Fournier’s gangrene of the penis caused by Streptococcus dysgalactiae subspecies equisimilis: case report and incidence study in a tertiary-care hospital
© Anantha et al.; licensee BioMed Central Ltd. 2013
Received: 11 March 2013
Accepted: 19 August 2013
Published: 20 August 2013
Fournier’s gangrene is a rare necrotizing soft tissue infection of the scrotum and penis. We report, to our knowledge, the first case of Fournier’s gangrene caused by Streptococcus dysgalactiae subsp. equisimilis (SDSE), a strain of pyogenic β-hemolytic streptococci that is increasingly being recognized as an important human pathogen.
We describe a healthy 59 year-old Caucasian male who presented to the emergency department with Fournier’s gangrene of the penis and scrotum, with extension to the anterior abdominal wall. He underwent urgent surgical debridement of his scrotum, penis, and anterior abdomen. Swabs from the scrotum grew Gram-positive cocci, which were initially identified as Streptococcus anginosus group by matrix-assisted laser desorption ionization–time of flight mass spectrometry (MALDI-TOF MS). However, polymerase chain reaction (PCR) amplification and sequencing of the 16S rRNA gene identified the isolate as Streptococcus dysgalatiae subspecies equisimilis (SDSE). The incidences of invasive S. anginosus group and SDSE infections at the London Health Sciences Centre, a tertiary-care institution in southwestern Ontario, were determined between August 1, 2011 and August 31, 2012, revealing a slightly lower rate of SDSE (3.2 cases per 100,000 population) than other studies.
This case highlights a unique disease manifestation of the emerging human pathogen Streptococcus dysgalatiae subspecies equisimilis that has not been previously reported. This case also underscores the limitations of MALDI-TOF MS in differentiating between closely-related streptococcal species which may have different pathogenic profiles.
KeywordsStreptococcus dysgalactiae subsp. equisimilis Fournier’s gangrene MALDI-TOF MS Species identification
Fournier’s gangrene is a rare necrotizing infection of the male genitalia [1, 2]. It is classically characterized by intense pain and tenderness in the genitals, rapidly progressing to gangrene and septic shock. Risk factors include diabetes , immune compromise, drug use, obesity, and trauma to the perineum [2, 3]. Most cases of Fournier’s gangrene are polymicrobial , and commonly isolated microorganisms include Escherichia, Klebsiella, Bacteroides, Clostridium, streptococci and enterococci . Early therapy is critical, including surgical debridement, broad-spectrum antibiotics, and skin grafting [1–3]. We describe a case of Fournier’s gangrene in a healthy male caused by Streptococcus dysgalactiae subsp. equisimilis (SDSE), initially misidentified as Streptococcus anginosus group. SDSE is a pyogenic β-hemolytic Streptococcus that is emerging as a human pathogen with a similar disease profile to S. pyogenes[4–6]. While it primarily presents as skin and soft-tissue infections, including cellulitis and necrotizing fasciitis , SDSE can also cause endocarditis, rheumatic fever, and streptococcal toxic shock-like syndrome [5, 6]. With an ever-increasing clinical burden, there is a need to accurately identify invasive SDSE infections.
A 59 year-old, previously healthy Caucasian male presented with scrotal and penile pain for six days, and brownish-black discoloration of the scrotum. His past medical and surgical history was non-contributory and he did not take any medications. There was no history of trauma or sepsis in the genital area, and there were no symptoms of dysuria or hematuria. A review of systems was unremarkable. On examination, the patient was alert and oriented, but appeared unwell. He was also febrile (38.3°C), tachycardic (heart rate 116/min), and normotensive (blood pressure 136/79). His lower abdomen was erythematous with palpable subcutaneous emphysema extending to the umbilicus. His penile shaft was swollen and tender, and his scrotum was necrotic. Bloodwork revealed a white cell count of 17 × 109/L, hyponatremia (125 mmol/L), hypochloremia (86 mmol/L), and elevated serum lactate (3.1 mmol/L). The patient’s international normalized ratio (INR) was 1.6, and his serum alanine-aminotransferase (ALT), and serum aspartate-aminotransferase (AST) were elevated at 127 U/L, and 66 U/L respectively. Blood glucose and serum creatinine were within normal limits.
Intravenous treatment with vancomycin (1 g every 12 h), and piperacillin-tazobactam (4.5 g every 8 h) was initiated. The patient was urgently taken to the operating room and underwent extensive debridement of his scrotum, penile shaft, and anterior abdomen, while preserving the testes and abdominal muscles. A small abscess cavity communicating with the necrotizing infection was identified in the right buttock and debrided. A suprapubic catheter was placed for urinary diversion, and the patient was admitted to the intensive care unit. After 48 hours, he underwent a transverse colostomy to divert stool from his perineum, and skin grafting to close his anterior abdominal wounds and penile shaft. His testes were tunnelled into his thighs, and after sixteen days in hospital, he was discharged home.
List of primers used to amplify streptococcal superantigen genes, 16S ribosomal RNA (rRNA), and emm genes
16S rRNA forward
Invitrogen life technologies
16S rRNA reverse
Invitrogen life technologies
emm genotyping primer forward
Invitrogen life technologies
emm genotyping primer reverse
Invitrogen life technologies
Characteristics of invasive SDSE and S. anginosus group infections in a one-year period at London health sciences centre
Rate (per 100,000 population)
Source of organism, n (%)
Cerebrospinal fluid (CSF)
Antibiotic resistance, n (%)
Fournier’s gangrene was first identified in 1883, when the French dermatologist and venereologist Jean Alfred Fournier diagnosed a rapidly progressive gangrene of the genitalia with no discernible etiology in five young men . Now defined as a necrotizing fasciitis of the perineal or genital areas [1, 2], Fournier’s gangrene remains unusually rare, with an incidence ranging from 0.002% to 0.005% of annual hospital admissions . The infection can rapidly spread throughout the perineum, thighs, and torso, subsequently leading to gangrene, septic shock, and death if untreated. Although Group A streptococci were thought to be the sole cause of Fournier’s gangrene , subsequent clinical series have emphasized the polymicrobial nature of the disease [1, 16], which is hypothesized to synergize enzyme production and promote rapid multiplication and spread of infection . The most common causative microorganisms include facultative organisms (E. coli, Klebsiella, enterococci), along with anaerobes (Bacteroides, Fusobacterium, Clostridium, or anaerobic or microaerophilic Streptococci). This case is unique because the patient lacked the typical risk factors associated with Fournier’s gangrene, such as diabetes, immune compromise, obesity, drug use, or genital trauma [1–3], and his infection was caused by Streptococcus dysgalactiae subsp. equisimilis (SDSE).
SDSE, a pyogenic β-hemolytic streptococcus , usually colonizes the upper respiratory, gastrointestinal, and female genital tracts . However, it is increasingly being recognized as an important human pathogen , with a wide spectrum of disease similar to that caused by S. pyogenes, including endocarditis, rheumatic fever, and streptococcal toxic shock-like syndrome . In a recent population-based study, the burden of invasive SDSE infections approximated that of invasive S. pyogenes infections . SDSE primarily presents as skin and soft-tissue infections, including pyoderma, cellulitis, wound infections, abscesses, erysipelas, and necrotizing fasciitis . SDSE contains either Lancefield group antigens C or G, but needs to be distinguished from S. anginosus group strains, which frequently contain the same antigens. The use of MALDI-TOF MS to differentiate between streptococcal species has been established [19–21], although misidentification may occur because of striking similarities in proteomic profiles [22, 23]. The organism in our case was misidentified as S. anginosus group by MALDI-TOF MS, because sequencing the 16S rRNA segment confirmed the isolate as SDSE. While few laboratories report identification of β-hemolytic Group C and G streptococci to the species level , differentiation should not be ignored because SDSE is more invasive than the S. anginosus group , and may have virulence factors similar to S. pyogenes[26, 27]. Although the presence of streptococcal superantigens such as SpeG homologues have been described in SDSE [6, 28], our clinical isolate lacked Group A streptococcal superantigen genes and activity. Nevertheless, our case demonstrates the potential benefit of molecular assays in differentiating closely-related streptococcal species, although further studies are needed to assess their clinical impact. To our knowledge, there are no other reports of Fournier’s gangrene caused by Streptococcus dysgalactiae subspecies equisimils (SDSE). One study isolated Group C Streptococcus from the perineum of a diabetic male with Fournier’s gangrene , but the species was not reported, and a potential role for superantigens was not investigated.
At our institution, the incidences of invasive S. anginosus group and SDSE infections were 3.9 and 3.2 cases per 100,000 respectively (Table 2). The rate of invasive S. anginosus group infection is slightly higher than previous studies , while the rate of invasive SDSE infection is slightly lower. Although rare penicillin-resistant SDSE strains have been reported , isolates from our centre were sensitive to penicillin. Six percent of S. anginosus group isolates, however, were resistant to penicillin, which is higher than other studies . While the mechanisms of resistance have yet to be fully elucidated, the potential transfer of penicillin resistance determinants from related Streptococcal species , together with selective antibiotic pressure, may play a role in the emergence of penicillin resistance in the S. anginosus group [4, 33]. Therefore, the addition of an aminoglycoside to a cell wall-active agent may be appropriate for severe S. anginosus group infections to avoid delayed response of infection . Similar to other studies  showing widespread resistance to macrolides (16-24%) we also observed erythromycin resistance (12% and 7% for S. anginosus group and SDSE isolates, respectively), albeit at a lower rate. Additionally, 12% of S. anginosus group isolates and 14% of SDSE isolates were resistant to clindamycin, suggesting that despite the popularity of macrolide and clindamycin use in infected patients, they may not be appropriate for all cases.
In conclusion, we present a case of Fournier’s gangrene of the penis caused by SDSE, highlighting a unique disease presentation of the organism, and underscoring the limitations of MALDI-TOF MS in differentiating between closely-related streptococcal species which may hixave differing pathogenic profiles.
Written informed consent was obtained from the patient for publication of this case report. A copy of the written consent is available for review by the editor of this journal.
This study was funded by the Division of General Surgery, Western University, and the Canadian Institutes of Health Research (CIHR) operating grant MOP-64176.
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