Young Urologists/Residents Corner A chance of 1 out of 50,000 A rare case of devastating Fournier’s Gangrene after circumcision Dr. Fabian Aschwanden Dept. of Urology Luzerner Kantonsspital Lucerne (CH) fabian.aschwanden@ luks.ch
Prof. Agostino Mattei Dept. of Urology Luzerner Kantonsspital Lucerne (CH)
agostino.mattei@ luks.ch
After a 66-year-old man had been diagnosed with devastating Fournier’s Gangrene (FG) after circumcision, he was treated with antibiotics and surgical debridement. He spent six days in the intensive care unit. Subsequently, plastic surgery was performed. After 38 days of hospitalisation, he was successfully discharged. We discuss risk factors, presentation and management of FG. What is Fournier’s Gangrene? FG is an aggressive and potentially life-threatening necrotising bacterial infection, affecting the fascial planes and soft tissues of the perineum and the external genitals. The disease, first reported by Fournier in 1883, is mainly associated with men over the age of 50, suffering from immunodeficiency, particularly diabetes. Further risk factors are malnutrition, malignancy and alcohol overindulgence1. Clinical presentation typically includes painful swelling of the perineum and scrotum combined with systemic signs of sepsis. Due to the rapidity of the infection’s progression along the fascial planes and the related mortality, prompt diagnosis and treatment is crucial.
"Diabetes is a well-known risk factor for FG." Case presentation The 66-year-old patient was in a good general health prior to the incident, despite having suffered from a cough for the last days. His past medical history included diabetes, arterial hypertension, dyslipidaemia, and benign prostate enlargement. Accordingly, his regular medication consisted of a SGLT-2 inhibitor, metformin, aspirin, an HMG-CoA reductase inhibitor, and tamsulosin. Due to recurrent balanoposthitis with secondary cicatrisation, routine circumcision and meatus plastic was performed in an outpatient setting. Two days after the procedure, he appeared in the emergency department. He complained about a painful swelling of the penis and the scrotum, anuria for more than 12 hours and distinct malaise. However, fever and shivering were denied. He was in a poor general condition showing severe hypotension (55/45 mmHg), tachycardia (110 bpm), tachypnoea (30/min), but no fever (36.6°C). The Sequential Organ Failure Assessment (SOFA) score was 7 points (maximum of
24), predicting a mortality of 21.5%2. A livid, oedematous swelling of the scrotum and the penis shaft with beginning partial skin blistering and necrosis as well as an erythema spreading to the suprapubic region were observed. However, skin crackling during examination was not present. Blood results showed leucocytosis (12.3 G/L), a CRP of 210 mg/L, lactate acidosis (lactate 7.8 mmol/L), acute kidney failure (eGFR 21ml/min/1.73m2), while HbA1c was 7.3%. On the CT-scan, diffuse soft tissue swelling of the penis and the scrotum without gas cavities were evident (see figure 1). After immediate intravenous fluid resuscitation and commencement of intravenous piperacillin, tazobactam and clindamycin, the patient was taken to the operating room for debridement. A widespread excision and debridement, including the whole scrotum, was performed (see figure 2). The testes, the glans penis and the deeper structures were not affected by the infection and could be spared. Negative pressure wound therapy (NPWT) and a faecal management system (Dignishield®) for wound protection were installed. After the operation, the patient was admitted to the intensive care unit for a total of six days; he was intubated for four days. Ultimately, he underwent five further debridements. The antibiotics were rationalised according to the microbiology, being positive for streptococcus pyogenes.
"Therapy with SGLT-2 inhibitors and infection of the upper respiratory tract are suspected to increase the risk of FG." By day 20 of his admission, a vertical posteromedial thigh flap and split skin graft were performed by the plastic surgery team (see figure 3). Additional surgical revision had to be performed due to a postoperative bleeding after plastic surgery. Finally, the patient was discharged after a hospitalisation of 38 days and a total of 8 surgeries. Subsequently, he spent another 47 days in rehabilitation before going home. The wound conditions three months after the operation are shown in figure 4. Discussion Circumcision is a rare cause for FG. Galukande et al. report two cases out of 100,000 circumcisions, which suggests an incidence of 2/100,0003. According to a recent study, SGLT-2 inhibitors are considered to be associated with an elevated risk of FG4. Our patient was suffering from diabetes and was under SGLT-2 inhibitor treatment; however, this treatment was paused approximately 10 days before surgery. It remains unknown if it was paused because of the suspected elevated risk of FG or for other reasons. Commonly, FG is a polymicrobial infection; in this case only S. pyogenes was identified. S. pyogenes is a typical pathogen that causes infections of the upper respiratory tract. It is hypothesised that in case of an infection of the upper respiratory tract, S. pyogenes probably enters the blood stream and binds from there to vimentin which is excessively expressed in injured muscle cells5, leading to necrotising fasciitis. As our patient was suffering from a cough, an infection of the upper respiratory tract seems indeed possible but cannot be proven retrospectively.
Figure 3: Status before, while, and directly after plastic surgery
Conclusion FG is a devastating necrotising infection, requiring immediate surgical treatment. Diabetes is a wellknown risk factor for FG. Furthermore, therapy with SGLT-2 inhibitors and infection of the upper respiratory tract are suspected to increase the risk. However, further investigations are necessary to evaluate the association between upper respiratory tract infections, as well as therapy with SGLT-2 inhibitors, and the risk of FG.
4. Bersoff-Matcha SJ, Chamberlain C, Cao C, Kortepeter C, Chong WH. Fournier gangrene associated with sodium-glucose cotransporter-2 inhibitors: A review of spontaneous postmarketing cases. Ann Intern Med. 2019;170(11):764-769. doi:10.7326/M19-0085 5. Bryant AE, Bayer CR, Huntington JD, Stevens DL. Group A Streptococcal Myonecrosis: Increased Vimentin Expression after Skeletal-Muscle Injury Mediates the Binding of Streptococcus pyogenes . J Infect Dis. 2006;193(12):1685-1692. doi:10.1086/504261
References
Figure 1: CT scan showing diffuse soft tissue swelling of the scrotum
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Figure 2: Wound conditions after debridement
1. Chennamsetty A, Khourdaji I, Burks F, Killinger KA. Contemporary diagnosis and management of Fournier’s gangrene. Ther Adv Urol. 2015;7(4):203-215. doi:10.1177/1756287215584740 2. Vincent JL, Moreno R, Takala J, et al. The SOFA (Sepsis-related Organ Failure Assessment) score to describe organ dysfunction/failure. On behalf of the Working Group on Sepsis-Related Problems of the European Society of Intensive Care Medicine. Intensive Care Med. 1996;22(7):707-710. http://www.ncbi.nlm.nih. gov/pubmed/8844239. Accessed January 25, 2020. 3. Galukande M, Sekavuga DB, Muganzi A, Coutinho A. Fournier’s gangrene after adult male circumcision. Int J Emerg Med. 2014;7(1):4-7. doi:10.1186/s12245-014-0037-0
Figure 4: Wound conditions three months after the operation
March/May 2020