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Case Report
1 School of Medicine, University of Texas Medical Branch, Galveston, Texas, USA
2 Division of Urology, University of Texas Medical Branch, Galveston, Texas, USA
Address correspondence to:
Frank Ventura
606 Ball Street, Galveston, Texas 77550,
USA
Message to Corresponding Author
Article ID: 100018Z15FV2021
Introduction: Fournier’s gangrene (FG) is a rare necrotizing fasciitis affecting the perineum. Symptoms include tender, edematous scrotal tissue along with fever and can rapidly disseminate. Though FG is frequently a polymicrobial infection of Staphylococcus, Escherichia coli, and Pseudomonas, other pathogens may be involved. Here, we present a rare case of FG that isolated Actinomyces species from its soft tissue culture.
Case Report: A 61-year-old male with a history of uncontrolled type two diabetes mellitus and hypertension presented with a 1-week history of scrotal swelling and pain. He developed urge incontinence, fever, and nausea several days after symptom onset. Vitals demonstrated fever (100.8F) and the patient had bilateral scrotal swelling with erythema and warmth to palpation. Genitourinary (GU) exam was insignificant for crepitus, necrosis, or open wounds. Labs showed leukocytosis. Computed tomography (CT) confirmed the presence of soft tissue gas in the perineum and scrotum. The patient was started on empiric antibiotics and underwent emergency scrotal exploration and debridement. Tissue culture at the time presented with Actinomyces spp. with negative blood cultures. Antibiotics were deescalated to Unasyn. The patient underwent two additional debridements with repeat cultures negative for Actinomyces. A partial split thickness skin graft was performed to reconstruct the scrotum and antibiotics were discontinued.
Conclusion: The importance of early detection and intervention in patients with FG cannot be overstated. This case highlights a rare, likely underreported role of actinomyces in FG with absence of crepitus or necrosis on physical exam and emphasizes the importance of appropriately debriding and tailoring antibiotics to provide effective management.
Keywords: Case report, Fournier gangrene, Infectious disease, Urology
Fournier’s gangrene (FG) is a rare necrotizing fasciitis that commonly affects the penoscrotal and perineal regions locally and may disseminate along fascial layers to infect the abdominal wall and beyond. Symptoms include tender, edematous scrotal soft tissue along with fever eventually progressing to septic shock and death. As FG rapidly disseminates, it is considered a medical emergency that requires urgent surgical debridement and broad-spectrum antibiotics [1],[2]. If left untreated, it can progress to septic shock and death, with the mortality rate ranging between 20% and 40% [1],[3]. While many cases are idiopathic, FG can be secondary to trauma or infection of the perineum [4]. Though FG is frequently a polymicrobial infection of Escherichia coli, Klebsiella, Bacteroides, and Clostridia, other pathogens may be involved. Here, we present a rare case of FG that isolated Actinomyces species from its soft tissue culture.
A 61-year-old obese male with a history of uncontrolled type two diabetes mellitus (T2DM) and hypertension (HTN) presented with a 1-week history of scrotal swelling and pain. He also developed urge incontinence, fever and chills, and nausea several days after the initial symptom onset. Vitals upon ED admission were significant for fever (100.8F) and the patient had bilateral scrotal swelling with erythema and warmth on palpation. Physical exam was insignificant for obvious crepitus, necrosis, or open wounds (Figure 1). Labs showed leukocytosis. Computed tomography of the pelvis confirmed the presence of soft tissue gas in the perineum and scrotum (Figure 2).
The patient was started on vancomycin and meropenem for empiric coverage and brought to the operating room for emergency scrotal exploration and debridement. Urologic surgical incision of the left scrotum was created to drain the loculated regions of purulent material. Gangrenous tissue of the left scrotum was debrided (Figure 3). The left spermatic cord and testicle appeared viable and was preserved. Necrotic tissue disseminated into the right scrotum and required a right hemiscrotectomy with preservation of the right spermatic cord and testicle. General surgery was consulted to evaluate gluteal involvement given proximity of the infection to the anorectal canal. Dissemination in the left gluteal muscle was noted and appropriately debrided. Tissue culture at the time presented as 2+ mixed organisms and 3+ Actinomyces spp. with negative blood cultures. Infectious disease deescalated antimicrobrials to Unasyn 3g IV Q6H to target the specific pathogen. The patient underwent a 2nd debridement two days afterward (Figure 4). Culture at this time came back 1+ Candida spp, at which point the patient was started on micafungin 100 mg IV Q24H. A 3rd debridement was required before closure.
A partial split thickness skin graft (STSG) was performed by Burn surgery to reconstruct the scrotum 14 days postadmission (Figure 5). Burns reperformed STSG on post-admission day 15 with appropriate closure (Figure 6). Antibiotics were discontinued at this time and the patient was discharged to return to clinic in 1 week for follow-up and reassessment of scrotal closure.
Fournier gangrene has been established as a necrotizing fasciitis of the penoscrotal and perineal regions caused by a polymicrobial infection [3],[5]. Classically, it has been understood that endogenous aerobic and anaerobic organisms residing in the genitourinary and colorectal sites are the most common culprits [6],[7]. Staphylococcal, Enterococci, E. coli, Pseudomonas, and Group B Streptococcus species have been underlined in the past literature as the most common organisms (Table 1) [5],[6],[7],[8]. Less likely to be found are anaerobic species, including rare cases of Actinomyces. Actinomyces is an anaerobic gram-positive filamentous rod that produces abscesses and drains through sinus tracts [9]. Actinomyces spp. are also present in the oral mucosa, pharynx, gut, skin, and female genitourinary tract [10],[11]. As FG is typically seen in male patients, the less frequent colonization of the male genitourinary tract may contribute to Actinomyces’ loose relation to the disease. Furthermore, the identification of anaerobic organisms is currently challenging. This difficulty may contribute to the underdiagnosed cases of FG secondary to actinomyces, and clinicians should remain wary of these potential pathogens when treating the disease.
Fournier’s gangrene secondary to Actinomyces species is a rare finding that has not been well established in the literature. Of the few cases, there seems to be an overlap with our patient’s clinical presentation. In 2014, a case report was published conveying the findings of FG in a 73-year-old male with vascular disease who presented with inguinal pain [12]. This patient’s physical exam findings were similar to our patient’s exam as both demonstrated perineal edema, skin color changes, but without crepitus. Computed tomography findings were also similar, illustrating a gaseous collection in the subcutaneous tissue; ultimately, confirming the diagnosis of FG. After cultures were obtained, Actinomyces and Clostridium species were confirmed at the site of infection. Our 61-year-old patient also showed mild findings of scrotal/perineal swelling, erythema, and fever. However, he lacked the severe findings of crepitus and necrosis, which is classically associated with FG. These unique presentations of mild physical exam findings without necrosis or crepitus may be related to FG secondary to Actinomyces spp.
A second case report was published in 2019 regarding an 84-year-old African American female patient. The patient had similar risk factors to our patient such as obesity, type 2 diabetes mellitus, and hypertension. She presented with severe right thigh pain and previously had a bump on her right groin that ruptured in the week prior. She presented with findings significant for fever of 38.3°C, physical exam with inner thigh induration, erythema, edematous, and discolored skin. No bullae or crepitus were noted on exam. Although there was no crepitus as is typical for a FG physical exam presentation, her CT scan showed diffuse soft tissue gas present in the gracilis muscle extending into the perineum, right inguinal region, right labia major, and right ischiorectal fossa confirming a diagnosis of FG [13]. This case of FG involving Actinomyces infection is of similar presentation to our patient.
Overall, the presence or absence of crepitus could be important in making the diagnosis of FG secondary to Actinomyces, however, further research needs to be done to make this association. Some studies suggest Actinomyces does not create an exotoxin that is frequently seen in other pathogens, which may contribute to the lack of crepitus and necrosis [10]. It may also contribute to a more chronic infection compared to the typical FG presentation. Due to underreported cases of Actinomyces’ role in FG, however, the pathogen’s clinical significance is yet to be determined. As seen in our case as well as the report by Tena et al., the patients clinically improved with appropriate antibiotics and repeat debridement.
It is crucial for clinicians to recognize the early signs of FG and provide timely management. Without emergent surgical involvement, patients will need more consecutive debridements, the risk of mortality substantially worsens, and complications of septic shock and acute renal failure become more likely [14],[15],[16]. Not only is initial debridement essential, but physicians must pay close attention to the infecting pathogens and their antibiograms. Common pathogens including Staphylococci, gram negative bacteria, and beta-hemolytic bacteria should be suspected upon empiric treatment, however, cultures with susceptibilities should be obtained to tailor treatment to the offending pathogen. Due to the severity of the disease and wide variations in offending agents, physicians should stay wary when managing FG.
The importance of early intervention in patients with FG cannot be overstated. While appropriate empiric antibiotic therapy is important, due to the wide range of organisms that may be present, early surgical debridement is crucial in minimizing mortality and maximizing the preservation of viable tissue. Alterations to the antibiotic regimen may be needed based on tissue cultures for each individual case. This case highlights a rare, likely underreported case, role of Actinomyces in FG and emphasizes the importance of physicians appropriately debriding and tailoring antibiotics to provide effective management for the patient.
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We would like to acknowledge the Division of Urology at University of Texas Medical Branch for their support in accomplishing this case report.
Author ContributionsFrank Ventura - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be 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.
Wissam Harmouch - Acquisition of data, Analysis of data, Drafting the work, Final approval of the version to be published, Agree to be 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.
Nicholas Tahmassi - Acquisition of data, Analysis of data, Drafting the work, Final approval of the version to be published, Agree to be 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.
David Fernandez - Acquisition of data, Analysis of data, Drafting the work, Final approval of the version to be published, Agree to be 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.
Aditya Srinivasan - Conception of the work, Design of the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be 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.
Joseph Sonstein - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be 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.
Guaranter of SubmissionThe corresponding author is the guarantor of submission.
Source of SupportNone
Consent StatementWritten informed consent was obtained from the patient for publication of this article.
Data AvailabilityAll relevant data are within the paper and its Supporting Information files.
Conflict of InterestAuthors declare no conflict of interest.
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