasciitis are the abdomen, perineum, and extremities (6). It is therefore scarce to see necrotizing fasciitis in the breast, with a handful of case reports published worldwide (5). Initial presentation of this condition in the breast can show up as breast swelling and erythema, masking the diagnosis and causing possible misdiagnosis with other more common inflammatory breast conditions such as breast abscess, breast cancer, or even mastitis (7). The mainstay treatment for necrotizing fasciitis is the usage of antibiotics and tissue debridement, with tissue reconstruction being done after the initial damage has been irradicated (1).

Rationale and knowledge gap

Due to the underreporting of this condition and associated misdiagnosis seen in the literature we present our case report which is of a 54-year-old diabetic female from Kuwait who presented with a breast swelling initially to the emergency department and was misdiagnosed as mastitis, the patient was sent home with antibiotics and later worsened in which she was later diagnosed with breast necrotizing fasciitis and treated resulting in full recovery.

Objective

Our aim is to shed light on this rare disease, to add information to the current literature aiding further healthcare personnel in providing more complete management for conditions with breast swelling in which misdiagnoses may hold dangerous implication on patient’s health. We also aim to show through our management of the condition in utilizing Versajet hydro surgical system, a different and nuanced ways in performing debridement, that might aid patient treatment and recovery. We present this case in accordance with the CARE reporting checklist (available at https://asj.amegroups.com/article/view/10.21037/asj-24-35/rc).

Case presentation

A 54-year-old lady, known case of uncontrolled type 2 diabetes mellitus (T2DM) on insulin and obesity with unremarkable family history. She presented with a painful breast lump of one day duration.

Examination shows that the lump is in the lower medial quadrant of her left breast, measuring 5 mm was red with a black spot at its center and no discharge. There was no history of insect bites. Labs in emergency has white blood cells (WBC) 8×10⁹/L, hemoglobin (Hb) 11.8 g/d, and serum creatinine of 89 µmol/L. The patient was initially treated with amoxicillin/clavulanic acid 1 g twice daily but with no response. The lump increased to 2 cm in size 2 days later and her condition deteriorated with increasing size of lump and increasing severity of pain with no relief, bringing the patient again to the emergency department. The patient was admitted to general surgery with a diagnosis of non-lactational mastitis based on the symptoms and the findings of breast ultrasound, which showed inflammation with diffuse skin thickening, subcutaneous interstitial edema, and underlying edematous echogenic fat lobules, along with streaks of free fluid. No walled or localized collections were observed. No air foci visualized on ultrasound. Patient was started on clindamycin 600 mg three times daily during her first two days of hospitalization. Afterwards she was switched to piperacillin-tazobactam 4.5 g three times daily for 7 days, based on the antibiotics protocol designed for the hospital with the infectious diseases department, there were however no signs of improvement. Furthermore, on the third day of admission her left breast became tender, ridged, and swollen, with the formation of bullae around the nipple (Figure 1A,1B). These bullae produced a yellow offensive discharge. Her vitals showed fever with 38.8, with heart rate (HR) slightly increased 101 beats/min, and blood pressure within normal limits of 125/63 mmHg. Although signs remaining within the normal range, she appeared toxic. Laboratory investigations showed a white cell count of 10.7×10⁹/L, a normal Hb level of 10.7 g/d, a serum creatinine level of 120 µmol/L, glucose 24 mmol/L, sodium 144 mmol/L and C-reactive protein (CRP) of 274 mg/L, LRINEC score was 7. The patient underwent CT scanning of her breast which showed superficial facial thickening associated with superficial and intermuscular gas foci centered to the left breast parenchyma extending to the right breast parenchyma, suggestive of necrotizing fasciitis (Figure 1C).

Figure 1 The photos of the case procedure: (A) the patient’s breast with right breast having a normal appearance and left breast showing signs of change in color and slight bullae development; (B) the patient’s left breast showing signs of change in color and slight bullae development; (C) left breast with superficial thickening associated with superficial and inter-muscular gas foci seen in the center of the left breast parenchyma extending into the right breast parenchyma. Associated skin thickening in both breasts more evident in on the left breast. CT shows extensive soft tissue inflammation/infection secondary to gas forming organism, suggestive of necrotizing fasciitis; (D) right breast undergone partial mastectomy and complete mastectomy of the left breast, clear necrotic tissue can be seen; (E) the patient’s breast after a total of three sessions of debridement and usage of NPWT, with viable tissue and development of granulation tissue seen; (F) the patient’s breast after reconstruction using split thickness skin grafting. CT, computed tomography; NPWT, negative pressure wound therapy.

During the third day of admission the patient was then taken for an emergency debridement of the left breast and right breast and necrotic areas with preservation of healthy tissue. During the surgery, the entire left breast tissue was found to be necrotic, displaying dusky-grey subcutaneous fat and containing pus with air pockets. Consequently, a mastectomy was performed on the left breast and half of the right breast, with the nipple on the right side being spared. Underlying muscles showed a healthy appearance and hence were left untouched during debridement (Figure 1D). Intraoperative cleaning was carried out using hydrogen peroxide, betadine, and 0.9% sodium chloride, followed by packing with betadine-soaked gauze and application of a pressure dressing to be changed the next day in theater. Subsequently, she was admitted to the intensive care unit (ICU) for observation and for a second look procedure the next day, aimed at assessing the extent of the necrosis. In the second surgery, conducted 24 hours later, debridement of the necrotic tissue was performed, extending from the sternum’s top to the upper left chest, dressing was changed on daily basis every morning during rounds. Samples of the tissue were sent for microbiological analysis, which revealed the isolation of Streptococcus mitis/oralis, Streptococcus salivarius, Streptococcus constellatus, Finegoldia magna and the histology confirmed the absence of malignancy.

Her antibiotics coverage was changed based on sensitivity of operative samples after consultation with the infectious diseases department, in which a 10-day intravenous course of ceftriaxone 2 g given once daily was initiated. The wound was evaluated daily in collaboration with the wound care team and displayed a gradual healing process with the development of healthy granulated tissue. As the subsequent swab culture showed no growth for microorganisms and her clinical and physical condition continued to improve, the plastic surgery team was involved in her case. On the 17^th day of her hospitalization, she underwent a third debridement (14 days after second debridement), performed using the Versajet hydro-surgery system, which removed approximately 10% of the necrotic tissue (as seen in Figure 1E). Subsequently, negative pressure wound therapy (NPWT) was applied after the third debridement and changed every 5 days. This led to the consideration of moving forward with a split-thickness skin graft (STSG) for the exposed area, which took place day 27 post-op (10 days after the third debridement). Harvesting of the skin graft was taken from the left thigh alone (Figure 1F). NPWT was placed over the STSG (protected by Vaseline gauze) for another 5 days where graft take was assessed and showed good graft take. The entire post-operative course of the patient was uneventful and carried no unanticipated events.

Patient clinical condition improved, and she was discharged home by day 40 post-op, with scheduled follow-up appointments at outpatient clinics, during follow-up the patient was offered other breast reconstruction methods, but she refused and was accepting of the current breast appearance. Her progress was satisfactory.

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) 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 and is attached in this manuscript.

Discussion

We presented a case of a 54-year-old female patient who came to the emergency department with a day-old breast swelling, the swelling seemed benign in nature and was targeted with antibiotics at first targeting a preliminary diagnosis of mastitis, which did not improve the patient’s condition leading to her return a day later to the emergency department with worsened symptoms (painful breast, swelling and bullae formation around the nipple) and affected vital signs. To which a suspicion of a more aggressive condition was considered showing a LRINEC score of 7 (intermediate suspicion) and a CT scan showing tissue emphysema diagnostic of necrotizing fasciitis. This led to the patient being treated with serial debridement episodes leading to an eventual recovery.

Strengths found in this case report is the transparent reporting and availability of clear timeline for this case, enabling clear tracking of the patient’s symptoms and management since admission. The glaring weakness on the other hand can be seen in the incomplete management seen at the first emergency department visit of the patient, as an early ultrasound could have been taken that would have guided the diagnosis in a better way, as well as performing the LRINEC (Laboratory Risk Indicator for Necrotizing Fasciitis) score earlier on might have showed the level of suspicion to necrotizing fasciitis. This delayed the management of the case and might have prevented the patient from further deterioration and might have led to less aggressive debridement protecting the right breast from partial mastectomy leading to an overall less duration of stay in the hospital. However, it can be pointed out that exposure to cases like this is extremely rare, this can be strengthened by the low number of published articles regarding this topic, this as a result placed necrotizing fasciitis on the unlikely differential diagnosis list of the examining team specially that this is the first documented case to be published out of Kuwait.

Characteristic symptoms that would hint at breast necrotizing would be breast erythema, swelling, pain, necrosis, bullae/blisters, and crepitus. Bullae formation is a hallmark symptom (8). When compared to our case these symptoms were seen in our patient. Associated risk factors to breast necrotizing fasciitis were obesity, diabetes, peripheral vascular disease, and immunosuppression (9). In our case, the patient was obese and had diabetes. Causes of breast necrotizing fasciitis are idiopathic (6), core needle biopsy, human bites (10), post-breast augmentation (11), trauma/minor scratches (12), and heparin-associated (13). Most of these causes are related to the mechanism by which necrotizing fasciitis occurs, that is, by having a breach in the skin where bacteria would be introduced and cause an inflammatory attack (8). In our case there was no clear etiologic factor, the early diagnosis was mastitis. Mastitis is breast tissue inflammation that sometimes involves infection. Symptoms of mastitis include swelling, pain, erythema, and sometimes nipple discharge. This condition is seen commonly in breastfeeding women; however, it can be also seen in postmenopausal women due to hormonal changes (14). Ultrasound findings of mastitis are hypoechoic areas showing inflamed fat lobules (15). One factor that could be hypothesized as a cause is the possibility of tick bites as the patient has an outdoor garden on her house which could have precipitated a tick bite that created the breach in the skin, this mixed with her risk factors might have been what led to the development of necrotizing fasciitis. Our case was treated with antibiotics and mastectomy with serial breast debridement, which resulted in partial mastectomy of the initially unaffected right breast due to necrosis progression. During some of the debridement episodes, we used Versajet hydro surgery system for debridement. Versajet is a device that employs the Venturi principle. Versajet pumps saline solution through a small tube end creating a suction effect hence performing debridement and suction of tissues carrying a reduced damaging burden on tissues (16). Versajet is typically used in debriding diabetic ulcers and second degree burns and sometimes wounds requiring skin grafts. It is not typically mentioned to be used in necrotizing fasciitis, except for one case reports where both cases had it in their lower limbs (thigh and leg) (17,18). Mastectomy was the most frequently observed treatment of breast necrotizing fasciitis reported (8). The usage of NPWT supplemented further reconstruction of tissue done in our case. Then, tissue closure was done with an STSG over the debrided breast area.

Conclusions

Breast necrotizing fasciitis is a life-threatening rare condition that is difficult to diagnose and can be missed due to its inflammatory nature where other diagnoses may take its place. Knowledge of the hallmark symptoms and a complete timely management algorithm with a staged approach together enable successful patient care and may avoid causing further harm to patients.

Acknowledgments

We would like to acknowledge the Radiology Department in Sheikh Jaber Al-Ahmad Al-Sabah Hospital in Kuwait for their contribution in this case.

Funding: None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://asj.amegroups.com/article/view/10.21037/asj-24-35/rc

Peer Review File: Available at https://asj.amegroups.com/article/view/10.21037/asj-24-35/prf

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://asj.amegroups.com/article/view/10.21037/asj-24-35/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 and/or national research committee(s) 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 and is attached in this manuscript.

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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