Fournier's gangrene with Streptococcus Anginosus in the setting of hidrandenitis suppurativa perineal abscess: A case report

Abstract

Introduction

Fournier's gangrene is a rare but life-threatening form of necrotizing soft tissue infection involving the perineal, genital, or perianal region, commonly caused by a mix of aerobic and anaerobic organisms. Initially discovered in dental abscesses, Streptococcus anginosus have been increasingly reported in pyogenic and systemic infections with abscess formation. We present a rare case of perineal abscess that developed into Fournier's gangrene in which the causative pathogen isolated was S. anginosus.

Presentation of case

A 58-year-old male with uncontrolled type 2 diabetes, hypertension and hidradenitis suppurativa of the groin, presented with worsening testicular pain. He was found to have a necrotizing soft tissue infection of the perineum, consistent with Fournier's gangrene. He was successfully treated with multiple surgical debridement and broad-spectrum intravenous antibiotics. He was transitioned to oral antibiotics before transferring to a tertiary care facility for reconstruction.

Discussion

The setting of uncontrolled diabetes and hidradenitis suppurativa may be the likely etiology for this peculiar case of Fournier's gangrene secondary to S. anginosus. Compromised tissue integrity and impaired local immune defenses from these etiologies predisposes to the development of Fournier's gangrene. Historically, these abscesses typically resolve after intravenous antibiotics and incision and drainage. However, the abscess in this case did not resolve but rather progressed to Fournier's gangrene. Perineal abscesses that grow S. anginosus should raise a high index of suspicion for worse outcomes.

Conclusion

In conclusion, we recommend a multidisciplinary approach and rapid diagnosis for the management of S. anginosus in the setting of a perineal abscess, with early aggressive surgical debridement and broad-spectrum antibiotics.

Highlights

• •Streptococcus anginosus cause systemic infections with abscess formation.
• •Uncontrolled diabetes increases risk of Fournier's gangrene.
• •Streptococcus anginosus is a rare, isolated cause of Fournier's gangrene.
• •Fournier's gangrene requires aggressive surgical intervention.

1. Introduction

Fournier's gangrene (FG) is a rare but life-threatening form of necrotizing soft tissue infection involving the perineal, genital, or perianal region, commonly caused by a mix of aerobic and anaerobic organisms, including Escherichia, Clostridium, Fusobacterium, and microaerophilic Streptococcus species. First identified in 1883, Jean-Alfred Fournier described FG in a published case series of five previously healthy young men that presented with decayed fascia, gray exudates, and diffuse tissue destruction. The formation of a perineal abscess occurs when one of the glands within the anus becomes clogged and infected. This may present with a patient having a painful infectious boil that may complicate into an indolent necrotizing fasciitis. FG carries an identifiable etiology that is not limited to young males with risk factors such as diabetes, inflammatory bowel disease, and smoking [1]. Management of FG requires high clinical suspicion as treatment is imperative on early recognition, debridement, and broad-spectrum antibiotics. If left untreated, FG may rapidly progress into multiple organ failure and subsequently death [2]. Streptococcus anginosus group are facultatively anaerobic, gram-positive bacteria that are one of the subsets of Streptococcus viridians group and are found within the normal human flora. Initially discovered in dental abscesses, S. anginosus have been increasingly reported in pyogenic and systemic infections with abscess formation. They are mostly found to colonize in the oropharyngeal, gastrointestinal, and urogenital tracts. Their role in human infection is unclear, but the leading theory is the synergistic interactions with anaerobes and other commensal organisms that lead to abscess formation [3]. Here we present a rare case of perineal abscess that developed into FG in which the lone causative pathogen isolated was S. anginosus.

2. Methods

This case report was written in line with the SCARE guidelines [4].

3. Case presentation

A 58-year-old male with uncontrolled type 2 diabetes, hypertension, and 30-pack-year smoking history presented to the community hospital emergency department with worsening testicular pain. Three days prior to admission, the patient noticed a painful quarter-sized lump around his left testicle. The constant, sharp, and non-radiating pain was initially rated as 5/10 pain in severity, which progressed to 8/10 on the pain scale, prompting him to come to the hospital. The patient also has a history of recurrent hidradenitis suppurativa (HS) on his axillary and groin region which has been treated in the past with incision and drainage (I&D) and antibiotics.

Initial physical examination demonstrated scrotal and perineal edema and scrotal tenderness. No obvious perianal abscesses were noted. He was found to be hypertensive (blood pressure, 165/73 mmHg), tachycardic (heart rate, 117 bpm), tachypneic (respiratory rate, 21 breaths/min), and febrile at 100.4F. His white blood count was 31.5 × 10⁹/L. His glucose was 400 mg/dL. HIV, syphilis, chlamydia, and gonorrhea workup were all negative. Ultrasound of scrotum revealed normal appearing testes with no masses or torsion but did show right epididymal head cyst vs spermatocele measuring 4 mm. Per sepsis criteria, he was started on Vancomycin/Ceftriaxone.

Urology was consulted and performed the initial perineal abscess I&D, with cultures growing Streptococcus anginosus with sensitivities to Cefotaxime, Penicillin, Ampicillin, Ceftriaxone, and Vancomycin. Initial I&D found a perineal abscess extending along the left groin crease and toward rectum. No rectal abscess was found on the rectal exam. All necrotic skin and/or tissue was excised. Despite I&D and Vancomycin/Ceftriaxone, his white blood count increased from 19.1 × 10⁹/L to 33.9 × 10⁹/L. CT scan after initial I&D revealed moderate subcutaneous fat stranding and emphysematous changes extending along the left groin and anterior pelvic soft tissues [Fig. 1A-B]. General Surgery was then consulted for further evaluation and debridement, who suspected high risk of necrotizing soft tissue infection per LRINEC score. Infectious Disease was also consulted and the antibiotic regimen was subsequently modified to Vancomycin, Ceftriaxone, Metronidazole, and Clindamycin, which was added for ostensible synergy in the setting of significant infection.

Fig. 1.

A, B CT of abdomen, pelvis after initial I&D. Findings reveal moderate subcutaneous fat stranding and emphysematous changes extending along the left groin and anterior pelvic soft tissues.

During the second debridement with general surgery and urology, foul-smelling “dishwater” fluid was encountered, and a necrotizing soft tissue infection of the left groin, scrotum and base of the penis was identified, consistent with Fournier's gangrene. Pus was observed at the superior aspect of the base of the penis and base of the left lateral scrotal wall. Necrotic tissue was also found at the mons pubis and proximal cord. No pus was observed in the right scrotum. His WBC further increased to 42 × 10⁹/L the following day. Urology and general surgery brought the patient back into the OR for additional wound exploration, and third debridement of the left gluteal region, scrotum, left cremaster muscle, and penis [Fig. 2]. Based on the operative evaluation, there was not a significant amount of necrotic tissue found to justify the increase in white blood count [Fig. 3A-B]. Local wound care was administered with daily wet-to-dry dressing changes using Vashe three times daily, later de-escalating to two times daily until date of discharge.

Fig. 2.

Pre-op of third debridement. Minimal necrotic tissue noted in the left gluteal region, scrotum, left cremaster muscle, and penis.

Fig. 3.

A, B Post-op of third debridement. All necrotic tissue removed from the left gluteal region, scrotum, left cremaster muscle, and penis.

MRI pelvis was ordered 1 day after third debridement due to increasing white blood counts. Results revealed no drainable residual abscesses. As a result, Vancomycin, Ceftriaxone, and Metronidazole were discontinued. Infectious disease de-escalated antibiotic regimen to Ampicillin-Sulbactam and Fluconazole. Ultrasound of the scrotum three days after third debridement showed normal arterial and venous flow. 10 days after the third debridement, his white blood count decreased to 8 × 10⁹/L. He was later transitioned to oral Amoxicillin-Clavulanate before being transferred to a tertiary care facility for reconstruction surgery.

4. Discussion

The presence of uncontrolled DM and a history of recurrent HS are likely contributing factors in this peculiar case of FG secondary to S. anginosus. Diabetes, alcohol misuse disorder, obesity, and immunocompromised state are all high-risk factors for FG [5]. Studies have demonstrated individuals with uncontrolled diabetes are especially susceptible to soft tissue infections due to dysfunctional neutrophil and macrophage function, and suppression of cytokine production [6]. Additionally, hyperglycemia also increases the likelihood of colonization by pathogenic microbes [7].

HS is a chronic inflammatory skin disorder characterized by the presence of deep-seated abscesses and nodules, and sinus draining tracts commonly located in the axilla, inguinal and perineal area. It occurs because of occlusion and subsequent rupture of hair follicles, triggering inflammatory responses and leading to the formation of abscesses. The chronic inflammation and soft tissue damage in HS predisposes individuals to FG, through compromised tissue integrity and impaired local immune defenses. Historically, these abscesses typically resolve after initiation of IV antibiotics and I&D. However, the abscess in this case did not resolve but rather progressed to FG, requiring Urology and General Surgery to perform multiple debridement. While S. anginosus has been shown to produce gas, the emphysematous changes were thought to be attributed to post-I&D changes [8]. Perineal abscess that grows S. anginosus should raise a high index of suspicion for worse outcomes. Individuals with heart disease, diabetes, kidney disease, and renal failure are associated with higher mortality rates. Unless the original infection originated from the urinary tract, the urinary function is typically spared without any strictures. The coordination of care between General Surgery, Urology, Infectious Disease, and the hospitalist team was imperative to the successful treatment of this fastidious infection. Early aggressive surgical debridement is vital, as patients with delayed treatment had significantly worse outcomes [5]. Additionally, patients that required increased numbers of operations are found to have a 27 % increase in mortality rate [9]. However, despite improvement in recognition and treatment modalities, the mortality rate for FG remains relatively high at 7.5 % [9].

When considering both uncontrolled DM and HS, there are studies that relate uncontrolled DM to the development of FG, particularly individuals with long-standing hyperglycemia and poor glucose control [10]. While HS is not a direct cause of Fournier's gangrene, there have been sporadic reports of individuals with HS developing Fournier's gangrene [11,12]. Ultimately, the frequency and nature of this association remain unclear, and the underlying mechanisms linking the two conditions are not well understood.

5. Conclusion

In conclusion, we recommend a multidisciplinary approach and rapid diagnosis for the management of S. anginosus in the setting of a perineal abscess, with early aggressive surgical debridement and broad-spectrum antibiotics to improve survival rates.

Consent

Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Ethical approval

Ethical approval for this case report was provided by the Institutional Review Board at Community Memorial Healthcare System, Ventura, CA, USA on September 21, 2023.

CMHS IRB 2023-CR306.

Funding

No funding.

Author contribution

Andreas Lau BS – data collection, writing the paper, and critical review

Nobel Nguyen DO – study conception, supervision, writing the paper, and critical review

Alvin Hui BS, MS – writing the paper

Johnson Ong DO – critical review

Michael Salehpour MD – critical review and final approval of the article

Guarantor

Michael Salehpour MD.

Conflict of interest statement

No conflicts from any authors.