A Case of Necrotizing Fasciitis of the Breast Following Lumpectomy and Oncoplastic Closure
© 2024 HMP Global. All Rights Reserved.
Any views and opinions expressed are those of the author(s) and/or participants and do not necessarily reflect the views, policy, or position of Wounds or HMP Global, their employees, and affiliates.
Abstract
Background. NF is a life-threatening soft tissue infection that most commonly occurs in the lower extremity. While presenting symptoms such as erythema, severe pain, sepsis, and wound crepitation are well documented, diagnosis of NF of the breast often is obscured by a low clinical index of suspicion due to its relative rarity as well as by the breast parenchyma that physically separates the underlying fascia and overlying skin. Several risk factors have previously been identified, such as underlying infection, diabetes, advanced age, and immunosuppression. However, the gross morbidity and high mortality associated with NF warrant continued surveillance of contributing factors across any anatomic location. Fifteen cases in the literature document the development of NF following breast surgery. Case Report. The authors of this case report aim to expand on the current literature through the presentation of a unique case of NF of the breast following right breast lumpectomy and oncoplastic closure with left reduction mammaplasty in an immunocompromised patient found to have concurrent perforated sigmoid diverticulitis. Conclusion. This case exemplifies how frequent postoperative surveillance, a low threshold for intervention, and efficient coordination of care are vital to minimizing the morbidity and mortality risks associated with NF of the breast.
Abbreviations
Abbreviations: BMI, body mass index; CI, confidence interval; ER, estrogen receptor; GAS, group A Streptococcus; HER2, human epidermal growth factor receptor 2; IV, intravenous; NAC, nipple-areolar complex; NF, necrotizing fasciitis; NPWT, negative pressure wound therapy; OD, odds ratio; PDS, polydioxanone; POD, postoperative day; PR, progesterone receptor; SpO2, oxygen saturation as measured by pulse oximetry; TIVA, total IV anesthesia.
Introduction
NF is a rapidly progressive infection of the fascia and subcutaneous tissues that can quickly become life-threatening, with an associated mortality rate of 20%.1 While the condition is rare, occurring in an estimated 0.4 per 100 000 individuals, prompt diagnosis, administration of broad-spectrum antibiotics, and surgical intervention are necessary to avoid the potential for rapid decompensation.2,3 Infections are most often polymicrobial (type I), consisting of both aerobic (eg, Pseudomonas aeruginosa) and anaerobic (eg, GAS, Bacteroides, Clostridium) species. Less frequently, monomicrobial (type II) cases of NF typically involve GAS or other β-hemolytic streptococci. More commonly, polymicrobial NF presents along the lower extremity in comorbid patients with chronic diseases such as diabetes mellitus and immunocompromised states.4,5 One study assessing over 1500 patients with NF reported that approximately 77% of cases occurred in the lower limb, while only 1.9% of cases occurred along the trunk.6
Specifically, NF of the breast is exceedingly rare. A recent systematic review revealed that among 40 total cases of breast NF reported in the literature, just 15 (37.5%) occurred after breast surgery, while the majority of cases (52.5%) were primary without any known inciting factor.7 The rarity of NF of the breast predisposes it to frequent misdiagnosis as cellulitis or abscesses, and to treatment solely with antibiotic therapy without immediate surgical intervention.8 Subsequent delays in appropriate surgical debridement have the potential to foster rapid spread of infection, leading to systemic toxicity, septic shock, and, ultimately, death. In cases of missed diagnosis of NF, total mastectomy followed by delayed skin closure after resolution of the infection often are necessary to achieve favorable outcomes.8,9
Therapeutic surgical decision-making hinges on the ability to diagnose NF efficiently and accurately, which emphasizes the need for continued surveillance of possible contributing factors. The authors of the current case report sought to expand on the literature by presenting a unique case of NF of the breast following right lumpectomy and oncoplastic closure with concurrent left reduction mammaplasty in an immunocompromised patient with perforated diverticulitis of the distal sigmoid. Signed consent was obtained by the patient prior to submission of this case report.
Case Report
A 61-year-old postmenopausal female with stage IA right invasive intraductal breast carcinoma with concomitant ductal carcinoma in situ (T1aN0M0, ER/PR+, HER2-negative) presented to the authors’ institution (MedStar Georgetown University Hospital, Washington, DC) for surgical management and subsequent breast reconstruction. A detailed timeline of the patient’s cancer diagnosis, surgical management, and ensuing postoperative course is outlined in Figure 1.
At the time of consultation for surgery, the patient had a Charlson Comorbidity Index of 5, which signified a substantial comorbidity burden and elevated 10-year mortality risk.10 Her medical history was remarkable for a former 40 pack-year smoking history (quit date: January 2020), BMI of 44.38, hypertension, hepatic steatosis, malignant hyperthermia, and plaque psoriasis requiring chronic immunotherapy with risankizumab, which was discontinued 8 days prior to surgery. The patient also had a complicated surgical history, including a total abdominal hysterectomy complicated by bowel injury, which ultimately necessitated abdominal wall reconstruction with a protracted postoperative course requiring NPWT and healing by secondary intention.
Preoperative planning is depicted in Figure 2A. Immediately prior to surgery, the patient received an IV infusion of clindamycin, 900 mg, in the setting of a documented anaphylactic penicillin allergy. Given her family history of malignant hyperthermia, the patient received general anesthesia in the form of TIVA. Through bilateral Wise pattern incisions, she underwent a right-sided lumpectomy of the 5-mm × 4-mm × 4-mm mass with sentinel lymph node biopsy, removal of 2 lymph nodes, and oncoplastic reconstruction with an inferiorly based 15-cm × 10-cm dermoglandular pedicle flap. Concurrently, a reduction mammaplasty of the left breast totaling 576 g was performed. The surgical team then used the SPY Elite fluorescence imaging system (Stryker) to ensure adequate vascularity. No evidence of skin compromise was found in either breast. Both breasts were then irrigated twice with a triple antibiotic solution (1 g of cefazolin, 50 000 U of bacitracin, 80 mg of gentamicin), and 2 15F Blake drains (Ethicon, Inc; hereafter silicone drains) were placed on each side. Both breasts were closed using a combination of PDS, Monocryl absorbable monofilament (Ethicon, Inc; hereafter poliglecaprone-25), and Stratafix sutures (Ethicon, Inc; hereafter knotless antibacterial, absorbable, barbed sutures) (Figure 2B). Finally, NPWT devices (Prevena Incision Management System; 3M) were placed to overlie the incisions bilaterally. The patient was discharged home on POD 0, as is common practice following oncoplastic and reduction mammoplasty procedures.11,12
When the patient presented to the authors’ clinic on POD 7 for follow-up, drain output was less than 10 mL per day bilaterally, prompting their removal. The bilateral drains and incisional NPWT were removed without difficulty. At that time, the patient reported that her pain was adequately controlled by her postoperative regimen of acetaminophen, ibuprofen, and as-needed oxycodone. The patient recalled that she experienced an episode of fever (103˚F) on POD 6, which resolved without the need for additional antipyretics. Nonetheless, her physical examination was benign, with no evidence of local or systemic infection (ie, afebrile, with no erythema, induration, or excessive purulent drainage). Given these findings, the decision was made to restart risankizumab as previously prescribed.
On POD 9, the patient presented to the emergency department with mild confusion, worsening shortness of breath, increased pain, and copious malodorous brown drainage from the incision site. Upon arrival she was found to be hypotensive (blood pressure of 88/49 mm Hg) and tachypneic (respiratory rate of 28 breaths per minute), with a room air SpO2 of 92%. Her laboratory results were significant for lactic acidosis (3.1 mmol/L), mild leukocytosis (white blood cell count, 12.1/μL), and hyponatremia (sodium level, 122 mmol/L). A computed tomography scan of the chest without contrast revealed more soft tissue gas along the right side of the anterior chest wall than would be expected for postoperative changes, raising the possibility of gas-forming infection as well as moderate free air in the upper abdomen of indeterminate source (Figure 3A). A subsequent computed tomography scan of the abdomen and pelvis revealed significant free air related to perforated diverticulitis at the level of the distal sigmoid colon. Physical examination at that time was remarkable for moderate abdominal tenderness to palpation, erythema along the right breast incision site, 3 areas of necrotic skin measuring approximately 2 cm × 1 cm each, marked tenderness to palpation in the right breast, and grossly malodorous discharge from the incision site (Figure 3B).
Because of concern for NF, the patient was given broad-spectrum parenteral antibiotics (2 g of vancomycin, 1 g of cefepime, 600 mg of clindamycin) and promptly taken to the operating room for surgical debridement of the breast. Intraoperative findings included approximately 500 mL of frank purulence as well as marked fat necrosis in between the inferior and superomedial breast pedicles of the right chest (Figure 4A). The fluid was evacuated, and the necrotic inferior and superomedial pedicles of the breast were debrided until healthy and viable tissue was achieved. All tracks, including the sentinel node biopsy track, track to the latissimus, and track to the superior clavicle were evaluated for the presence of free air. All dark, infectious-appearing areas were excised and irrigated with 6 L of cystoscopy tubing, and the right-sided defect was packed with Dakin-soaked antimicrobial gauze (Kerlix AMD; Cardinal Health) (Figure 4B). No evidence of infection was observed in the left breast. Postoperatively, the patient was intubated, placed on pressors, and upgraded to the surgical intensive care unit.
The patient returned to the operating room 4 additional times (PODs 10, 11, 14, and 19) for debridement of the right chest through the superficial fascia totaling 675 cm2, and of the left chest to the superficial fascia totaling 143 cm2 (Figure 5A). On debridement the right breast superficial pectoralis fascia was found to be grossly necrotic, and it was subsequently removed off the pectoralis muscle. Conversely, the superficial fascia on the left side appeared viable and was left in place. Both breasts were packed with Dakin-soaked antimicrobial gauze between debridements; this gauze was changed daily. Intraoperative cultures on POD 11 were positive for Staphylococcus aureus, P aeruginosa, S agalactiae, S lugdunensis, and Klebsiella pneumoniae. Because the patient remained afebrile and her white blood cell count remained stable between 11 × 109/L and 13 × 109/L, the perforated sigmoid diverticulitis was managed conservatively throughout with serial imaging, continuous monitoring of laboratory values, broad-spectrum antibiotics, and fluid resuscitation as appropriate.
The patient was successfully extubated and pressors were discontinued on POD 13. Delayed closure of the left chest took place on POD 14 with placement of a 15F silicone drain followed by a combination of PDS, poliglecaprone-25 absorbable monofilament, and knotless antibacterial, absorbable, barbed sutures with overlying liquid adhesive on self-adhering latex-free mesh (Dermabond Prineo; Ethicon US, LLC). The right chest was debrided twice more, on PODs 16 and 19. On POD 19, a 15F silicone drain was placed and the chest was closed in delayed fashion using a combination of PDS and fast-absorbing gut sutures with overlying liquid adhesive on self-adhering latex-free mesh. On POD 22, the patient was discharged home on a tailored parenteral antibiotic regimen (fluconazole, 400 mg, once daily; ertapenem, 1 mg, once daily) (Figure 5B). She received a total of 4 weeks of IV antibiotic therapy prior to de-escalation to oral antimicrobials, which were continued for another 2 weeks.
By 1-month follow-up, the right chest wall incision was clean, dry, and intact with no signs of infection or fluid collection (Figure 5C), and the patient’s nipples remained viable bilaterally. Delayed wound healing was noted at the left inframammary fold and inferior NAC; however, both locations were composed of healthy granulation tissue without palpable fluid collections. At the most recent follow-up visit 3 months postoperatively, the patient continued to display no signs of infection, as well as viable nipples bilaterally and steady improvement in the healing of the left inframammary fold and NAC.
Discussion
This report presents a unique case of NF of the breast following lumpectomy and oncoplastic closure. While NF following breast surgery has previously been reported, most instances have occurred after mastectomy. To the authors’ knowledge, this is the first case to develop after lumpectomy with oncoplastic closure. Although this case is rare, it demonstrates that NF of the breast must remain part of the differential diagnosis, even after episodes of minor trauma.
Known risk factors for NF include underlying infection, penetrating injury, trauma, burns, advanced age, peripheral vascular disease, obesity, immunosuppression therapy, and chronic disease (eg, diabetes).7,13,14 The patient in the current case report had multiple risk factors, including advanced age of 61 years, obesity (BMI, 44.38), immunosuppression for plaque psoriasis therapy (risankizumab), iatrogenic tissue damage secondary to breast surgery, and underlying perforated diverticulitis. The patient’s 40 pack-year smoking history likely contributed to worse outcomes, as both active and former smokers experience significantly more postoperative complications following breast reconstruction than never-smokers (OR, 1.91; 95% CI, 1.69-2.17).15
Risankizumab can be associated with serious infections such as bullous cellulitis, sepsis, and neck abscesses.16-18 Serious infections are infrequent, however, occurring in approximately 1.7 per 100 persons per year according to a pooled long-term safety analysis of 1306 patients.16 Nevertheless, it would stand to reason that the immunosuppressed state of the patient in the current case series increased her overall susceptibility to a severe polymicrobial infection.19 Cases of perforated diverticulitis manifesting as NF have been previously reported, most often occurring along the thigh, groin, or scrotum via retroperitoneal spread.20-23 In the patient in the current case report, hematogenous spread of the infection may have facilitated the development of NF in the anatomically disparate location of the breast.
The patient in the current case report received TIVA rather than inhalation-based general anesthesia due to her history of malignant hyperthermia. In a propensity score-matched registry study, compared to TIVA, the use of inhalation-based general anesthesia was associated with significantly lower rates of postoperative surgical complications (OR, 0.76; 95% CI, 0.71-0.83), including abscess and sepsis, following colorectal cancer surgery.24 Although the patient in the current case report underwent a disparate operation secondary to malignancy, the exposure to TIVA may have further magnified her baseline risk for postoperative complications.
Although there is no consensus on the definitive management of NF, a recent review of 18 published cases of NF indicates a general trend toward total mastectomy, which was performed in 9 cases (50%).8 Total mastectomy was avoided in this case; the patient was successfully treated with serial debridements and an extended course of IV antibiotics, which ultimately permitted conservation of the NAC bilaterally. Her relatively positive outcome was achieved through the rapid recognition of NF in addition to fluid collaboration between infectious diseases and plastic surgery colleagues in the orchestration of necessary medical and surgical interventions.
The successful outcome of the patient in the current case report was, in part, facilitated by her classical presentation of confusion, erythema, pain out of proportion to physical examination, and visualization of soft tissue gas on imaging. Additionally, she presented to the emergency department within hours of symptom onset, enabling early intervention and communication with her care team. This may explain the feasibility of the more conservative treatment in this patient in contrast to the cases of NF in the literature in which the ultimate necessity for mastectomy likely was a consequence of patients’ delayed presentation.25-27
To diagnose NF in a timely fashion, clinicians must maintain a high index of suspicion and be wary of the possibility of nonclassical symptomatology. In the breast, this diagnosis may be physically obscured by copious breast parenchyma separating the infected fascia from its overlying skin, especially in cases of primary NF.7 Shah et al28 were the first to describe a 6-step surgical management approach to primary NF of the breast, which included a radial “pseudotumor” excision of the breast of all involved tissues followed by delayed closure once all devitalized tissue had been sufficiently cleared.
While surgical approach to NF of the breast secondary to recent surgery has been less discretely described, the authors’ approach entailed several steps: (1) reopening the prior incision; (2) evacuation of any fluid collections; (3) thorough debridement of all necrotic-appearing tissue until healthy and viable tissue was achieved; (4) exploration of all local tracks entered during the prior surgery for the presence of free air or infected tissue (eg, sentinel node biopsy track); (5) copious irrigation; (6) packing with antimicrobial gauze; (7) repeat debridements until only healthy, viable tissue remains and cultures display no growth; and (8) delayed incisional closure. This approach is guided by intuition and respect for the pathophysiology of NF. Ultimately, the level of resection depends on the extent of disease and expeditious decisions preoperatively.
In the current case, care was optimized through the coordination of a multidisciplinary team across the specialties of infectious diseases, plastic surgery, general surgery, intensive care, and nurse navigation, who assisted in coordination of follow-up care. Such teamwork permits expert involvement and maximizes the duration of appropriate antibiotics, meticulous debridement and excision, and swift escalation to higher-level care. This case demonstrates how interdisciplinary care can facilitate a systematic approach to a potentially devastating disease and support the optimization of outcomes.
Limitations
This case report is limited by its retrospective nature, which relies on the quality of the electronic medical record. Nonetheless, this unique case of NF of the breast following lumpectomy and oncoplastic closure builds on the current literature by emphasizing the role of prompt recognition and multidisciplinary care in optimizing patient outcomes.
Conclusion
This report details the case of a female who developed NF of the breast precipitated by lumpectomy and oncoplastic closure. Because many patients who undergo surgical resection of breast cancer present with risk factors for developing NF, frequent postoperative surveillance, a low threshold for intervention, and efficient coordination of care are vital to minimizing associated morbidity and mortality risks.
Acknowledgments
Authors: Lauren E. Berger, BA1,2; Daisy L. Spoer, MS1,3; Samuel S. Huffman, BS1,3; Eleanor Drew, MD4; Ian T. Greenwalt, MD5; and Kenneth L. Fan, MD1
Affiliations: 1Department of Plastic and Reconstructive Surgery, MedStar Georgetown University Hospital, Washington, DC; 2Rutgers Robert Wood Johnson Medical School, New Brunswick, NJ; 3Georgetown University School of Medicine, Washington, DC; 4Division of Critical Care Surgery, Department of Surgery, MedStar Georgetown University Hospital, Washington, DC; 5Division of Breast Surgery, Department of Surgery, MedStar Georgetown University Hospital, Washington, DC
Author Contributions: L.E.B.: literature review, drafting manuscript, figure creation, manuscript edits; D.L.S.: drafting manuscript, manuscript edits; S.S.H: drafting manuscript, manuscript edits; E.D.: care of patient, case presentation, manuscript edits; I.T.G.: care of patient, case presentation, manuscript edits; K.L.F.: care of patient, case presentation, manuscript edits
ORCID: Berger, 0000-0002-5747-0009
Disclosure: The authors disclose no financial or other conflicts of interest.
Correspondence: Kenneth L. Fan, MD; MedStar Georgetown University Hospital, Department of Plastic and Reconstructive Surgery, 3800 Reservoir Rd NW, Washington, DC 20007; kenneth.l.fan@medstar.net
Manuscript Accepted: November 22, 2023
References
1. Golger A, Ching S, Goldsmith CH, Pennie RA, Bain JR. Mortality in patients with necrotizing fasciitis. Plast Reconstr Surg. 2007;119(6):1803-1807. doi:10.1097/01.prs.0000259040.71478.27
2. Simonsen SE, Van Orman E, Hatch B, et al. Cellulitis incidence in a defined population. Epidemiol Infect. 2006;134(2):293-299. doi:10.1017/S095026880500484X
3. Al-Qurayshi Z, Nichols RL, Killackey MT, Kandil E. Mortality risk in necrotizing fasciitis: national prevalence, trend, and burden. Surg Infect (Larchmt). 2020;21(10):840-852. doi:10.1089/sur.2019.277
4. Seal DV. Necrotizing fasciitis. Curr Opin Infect Dis. 2001;14(2):127-132. doi:10.1097/00001432-200104000-00003
5. Chen RJ, Gillespie C, Jassal K, Lee JC, Read M. Delayed presentation of breast necrotising fasciitis due to COVID-19 anxiety. ANZ J Surg. 2020;90(7-8):1485. doi:10.1111/ans.16102
6. Khamnuan P, Chongruksut W, Jearwattanakanok K, Patumanond J, Yodluangfun S, Tantraworasin A. Necrotizing fasciitis: risk factors of mortality. Risk Manag Healthc Policy. 2015;8:1-7. doi:10.2147/RMHP.S77691
7. Cai Y, Cai Y, Shi W, Feng Q, Zhu L. Necrotizing fasciitis of the breast: a review of the literature. Surg Infect (Larchmt). 2021;22(4):363-373. doi:10.1089/sur.2020.071
8. ALShareef B, ALSaleh N. Necrotizing fasciitis of the breast: case report with literature review. Case Rep Surg. 2018;2018:1370680. doi:10.1155/2018/1370680
9. Sammoni A, Mohamad O, Abdalah A, Alghazali MB, Al-Aissami M. Management of necrotizing fasciitis and the use of sealed irrigation system: a case report. Ann Med Surg (Lond). 2022;75:103432. doi:10.1016/j.amsu.2022.103432
10. Charlson ME, Pompei P, Ales KL, MacKenzie CR. A new method of classifying prognostic comorbidity in longitudinal studies: development and validation. J Chronic Dis. 1987;40(5):373-383. doi:10.1016/0021-9681(87)90171-8
11. Blankensteijn LL, Crystal DT, Egeler SA, et al. The influence of surgical specialty on oncoplastic breast reconstruction. Plast Reconstr Surg Glob Open. 2019;7(5):e2248. doi:10.1097/GOX.0000000000002248
12. Carpelan A, Kauhanen S, Mattila K, Jahkola T, Tukiainen E. Mammaplasty as an outpatient procedure: a retrospective analysis of outcome and success rate. Scand J Surg. 2015;104(2):96-102. doi:10.1177/1457496914526872
13. Puvanendran R, Huey JC, Pasupathy S. Necrotizing fasciitis. Can Fam Physician. 2009;55(10):981-987.
14. Gupta Y, Chhetry M, Pathak KR, et al. Risk factors for necrotizing fasciitis and its outcome at a tertiary care centre. J Ayub Med Coll Abbottabad. 2016;28(4):680-682.
15. Theocharidis V, Katsaros I, Sgouromallis E, et al. Current evidence on the role of smoking in plastic surgery elective procedures: a systematic review and meta-analysis. J Plast Reconstr Aesthet Surg. 2018;71(5):624-636. doi:10.1016/j.bjps.2018.01.011
16. Gordon KB, Lebwohl M, Papp KA, et al. Long-term safety of risankizumab from 17 clinical trials in patients with moderate-to-severe plaque psoriasis. Br J Dermatol. 2022;186(3):466-475. doi:10.1111/bjd.20818
17. Ojha N, Walsh K, Hess MJ. Non-necrotizing bullous cellulitis and bacteremia: a rare presentation of the Shewanella algae infection. Cureus. 2021;13(1):e12776. doi:10.7759/cureus.12776
18. Bernshteyn M, Kumar PA, Joshi S. Shewanella algae–a novel organism causing bacteremia: a rare case and literature review. Cureus. 2020;12(9):e10676. doi:10.7759/cureus.10676
19. Al Shukry S, Ommen J. Necrotizing fasciitis-report of ten cases and review of recent literature. J Med Life. 2013;6(2):189-194.
20. Underwood TJ, Southgate J, Talbot R, Nash GF. Perforated diverticulitis presenting as necrotising fasciitis of the leg. World J Emerg Surg. 2008;3:10. doi:10.1186/1749-7922-3-10
21. Piedra T, Martín-Cuesta L, Arnáiz J, et al. Necrotizing fasciitis secondary to diverticulitis. Emergency Radiology. 2007;13(6):345-348. doi: 10.1007/s10140-006-0566-9
22. Agaba EA, Kandel AR, Agaba PO, Wong LS. Subcutaneous emphysema, muscular necrosis, and necrotizing fasciitis: an unusual presentation of perforated sigmoid diverticulitis. South Med J. 2010;103(4):350-352. doi:10.1097/SMJ.0b013e3181c1a899
23. Klutke CG, Miles BJ, Obeid F. Unusual presentation of sigmoid diverticulitis as an acute scrotum. J Urol. 1988;139(2):380-381. doi:10.1016/s0022-5347(17)42425-6
24. Hasselager RP, Hallas J, Gögenur I. Inhalation anaesthesia compared with total intravenous anaesthesia and postoperative complications in colorectal cancer surgery: an observational registry-based study†. Br J Anaesth. 2022;129(3):416-426. doi:10.1016/j.bja.2022.03.019
25. Fayman K, Wang K, Curran R. A case report of primary necrotising fasciitis of the breast: a rare but deadly entity requiring rapid surgical management. Int J Surg Case Rep. 2017;31:221-224. doi:10.1016/j.ijscr.2017.01.049
26. Ward ND, Harris JW, Sloan DA. Necrotizing fasciitis of the breast requiring emergent radical mastectomy. Breast J. 2017;23(1):95-99. doi:10.1111/tbj.12686
27. Vishwanath G, Basarkod S, Katageri GM, Mirji P, Mallapur AS. Necrotizing fasciitis of the breast with shock and postpartum psychosis. J Clin Diagn Res. 2011;5(5):1117-1119.
28. Shah J, Sharma A, Johri A, Mearns B, O'Donoghue J, Thomas VA. Necrotising fasciitis of the breast. Br J Plast Surg. 2001;54(1):67-68. doi:10.1054/bjps.2000.3461