Clostridium sordellii Surgical Site Infection after Breast Mass Excision: Case Report

Abstract

Background:

Clostridium sordellii infection is a rare condition usually associated with obstetric operations. There have been few reports of C. sordelli in association with other invasive procedures. The mortality rate of this infection approaches 70%.

Methods:

Case report and review of the pertinent English-language literature.

Results:

We describe the first known C. sordellii infection after excision of a breast mass in an adult female. This patient had a prolonged hospital course in the surgical intensive care unit and endured multiple surgical debridements.

Conclusion:

Our patient showed many clinical signs that have been described in past cases of C. sordellii infection. Although the patient had substantial acute morbidity secondary to necessary aggressive treatment, she did survive ultimately. Our case serves the purpose of establishing future treatment for C. sordellii breast surgical site infections with the hope that future treatment may be adapted from our experience.

C lostridium sordellii infections are infrequent, lethal conditions that are associated most commonly with obstetric-related operations [1]. Clostridium sordellii is identified rarely as the causative agent in other invasive procedures or infectious processes. In the few cases described in the literature, the mortality rate approached 70% [1]. Most patients found to have C. sordellii infection decompensate rapidly and succumb to overwhelming septic shock over a period of days.

We present the first known C. sordellii infection after breast mass excision in a young adult female. Our patient endured toxic shock complicated by acute kidney injury and abdominal compartment syndrome. Her complicated clinical course exhibited many of the findings described by previous authors [1,2]. This infection has never before been described after breast surgery.

Case Report

A 37-year-old female with a history of diabetes mellitus, obesity, and asthma underwent an uneventful excision of a fibromyxoid sarcoma of the right breast. On the second post-operative day (POD), the patient presented to the surgeon's office with localized swelling, and she reported subjective fever. Her vital signs were normal (without hypotension or tachycardia), and she was afebrile. Because of the increased edema and the surgeon's concern about an infected hematoma, the patient underwent incision and drainage on POD 2 but no cultures were obtained. Pre-operatively, the patient was started on empiric ertapenem and carbenicillin. Clotted hematoma with purulent material was identified, but there was no evidence of tissue necrosis. The wound was packed with povidone-iodine-soaked gauze dressings, and the wound was managed subsequently with daily dressing changes.

By POD 6, there were worsening local and systemic signs of infection, with increased edema, erythema, and leukocytosis. The patient was taken to the operating room for debridement, and wound cultures were obtained. A deep incisional surgical site infection was evident with extension through the fascial layer of the pectoralis muscle and associated myonecrosis. Cultures grew gram-positive bacilli, which were identified as C. sordellii by the semi-automated Vitek^® 2 (bioMérieux, Inc. Durham, NC) system. The presence of this organism was confirmed with additional biochemical testing. Decisions regarding antibiotic management were made by a general surgeon and internist. On POD 8 after the original surgery, the patient deteriorated rapidly, requiring intubation, and was transferred to our tertiary-care facility.

On arrival, the patient was hypotensive and anemic; a red blood cell transfusion had been started at the outside hospital. The patient's white blood cell (WBC) count was 37,000/mm³ with 13% bands. Cultures were obtained from blood, urine, and via bronchoalveolar lavage on arrival at our institution and on multiple subsequent occasions when the patient developed acute systemic inflammatory responses. All cultures at our facility were negative. She received 14 million units of penicillin G administered on arrival at the surgical intensive care unit and was started on doripenem and linezolid. All antibiotic decisions were made in collaboration with an infectious disease specialist and surgical critical care physician. Outside hospital susceptibility testing demonstrated that the C. sordellii strain was sensitive to beta-lactams, aminoglycosides, macrolides, and fluoroquinolones.

On PODs 10–13, wound care included hyperbaric oxygen therapy and multiple surgical debridements on consecutive days. With debridements, the wound measured 33×20 cm. There was myonecrosis of the pectoralis major muscle identified during the series of five debridements. After debridement, there was no further myonecrosis identifiable. A vacuum dressing was added to the wound on POD 13.

The patient developed septic shock 24 h after the vacuum dressing placement. Fluid resuscitation was initiated in addition to norepinephrine and vasopressin infusions. The vacuum dressing was removed 72 h after its application (POD 16), and the deep medial portion of the wound was noted to have signs of necrosis, but no purulence. Wound management was changed to daily gauze dressing changes. The day the vacuum dressing was removed, the patient developed abdominal compartment syndrome. Urine output decreased acutely, and bladder pressures were elevated to > 30 mm Hg (normal < 20 mm Hg). A decompressive laparotomy was performed on POD 17, and she was managed subsequently with an abdominal vacuum-assisted dressing. The patient developed acute kidney injury and was placed on continuous veno-venous hemodialysis for the following three days. The open abdomen was closed on POD 21, four days after decompression.

The patient recovered slowly over a period of weeks. Linezolid was continued through POD 24. Doripenem was discontinued on POD 26. Vancomycin was started on POD 27 to treat Clostridium difficile colitis empirically after an episode of diarrhea associated with fever and tachycardia. Vancomycin was discontinued on POD 29 after C. difficile testing was negative. She was eventually discharged home approximately 45 days after her original operation.

Discussion

Clostridium sordellii is a gram-positive, anaerobic, spore-forming bacterium first identified in 1922 [3]. The organism can be found naturally in soil and colonizes the vagina and intestines in humans transiently on rare occasions [1,4]. A recent study found C. sordellii naturally colonizing the rectum in 2.8% of sampled women (n=500), whereas positive screenings for vaginal C. sordellii were not identified, supporting the rarity of colonization [5]. The highest incidence of C. sordellii infection occurs after obstetric procedures, although injection drug users are a close second [1]. Rare infections have also been reported involving the upper and lower extremities, eyes, ears, heart, prostate, musculoskeletal tissue, and liver [1,6]. This is the first report describing C. sordellii in breast tissue.

This patient presented with signs and symptoms of a surgical site infection. She was not immunocompromised but did have substantial chronic co-morbid disease. The presence of diabetes mellitus and obesity increased the risk of our patient contracting an incisional infection [7]. Although the treating clinician recognized the surgical site infection, the patient was managed inappropriately. Patients who undergo incision and drainage for deep incisional or organ-space surgical site infections must have wound cultures and consideration of appropriate empiric antibiotics. The Surviving Sepsis guidelines recommend starting two-agent therapy for sepsis that covers all likely pathogens, with special attention being given to methicillin-resistant species, as the prevalence is increasing among surgical site infections [8].

Patients with C. sordellii infections often are immunocompromised by underlying malignant disease or use of immunosuppressant drugs [1,2]. Infection also has been linked to the use of mifepristone (RU-486) for medical abortion [6]. The effect of mifepristone and misoprostol on C. sordellii pathogenesis is not well understood. Immunoregulatory mechanisms and direct effects on the bacterium are suspected to be involved [9,10]. At present, any association between C. sordellii and these drugs is speculative.

Unlike obstetric or immunocompromised patients, black-tar heroin users acquire C. sordellii infections through the administration of contaminated heroin supplies and use of non-sterile injection techniques [11 –13]. The local anaerobic conditions cause tissue destruction at the injection site and are suspected to help clostridial species proliferate [14]. Subsequent toxin release precipitates edema and necrosis of soft tissue [14]. It is speculated that any exposure of inoculated tissue to anaerobic environments will increase the lethality of this infection [1]. A recent study suggests the administration of antibodies directed against toxin mediators reduces edema and increases the effectiveness of concurrent supportive and operative treatment [15].

Independent of the site of the inoculation, patients typically present with nausea, vertigo, and localized pain from the infection site without exhibiting fever [1]. These signs, along with hypotension and tachycardia, are precipitated by toxin release causing capillary leakage in solid organs and peripheral tissues [1]. Edema of peripheral tissues may lead to compartment syndrome, further escalating tissue damage and metabolic derangement [1].

In regard to laboratory data, marked leukocytosis commonly is observed with C. sordellii infections (>30,000 cells/mm³) [6]. One study reported leukocytosis in 34 of 37 patients infected with various species of Clostridium that resulted in death [16]. A white blood cell count > 75,000/mm³ is indicative of likely death from C. sordellii infection because of rapidly progressive septic or toxic shock [1].

Clostridium sordellii toxic shock is a unique syndrome that has been described by previous authors [1,2,7,11,12]. Two main toxins produce this reaction: Hemorrhagic toxin (TcsH) and lethal toxin (TcsL) [1]. Both TcsH and TcsL work by glycosylating GTPase proteins and disrupting normal cell-cycle regulation, apoptosis, gene transcription, and the cytoskeleton of the cell [1,16]. Until recently, the virulence of TcsL was not known. However, Carter et al. demonstrated that TcsL is essential for lethality. In a mouse model, TcsL increased vascular permeability and led to subsequent tissue edema [17]. The edema and cellular dysfunction produced by TcsH and TcsL eventually alter cellular actin proteins, leading to the breakdown of epithelial cell-cell adhesion proteins [1]. The absence of cellular tight junctions allows capillary fluid to leak into intercellular spaces, producing edema of vital organs and peripheral tissues [1]. We believe this cytotoxic response was evident in our patient when she developed toxic shock syndrome and required massive resuscitation (50 L of colloid and crystalloid in addition to multiple vasopressors). Aggressive resuscitation eventually led to a decompressive laparotomy secondary to abdominal compartment syndrome.

Gwan-Nulla and Casal described toxic shock syndrome after application of a vacuum dressing appliance [18]. Theirs was the first relation described in the literature linking the application of a vacuum dressing appliance to development of toxic shock syndrome. Interestingly, our patient also demonstrated a temporal association between vacuum dressing placement and C. sordellii toxic shock syndrome. A mechanism for this response has not been delineated. We suspect that the enclosed environment of a vacuum dressing causes enhanced production of cytotoxins, or may enhance anaerobic conditions.

Our report is the first case of C. sordellii identified in a breast tissue infection post-operatively. Even more remarkable is the survival of this patient, given C. sordellii's high mortality rate. We believe that aggressive multimodal therapy was the main contributing factor to this patient's survival. Key management of this infection includes consideration of this pathogen early in the presentation on the basis of the signs and symptoms, site culture, maximum surgical debridement of necrotic tissue, early start of broad-spectrum antibiotics, and, possibly, hyperbaric oxygen therapy [19].

Footnotes

Author Disclosure Statement

The authors have no conflicts of interest.

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