Fournier’s gangrene secondary to male’s circumcision – a case report and review of the literature

Introduction

The Fournier's gangrene (FG) was first reported in 1764 by Bauriene, who described a case of scrotal gangrene in a 45-year-old man, an army butcher, that was due to a traumatic injury in the genital region and the left thigh from the horn of an ox, 4 days before the clinical diagnosis. Bauriene stated that in cases of scrotal gangrene, radical surgical debridement of all infected and necrotic tissues is required. The testicles are not always affected, and in these cases, to safeguard the male reproductive system they must not be resected [1]. However, the disease was named after Jean-Alfred Fournier (1832–1914), a Parisian venereologist, who described a series of five otherwise healthy young men with a rapid gangrene of penis and scrotum without any apparent cause [2]. This disease is rare, rapidly progressing and potentially fatal necrotizing fasciitis caused by soft-tissue infections due to a mixture of aerobic and anaerobic microorganisms. Early recognition of the signs and symptoms is essential to prevent fatal consequences, and it consists of several aggressive surgical debridements of the necrotized tissue, several surgical irrigations, broad-spectrum intravenous antibiotics and early resuscitation.

Case

A 57-year-old man with a medical history of type 2 diabetes, treatment with metformin, 500 mg twice a day, presented to our emergency department due to pubic, scrotal and perineal swelling; the glans was sutured to the scrotal skin with nylon 4-0 stiches, the shaft was not visible, being covered with scrotal skin, and a purulent discharge was present from the balanopreputial sulcus (Fig. 1). The patient referred severe pubic pain and pyrexia with a temperature of 39.5 °C, which had started 3 days ago. Two weeks prior the presentation, he underwent a circumcision for recurrent balanoposthitis at an urology clinic external to the hospital. Following the operation, the patient described a fever resistant to the amoxicillin tablets (Fidia Farmaceutici, Italy) with a dose of 1 g twice a day administrated for 1 week, and worsening tenderness and induration of the scrotum. An urgent computer tomography (CT) scan revealed subcutaneous emphysema with a pubic, scrotal and perineal abscess. The laboratory analysis showed an increased white blood cell count (18×10³/μL) with neutrophilia and a higher C-reactive protein level (CRP); the sequential organ failure assessment (SOFA) score was 2, with < 33.3% mortality [3], while the FGSI (Fournier’s ­gangrene severity index) was 5, with the probability of survival 78% [4]. The patient was subsequently taken to the operative room urgently for scrotal and pubic exploration under general anaesthesia. Skin incision was performed along the scrotal raphe, abundant purulent fluid immediately flew out from the scrotum, revealing a necrotizing fasciitis involving the scrotal septum, cremaster and dartos muscles, tunica vaginalis of the right testis and epididymis (Fig. 2). The shaft of the penis was involved by pus and detached by the subcutaneous tissue. Several irrigations were performed with a mixture of saline solution and gentamicin sulphate antibiotic. After removing the scrotal abscess, full debridement of the necrotic fascia and subcutaneous tissue was performed; then the residual portion of the tunica vaginalis of the right testis was sutured by a running absorbable 3-0 suture, to avoid hydrocele. Another longitudinal pubic skin incision was performed, which exposed the abscess with necrosis of the Camper's fascia and Scarpa's fascia. After the purulent fluid removal and irrigation, a fistula was noticed between the pubis and the scrotum. All necrotic fatty tissue and the superficial fascia in the tunnel were excised. The culture obtained by the necrotic tissue showed an infection by Staphyloccocus aureous and Escherichia coli. An intravenous antibiotic treatment was administered with piperacillin/tazobactam (Mylan, Italy) in a dose of 4.5 mg 4times a day and levofloxacin (Fresenius Kabi Italia, Italy) 600 mg, twice a day; then, because of the appearance of an urticarial reaction, only linezolid (Fresenius Kabi Italia) was administrated orally in a dose of 600 mg twice a day, for other 2 weeks. The patient returned to the operating room for another surgical debridement after 2, 4, 8 and 16 days from the first operation until all nonviable necrotic tissue was excised. The patient was discharged from the hospital after 30 days from the first operation, once the wounds were healed. After 3 months, the patient underwent a new surgery. Clinically, he presented a shortened penis with corpora cavernosa entirely covered by the skin of the scrotum (Fig. 3A, B). The procedure was performed under general anaesthesia. The glans was detached from the scrotum and debridement was performed to fully expose the corpora cavernosa surrounded by abundant scar tissue (Fig. 4). The suspensory ligament of the penis identified after removal of the fibrous tissue was stretched using the Z-plasty to increase the outward projection of the penis. Excess scrotal skin was removed and sutured to the root of the penis with 3-0 nylon stitches. Once the entire body of the penis was exposed, it was covered with a full thickness graft measuring 7 cm in width and 9 cm in length, taken from the abdominal fold and secured circumferentially to the body of the penis with a nylon 5-0 continuous suture (Fig. 5A, B). A bolster dressing was made to fix the graft to the recipient area.

After 5 days, the dressing was removed showing a graft take of 90%, a small necrotic area of the skin was surgically removed and left for healing by secondary intention. The patient returned home after 2 weeks. The patient resumed sexual activity after 2 months after the skin grafting. The aesthetic and functional result reported by the patient 1 year from the operation was very satisfactory (Fig. 6A, B).

Discussion

Fournier’s gangrene represents a necrotic fasciitis of genitalia, perineum and abdominal wall, rapidly progressing and potentially fatal, due to soft tissue infection. It is rarely truly idiopathic; however, recent studies indicate that there is an underlying aetiology. Colorectal sources (30–50%), urogenital sources (20–40%), cutaneous infections (20%) and local trauma are frequently identified as the cause of FG [5]. Urogenital factors include an indwelling catheter, traumatic catheterization in case of longstanding urethral stricture, urethral calculi, prostatic biopsy, bladder carcinoma, epididymitis, balanitis, urinary extravasation, circumcision, vasectomy, insertion of penile prosthesis, penile erosion, tension-free vaginal tape procedure, hydrocele aspiration, delayed rupture of ileum neobladder, intracavernosal cocaine injection, and genital piercing, as well as perineal trauma and human bites or scratches as sexual trauma. Males are more affected than females with a ratio of 10:1, and the age of onset is becoming higher (60–70s) [6]. Metabolic disorders such as diabetes, alcohol abuse, human immunodeficiency virus and chronic steroid abuse are often associated with FG; these factors decrease the host immunity, allowing a portal of entry for aerobic and anaerobic microorganisms into the perineum, scrotum or penis, which start a rapid multiplication and spread of infection. Because of its rarity, most of the limited knowledge about FG derives from case reports and retrospective studies with small-size samples. In many cases, literature reports no identifiable source. These findings can be explained by the retrospective non-multicentre studies limitation, with a potential source of bias patients that were transferred to the plastic surgery department from other hospitals in advanced stage. According to literature, the most commonly isolated aerobic microorganisms include Escherichia coli, Pseudomonas aeruginosa, Enterococcus faecalis, Klebsiella pneumonia, Streptococcus agalactiae, and Proteus, while the most commonly isolated anaerobic microorganism are Stafylococcus aureous and Bacteroides fragilis. The mean number of debridement is 3.3 (range 1–4) [7].

The clinical features of FG include sudden scrotal pain and swelling associated with systemic features such as fever > 38 °C [8]. Purulent discharge and area of necrosis, acute renal impairment, anuria, poor hygienic conditions, and necrotic tissue involving the external genitalia are common associated conditions at the clinical examination [9].

The diagnosis of FG is primarily based on clinical findings. The infected area is swollen, dusky and covered with macerated skin and presents with a characteristic feculent odour, attributed to the role of anaerobes in the infection. Systemic signs may be pronounced, usually out of proportion to the local extent of the disease. In those with severe clinical presentation, progression of the gangrenous process to malodorous drainage and sloughing in affected sites results in the deterioration of patient’s overall condition. Crepitus of the inflamed tissues is a common feature due to the presence of gas-forming organisms. As the subcutaneous inflammation worsens, necrosis and suppuration of subcutaneous tissues progresses to extensive necrosis. FG tends to spread along the fascial planes. Infection arising from the anal triangle can spread along the Colles fascia (superficial perineal fascia) and progresses anteriorly along the Dartos fascia to involve the scrotum and penis. It can also pass superiorly along the Scarpa fascia to involve the anterior abdominal wall. The Colles fascia is attached laterally to the pubic rami and fascia lata, and to the urogenital diaphragm posteriorly, thereby limiting the spread of infection in these directions. If the Colles fascia is interrupted, the infection can spread to the ischiorectal fossa and subsequently to the buttocks and thighs. Infection originating from the urogenital triangle, urethra, or periurethral glands can involve the Buck fascia, which initially limits infection to the ventral aspect of the penis. If the infection is not initially treated and the Buck fascia is penetrated, the infection may progress along the Colles and Dartos fasciae.

Patients can rapidly deteriorate as sepsis and multiorgan failure, the most common cause of death in these cases, develop [10]. FGSI is recommended to assess the mortality risk of FG. Described by Laor et al in 1995 [4], by modifying the acute physiology and chronic health evaluation scale to assign a numerical score to 9 parameters that predicted the severity and outcome of FG. FGSI was obtained by combining admission physiological (temperature, heart and respiratory rates) and laboratory parameters (white blood count, serum sodium, potassium, creatinine and venous bicarbonate). With FGSI > 9, the probability of death is 75%, and when the score is ≤ 9, the probability of survival is 78%. Other factors such as the duration of symptoms before hospital admission, extent of body surface area, number of surgical debridements, additional surgical manoeuvres (supra-pubic catheterization or colostomy), and microbiological cultures did not show significant differences [11].

Although the diagnosis of FG is most commonly made clinically, CT can be valuable in patients in whom the diagnosis is unclear or the extent of the disease is difficult to discern. CT has greater specificity for evaluating disease extent than does radiography or ultrasound (US). The CT features of FG include soft-tissue thickening and inflammation, asymmetric fascial thickening, fluid collection or abscess, fat stranding around the involved structures, and subcutaneous emphysema. Post treatment follow-up CT is valuable in assessing for the improvement or worsening of the disease to determine if additional therapy or surgery is needed. US is also useful in differentiating FG from inguinoscrotal incarcerated hernia; in the latter condition, gas is observed in the obstructed bowel lumen, away from the scrotal wall. US is superior to radiography since the scrotal content can be examined along with Doppler blood flow. Soft-tissue air is also more obvious at US than at radiography. Again, CT is superior to both US and radiography in demonstrating FG, its extent, and its underlying causes [12]. The management consists of urgent patient resuscitation, broad-spectrum antibiotic therapy, and surgical debridement. Parenteral broad-spectrum antibiotics are required, the therapy includes triple treatment with third-generation cephalosporins or aminoglycosides, penicillin and metronidazol and is adjusted according to the result of the cultures. In severe forms of the disease, antibiotics from the class of carbapenems are included in the complex processes of the antibacterial therapy [13]. Early surgical debridement is always recommended where necrotic tissue must be performed until the wound bed is clean. The use of adjunctive therapies such as hyperbaric oxygen therapy (HBOT) and vacuum assisted closure are supported in some aspects of the literature and disputed in others. HBOT has bactericide and bacteriostatic effects on anaerobic pathogens; in particular, it also improves bacterial lysis by leukocytes and stimulates collagen formation and superoxide dismutase with tissue survival. Some authors have reported a lower range of mortality for the patients who undergo HBOT, approx. 17.6 vs. 16–30% without HBOT [14].

Once the infection has been treated, there is a necessity of the reconstruction of the surgical wounds with the best functional and cosmetic results and minimal morbidity possible. The options include healing by secondary intention, primary closure or reconstructive procedures with skin grafts or flaps. The skin grafting is the most popular surgical option; its advantages include a simple one-stage procedure and low donor site morbidity as well as the possibility to cover large areas with reasonable functional and cosmetic results [15]. Donor areas also include the abdominal inferior fold, inguinal fold for full thickness skin graft, anterior aspect of the thigh and scrotum for split thickness skin graft; however, this technique should only be performed in case of healthy scrotal wound bed or chronic scrotal pain. Discomfort is often reported in case of using this area. The scar contraction of the graft could be problematic and may limit reconstruction of larger defects. There is also a risk of graft loss due to hematoma, shearing or infection. Full thickness skin graft is often used in penile reconstruction for its minimal contraction compared to thin skin grafts and it does not appear to interfere with sexual and erectile function. Perineal and perianal areas often present reconstructive challenges due to the risk of faecal and urine contamination, tissue maceration and trauma. For this reason, the reconstruction with split-thickness skin grafts is frequently impaired, making them useful options for small defects only [16–17].

Flap reconstruction has been reported for covering defects larger than half the area of the scrotum or extending beyond it. It has been reported in early stage reconstruction and represent an ideal option to cover the testes [18]. The flaps provide more durable protection, without relying on granulation tissue. These procedures are technically more complex and require a longer surgery time and they are associated with higher morbidity. Local advancement flaps, myocutaneous and fasciocutaneous flaps are often described. Scrotal flaps are an ideal option for small defect reconstruction, with low donor site morbidity and significant aesthetic results. They can also be used for penile and perineal reconstruction due to the scrotal skin extensibility [19]. Among fasciocutaneous type flaps, the medial thigh flap is frequently harvested for the scrotal reconstruction. Described by Hallock [20], it is based on the communicating suprafascial vascular plexus of the medial thigh. With its dimension of up to 9 × 20 cm, it provides a good aesthetic result with primary donor site closure. The anterolateral thigh flap is an ideal option for medium and large perineal, penile, anal and scrotal defects. The flap is based on the descending branch of the lateral femoral circumflex artery; the pliability of the tissue, the primary closure of the donor site, and the satisfactory aesthetic results are the main advantages of this type of reconstruction. In case of extensive defects, a pedicled deep inferior epigastric perforator is indicated, although its indications in male genital reconstruction are limited by its bulkiness [21]. In case of urethral defects, usually a full-thickness skin graft or an oral mucosa graft is considered.

Conclusion

FG is an urological emergency with a high mortality rate despite advances in the medical and surgical fields. The early recognition and immediate surgical intervention appears mandatory to face the aggressive nature of the infection. It requires a high clinical level of suspicion, combined with the knowledge of anatomy, risk factors, and aetiology for an accurate diagnosis. Although FG remains a clinical diagnosis, relevant laboratory and radiography investigations can serve as useful adjuncts to expedite surgical management, hemodynamic resuscitation, and antibiotic administration [22].

The gold standard of treatment seems to be the triad: intravenous fluid resuscitation, emergency surgical debridement, and broad-spectrum intravenous antibiotics [23]. The individual characteristics of the defect, patient preference and surgeon experience must be considered to choose the ideal option of reconstruction, which includes technically rapid and simple procedures such as skin grafting with lower donor site morbidity, associated, however, with less satisfactory aesthetic results, fasciocutaneous or perforatos flaps, which probably provide better testicular protection with lower incidence of contracture and higher quality of cover. These surgical procedures are technically more complex, and they are associated with higher donor-site morbidity; furthermore, due to its bulkiness, other operations could be necessary to improve the aesthetic results [24]. FG remains an urgent condition associated with a high mortality rate, requiring immediate treatment. More statistical reports and standard guidelines are necessary to improve the rate of survival.

Roles of authors: All authors have been actively involved in the planning, preparation, analysis and interpretation of the findings, enactment and processing of the article with the same contribution.

Disclosure: The authors declare there are no conflicts of interest regarding the publication of this article. The authors declare that this study has received no financial support. All procedures performed in this case were in accordance with ethical standards of the institutional and/or national research committee and with the Helsinki declaration and its later amendments or comparable ethical standards.

Massimiliano Tripoli PhD

Plastic and Reconstructive Surgery, Department of Surgical, Oncological and Oral Sciences University of Palermo

Via del Vespro 129

90127 Palermo

Italy

e-mail: matripoli@yahoo.it

Submitted: 17.12.2020

Accepted: 10.5.2021