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Clinical Infection & Immunity

Case Report

Volume 5, Number 3, September 2020, pages 66-70

Fulminant Myonecrosis From a Minor Puncture Wound Leading to Hip Disarticulation: A Rare Case of Clostridium septicum Gas Gangrene

Gas gangrene, a spreading infection mediated by toxins released by Clostridium species, is a potentially fatal and physically disabling disease due to its often-rapid progression. It carries a high rate of mortality and morbidity. Clostridium perfringens is the most common amongst the species, followed by Clostridium novyi. Clostridium septicum is relatively rare, accounting for only 1.3% of all clostridial infections [1].

Risk factors for developing fulminant soft tissue infections include trauma, immunosuppression, diabetes, and vascular disease. More specifically, gas gangrene is commonly associated with patients of post-surgery or trauma due to the presence of C. perfringes [2, 3].

C. septicum represents a novel subtype among Clostridium species. It causes devastating tissue necrosis in the absence of prior trauma and can present with metastatic myonecrosis, distant from the presumed portal of entry. This particular Clostridium species causes tissue necrosis by toxin formation that disrupts the cell membrane [4]. C. septicum infections are believed to have a greater prevalence among individuals who have malignant disease.

A 60-year-old gentleman presented to the emergency room after stepping on a nail. He received a tetanus toxoid injection and was discharged. Following discharge, he felt well and had no difficulties until 10 days later in the morning, he began experiencing aching soreness over his right thigh. Over the course of a few hours, this rapidly progressed to excruciating pain and swelling, and the patient presented to the emergency department (ED) in the early afternoon in a state of acute delirium.

Evaluation in the ED included a head computed tomography (CT) followed by a detailed clinical examination which disclosed discoloration, swelling, and crepitation of the right thigh. He then underwent a CT of the pelvis and lower extremities, which confirmed subcutaneous air in muscle and fascial regions of the right lower extremity (Figs. 1, 2). A provisional diagnosis of gas gangrene was made. Initial laboratory values were as follows: white blood cells (WBC) 2,500/mm³, hemoglobin (Hb) 11.4 g/dL, creatinine 1.5 mg/dL, albumin 2.6 g/dL and platelets 284 × 10³/mm³. He was consented for extensive debridement of the right leg and transferred to the operating room emergently.

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Figure 1. Computed tomography (CT) of pelvis/lower extremity (coronal view) showing subcutaneous air in right lower extremity.

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Figure 2. Computed tomography (CT) of pelvis/lower extremity (sagittal view) showing subcutaneous air in right lower extremity.

Following the pattern of obvious crepitation and discoloration of the underlying tissues, an elliptical incision was made from the anterior hip region to just proximal to the knee, overlying the quadriceps region of the right thigh. Subcutaneous tissues were divided, and there was obvious thrombosis of the blood vessels with very little bleeding. The underlying muscle groups were quickly exposed, and this revealed obviously necrotic muscle in the region of the vastus lateralis, intermedius, and medialis muscles, as well as the sartorius. Necrotic and nonfunctioning muscle was excised. Failure to respond to electrocautery or to pinch stimulation allowed us to track the regions of necrotic muscle. The necrotic area of the debridement included the medial compartment. The femoral vessels were exposed and were unfortunately surrounded by necrotic muscle. The necrotic muscle was debrided down to the femur and superiorly to the iliac crest and the region of the inguinal ligament. Intraoperative orthopedic and vascular consults were sought. A consensus was reached that the only way to gain definitive control of the necrotizing infection and allow subsequent rehabilitation would be to perform an amputation. The patient needed to be re-draped and prepped to better facilitate the amputation. He was placed in the lateral decubitus position on a beanbag. The abductors were released from the greater trochanter as well as the vastus lateralis and medial compartment musculature. The femoral vessels were isolated and tied using hand ties at the proximal aspect of the incision approximately at the level of the femoral acetabular joint. In addition, the sciatic nerve was isolated and tied off with size 0 Vicryl hand ties and ligated. There was an episode of brisk bleeding due to a cut branch of the external iliac artery.

The leg was completely removed from the body by creating a skin incision and cutting through the subcutaneous tissue with the Bovie cautery knife (Fig. 3). The posterior muscles, particularly the gluteus maximus, appeared to be more viable than the other muscle groups and were preserved for a flap which was brought around to the bottom and the front of the amputation site for future weightbearing. Any devitalized muscle that looked either necrotic or did not respond with stimulus after being touched with the Bovie cautery knife was excised. It was felt that a provisional vacuum-assisted closure (VAC) dressing placement, followed by repeat incision and drainage and final amputation would best serve the patient.

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Figure 3. X-ray showing post-disarticulation status of the right hip joint.

A large wound VAC was placed and set on 125 mm Hg suction. The patient was transferred to the Surgical Intensive Care Unit in critical yet stable condition. Cultures taken from the wound grew out C. septicum. Histology showed extensive myonecrosis and an invasive gram-positive bacilli infection (Fig. 4). He required additional debridement of necrotic muscle the following day. The patient underwent significant ventilatory and hemodynamic support postoperatively for a prolonged period of time. He was weaned from the vasopressors and successfully extubated a week later. He was subsequently discharged to a rehab facility after 3 weeks. Part of his wound required a split-thickness skin graft for closure in several weeks. He had to undergo intensive physical therapy in a rehabilitation facility for months following his surgical intervention. He was ultimately fitted with prosthesis.

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Figure 4. Section of muscle showing numerous gram-positive bacilli surrounding the disintegrating muscle bundles (Brown-Hopps stain, magnification: × 600).

C. septicum is a gram-positive, rod-shaped bacterium that is known to cause rapidly-spreading tissue necrosis, sepsis resulting in multiorgan failure, and death. Trauma to the tissues often acts as the portal of entry for the organism [5].

Malignancy and immunocompromised states are often associated in atraumatic cases [4]. There are case reports of C. septicum infection in patients with primary gastrointestinal malignancy with hepatic metastases [4], adenocarcinoma of the colon with fasciitis of the foot [6], spontaneous aortic dissection from C. septicum aortitis [7], as well as a splenic abscess in a diabetic patient [8].

Larson et al [9] found 10% association with malignancy in a series of 241 patients with Clostridium infection. Interestingly, the infections caused by C. septicum, in particular, have a much higher association with malignancy at 52.6% [10]. In another study (conducted between 1966 and 1993) with C. septicum infected patients, 50% had a primary malignancy, with colon malignancy accounting for 75%, and 40% were cecal [9]. A recent retrospective study found that four of 15 cases of C. septicum positive blood cultures were associated with concurrent colon carcinoma. The authors concluded that all patients with positive blood cultures for C. septicum, even without clinical suspicion of colon malignancy, should be referred for colonoscopy [11].

In addition to gastrointestinal malignancy, other potential risk factors include drug-induced immunosuppression and diabetes [12]. Non-traumatic, endogenous C. septicum tends to disproportionately effect the elderly population [12].

C. septicum’s primary virulence lies in the production of alpha toxin, a cytolysin which produces pores encoded by the csa gene and is secreted as a protoxin and can cause a rapid drop hemoglobin and hematocrit. This alpha toxin differs from the alpha toxin produced by C. perfringes and bears no structural, sequential, or functional relationship [13]. The lethal alpha toxin is responsible for both the hemolytic and necrotizing activity involved in the pathogenesis of gas gangrene. C. septicum also produces beta toxin - a DNase, gamma toxin - a hyaluronidase, and delta toxin - a septicolysin. Septicolysin acts by promoting platelet and neutrophil aggregation and impending neutrophil migration into tissues [14].

Clinically, however, there is no difference between an infection with C. septicum and C. perfringes. Nonspecific findings such as pyrexia, leukocytosis, and pain are the common presentations [15]. Identifying the type of clostridial bacterium helps predict mortality, as rates for C. septicum approach 63% in adults versus 11% in those infected with C. perfringens [15]. The majority of deaths occur within the first 24 h if diagnosis is missed and appropriate treatment measures are not promptly started [16].

Assadian et al [17] describe the significant implications, namely increased mortality, of C. perfringens infection involving the femoral vessels in intravenous drug abusers. Vascular involvement (as in our case) shows extensive infection beyond the realms of expected myonecrosis typically seen with gas gangrene [18]. C. septicum’s ability to invade and permeate healthy tissues differentiates it from its other Clostridium counterparts. Following exposure, healthy tissues become ischemic and necrotic which create an environment that precipitates toxin production and anaerobic fermentation manifesting as gas bubbles and producing crepitus found on clinical examination [18, 19]. This is the pathogenesis of C. septicum induced myonecrosis.

Treatment options include early aggressive surgical exploration both to establish the diagnosis if not already made, as well as to widely debride any devitalized tissue. Only viable musculature should be left behind, indicated by active bleeding with cutting or contraction following stimulation with electrical energy. Most cases, however, warrant a second look surgery, and often include fasciotomies to prevent compartment syndrome. Time is of essence.

Some of the well-known lower extremity scoring systems used in the assessment of the need for amputation in traumatic limb injury are Lange’s predictive salvage index, limb score index, the limb salvage index, mangled extremity syndrome index, and mangled extremity severity score (MESS) [20]. No such scoring system exists for necrotic extremity infections. Optimal functional limb recovery is balanced against the risk of sepsis and death associated with efforts of limb preservations [21]. In cases of significant deficit of neurovascular functions and no expected postoperative residual limb function, amputation may be the right choice. Further indications for amputation may be destruction of the tibial nerve and/or irreparable vascular injury in an ischemic limb. Early amputation and prosthetic fitting can potentially decrease morbidity, repeated debridement, and trips to the operating room (OR), leading to shorter hospital length of stay, reduced expenses, and faster rehabilitation [22].

Very early in the disease course, it is crucial to use concurrent systemic antibiotics to include high dose intravenous (IV) penicillin (18 - 24 million units/day) in divided doses along with IV clindamycin [23]. Clindamycin has a distinctive mechanism of action in preventing toxin release. Additionally, due to the high incidence of mixed infections, an antibiotic with potent gram-negative coverage is often initially started until the cultures and sensitivity are back. Ampicillin-sulbactam, piperacillin-tazobactam, or ticarcillin-clavulanate along with clindamycin or metronidazole are commonly used as empiric treatment in the US hospitals. Some surgeons prefer using vancomycin or metronidazole for C. septicum as these species may exhibit increased resistance to penicillin or clindamycin [24].

Once microbial sensitivities are determined, targeted antibiotic therapy should commence. Intravenous immune globulin has shown promising use, though there is not yet strong evidential data to support routine usage [25]. Hyperbaric oxygen may also play an adjunct role to surgical debridement.

In the presence of bacteremia and septic shock, patients may require inotropic support; therefore, it is often wise to involve the intensive care team early. Long-term management involves major reconstruction and grafting for which specialist plastic surgery input is required [26].

Infection with C. septicum carries a high mortality rate, and early detection is crucial. These infections often present with pain out of proportion to the examination with associated altered mental status. The presence of crepitus can be a late finding, so absence of this sign should not deter one from suspecting this diagnosis.

Our case demonstrates the fulminant nature of gas gangrene. Although uncommon, it is of the utmost importance that management is initiated early. This should include aggressive resuscitation, early systemic antibiotics, hyperbaric oxygen therapy if available, and radical surgical debridement.

Acknowledgments

None to declare.

Financial Disclosure

None to declare.

Conflict of Interest

The authors declare no conflict of interest.

Informed Consent

This patient like mist patients in trauma and emergency general surgery was lost to follow-up. So, we do not have informed consent, but we have tried to not use any identifying pointers in our case report.

Author Contributions

SB and EM jointly did the background search and wrote the manuscript. SB, as the corresponding author, did the final review and edit prior to submission.

Data Availability

Any inquiries regarding supporting data availability of this study should be directed to the corresponding author and can be provided on genuine request.

Disclaimer

This research was supported (in whole or in part) by HCA and/or an HCA affiliated entity. The views expressed in this publication represent those of the author(s) and do not necessarily represent the official views of HCA or any of its affiliated entities.

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