Fournier’s gangrene is an aggressive disease with rapidly progressive, infectious, necrotizing, life threatening fasciitis [1]. The clinical presentation is heterogeneous, ranging from insidious onset and slow progression to rapid onset and fulminant course, with systemic symptoms such as fever, chills, asthenia, nausea, vomiting, and signs of infection (such as rapid vesicle formation, necrosis, foul-smelling discharge with edema, tenderness, and redness). Fournier’s gangrene can occur in parts of the body such as the perineal, genital, and perianal regions and the infection can spread to the abdominal wall, leading to soft tissue necrosis and sepsis [1–3]. Necrosis can be caused by anaerobic and aerobic bacteria in the soft tissue blood vessels [4]. The mixed infection of anaerobic and aerobic bacteria in the soft tissue blood vessels synergistically activates different proteins (enzyme), resulting in rapid platelet aggregation, intravascular coagulation, and thrombosis, which leads to ischemic changes in the tissue. This leads to irreversible soft tissue necrosis as the partial pressure of oxygen, and blood flow in the vessels decreases, and bacteria overgrowth in the skin and subcutaneous tissues occurs [3,5].

Fournier’s gangrene is an extremely rare condition, appearing in urogenital and anorectal diseases, as well as in immunocompromised patients with diabetes, obesity, and malignant neoplasm. Delayed treatment of the infection results in mortality in up to 90% of cases due to the development of septic shock and its associated complications. Unfavorable prognosis in this disease highly depends on the timing of medical care received [6,7]. One of the most important determinants of overall outcome is early recognition and extensive surgical debridement upon initial diagnosis [4,6,7]. If it is not treated immediately, sepsis, toxic shock syndrome, and multiorgan failure may occur with a mortality approaching 100% [1,8].

The treatment of Fournier’s gangrene requires: (1) repeated invasive and painful aseptic dressing procedures (the number of which cannot be guaranteed); (2) an anesthesiologist, in addition to a lot of manpower; (3) medical resources to be mobilized for general anesthesia every time; (4) moving the patient to the operating room and all the risks involved; and (4) treatment of complications arising from multiple administrations of general anesthesia over a relatively short period of time.

In this case report, experience with a repeated invasive procedure is shared using noninvasive ventilation (NIV) without general anesthesia in an intensive care unit (ICU) to treat Fournier’s gangrene.

An 82-year-old woman presented to the Emergency Department with perineal pain, swelling, and weakness in both legs. She had a history of hypertension, uncontrolled Type 2 diabetes, and had a low anterior resection for rectal cancer in 2005. On physical examination, swelling and redness of the perineum was observed, with tenderness and heat sensation. The patient’s body temperature, when measured in the Emergency Department, was 36.7 °C, her heart rate was 113 bpm, and her blood pressure was 86/60 mmHg. In the initial laboratory findings, the level of C-reactive protein was 30.64 mg/dL, the white blood cell count was 10,980/uL, the segmented neutrophil rate was 88.4%, the level of procalcitonin was 25.42 ng/mL, the glucose measurement was 791 mg/dL and HbA1c was 12.2%. The Sequential Organ Failure Assessment score was 11. Abdominopelvic computed tomography was performed for further evaluation and revealed extensive subcutaneous emphysema and fat infiltration along the right labia majora, perineal/inguinal area, and lower abdominal wall, which was consistent with the necrotizing fasciitis called Fournier’s gangrene (Figure 1).

The patient underwent emergency perirectal abscess drainage, necrotic tissue debridement with perineal resection, and a transverse loop colostomy. After surgery, the patient was transferred to the intensive care unit (ICU) and was treated with vasoactive agent, and broad-spectrum antimicrobial agents for septic shock due to Fournier’s gangrene. Necrotic tissue debridement, wound cleaning, and moist dressing were performed in the operating room under general anesthesia daily from postoperative Days 1–7 and negative-pressure wound therapy (NPWT) was applied on postoperative Day 8.

On postoperative Day 9, the patient required extubation, but repeated tracheal intubation and extubation with general anesthesia every day was deemed a significant therapeutic burden given the patient’s age. It was also burdensome in terms of manpower, and medical resources such as booking the operating room, and moving the patient at the designated time. Therefore, the decision was made to apply wound debridement and NPWT without transferring the patient from the ICU to the operating room.

This process requires the patient to be in the lithotomy position and causes considerable pain during the procedure; therefore, it was decided to lightly sedate the patient. However, when using drugs for sedation, possible airway problems had to be considered, so, noninvasive ventilation was used. For the lithotomy position, the patient was moved to the bottom of the ICU bed, a leg cushion was placed on the height-adjustable shelf of the bed, and the patient’s legs were positioned and secured (Figure 2). The ICU bed was adjusted to a reverse Trendelenburg 5° to direct the flow of water downwards for wound irrigation. Extubation was performed on the postoperative Day 12, after which, NIV was used for periodic wound irrigation, curettage, and NPWT every 3 days under light sedation until postoperative Day 36.

The total number of operations performed in the ICU was 9, 2 of which were performed with intubation, and the remaining 7 were performed by applying NIV after extubation. Two operations performed under intubation, on postoperative Day 10 and 12, took 85 and 75 minutes, respectively. Extubation was performed after the operation on the postoperative Day 12. NIV was applied following the operation on postoperative Day 15. The operation time was 80 minutes on postoperative Day 15, 70 minutes on postoperative Day 17, 69 minutes on postoperative Day 19, 60 minutes on postoperative Day 23, and 30 minutes on postoperative Day 32.

The drugs used for light sedation were 2% propofol (20 mg/mL; 1,000 mg in 50 mL/vial) and remifentanil (20 μg/mL; 2 mg in 100 mL). Propofol 14.16 μg/kg/min (= 3 mL/hr) and remifentanil 0.01 μg/kg/min (= 3 mL/hr) were used as induction dose, the dose was gradually increased according to the patient’s sedation status, the progress of the operation, and up to a maximum dose of propofol 47.21 μg/kg/min (= 10 mL/hr) and remifentanil 0.05 μg/kg/min (= 10 mL/hr). The sedative dose was optimized by reducing the maximum dose of propofol 33.05 μg/kg/min (= 7 mL/hr) and remifentanil 0.03 μg/kg/min (= 7 mL/hr) from the 3^rd operation using NIV.

During the operation, the patient was lightly sedated with good compliance to NIV. Following the operation, the sedative being used was tapered and stopped without any problems. On postoperative Day 37, local flap and coverage with split thickness skin graft was performed for perineum reconstruction in the Plastic Surgery Department (Figure 3).

We report a case of Fournier’s gangrene, following the control of septic risk (emergency debridement, perineal resection, and a colostomy, wound debridement performed under general anesthesia daily for 1 week), NPWT was administered without general anesthesia and using only NIV. Fournier’s gangrene is a rare disease with many predisposing factors. Given the patient’s initial HbA1c, it was reasonable to assume that the patient’s underlying disease, uncontrolled Type 2 diabetes, influenced the onset of Fournier’s gangrene.

From January 1, 2020 to December 31, 2023, a total of 18 patients were diagnosed and treated for Fournier’s gangrene at our hospital. Among them, 15 patients were male, 4 of the 18 patients were in septic shock at the time of presentation, and 1 patient died (an elderly patient aged 90 years). Considering the incidence of Fournier’s gangrene, our experience in treating it is not significantly different from other hospitals.

Fournier’s gangrene is a serious and complex disease that requires a combination of 3 treatments: early surgical debridement to remove necrotic tissue for source control, broad-spectrum antimicrobial therapy, and intensive management [9]. In our hospital, aggressive wide excision is performed, depending on the extent of the initial lesion, designed to completely remove necrotic tissue. If the infection invades the perineum, a colostomy or urostomy will be performed under the decision of the surgeon to prevent further contamination. Because complete debridement of necrotic tissue is difficult to achieve in a single surgical exploration, re-exploration, and aseptic dressing are performed every 24–72 hours to assess if the lesion is improving or worsening. Once necrotic tissue is completely removed and the wound has begun to heal, NPWT is then considered. Intensive management is performed in ICU. To prevents additional infection of the wound, appropriate antibiotics are used and aseptic dressing is performed every 24–72 hours depending on the wound healing condition. Reconstruction is surgically performed in a Plastic Surgery Department using surgery such as local flap and coverage with split thickness skin grafting

Upon diagnosis, the patient in this case underwent emergency surgery and appropriate sepsis control treatment. In the 1^st operation, the patient’s wound was identified as an extensive gas foaming necrosis from the right ischiorectal fossa to the right-side labia majora, perineal and inguinal areas, and lower abdominal wall. Therefore, extensive debridement was performed on the extensive necrotizing fasciitis. According to the World Society of Emergency Surgery and Surgical Infection Society Europe, NPWT is recommended after complete removal of necrotic tissue [10]. Since the initial wound of the patient was extensively advanced, complete removal of the necrotic tissue was not possible in the 1^st operation. Moreover, there was a possibility of further necrosis of the surrounding tissues, so early application of NPWT would likely worsen the patient’s wound. Therefore, aseptic dressing was performed daily to identify and debride additional necrotic tissue, and NPWT was applied after the removal of most of the necrotic tissue.

Following the application of NPWT, the process required for the healing of the patient’s wound required general anesthesia, because it is a very painful process involving repeated and considerable amounts of necrotic tissue removal and wound irrigation. Multiple intubations and extubations under general anesthesia for frequently repeated procedures is painful for the patient and can lead to complications such as laryngeal trauma, ventilator-associated pneumonia, and adverse drug reactions from the administration of multiple agents such as sedatives. Therefore, multiple intubations and extubations under general anesthesia for frequently repeated procedures are a concern as they are thought to be a risk factor for poor patient outcomes despite the patient receiving the appropriate treatment. They also result in a significant drain in medical resources. On the other hand, the process can be performed without intubation under local anesthesia, but the patient’s position and pain are not easily controlled, which means that the procedure is likely to be poorly performed. To address these issues, NIV was used to perform invasive procedures under light sedation in the ICU.

During the operation, light sedation was applied for maintenance of the patient’s position and pain control whilst NIV was applied (in consideration of airway problems). NIV is a safe and effective alternative to mechanical ventilation in patients with respiratory failure, that can preserve respiratory defense mechanisms, and avoid the many side effects and complications which are associated with mechanical ventilation, including pneumonia, pneumothorax, barotrauma, and adverse effects of sedatives [11]. Sedation during NIV can be considered a relative contraindication. However, successful NIV application depends on patient acceptance and compliance. Anxiety, agitation, pain, and respiratory distress can increase the likelihood of NIV failure and ultimately affect patient safety. Therefore, in terms of patient safety, use of sedation is associated with and important for successful application of NIV by increasing patient compliance [12].

Performing aseptic dressing under local anesthesia without sedation is not effective in maintaining lithotomy position and controlling pain. Conversely, pain can reduce patient compliance with NIV which can lead to respiratory problems, and prolonged procedure times, increasing the risk to patient safety. By maintaining patient comfort the use of appropriate sedation is not essential for the effective application of NIV in synch with a ventilator but, it may be beneficial in certain circumstances [13]. Because the potential risks of over-sedation and the need for unintentional intubation cannot be ruled out, intubation should be available in the ICU at all times during procedures, and an intensivist experienced in sedation and intubation should be present during the procedure to continuously assess the degree of sedation. As a result, light sedation for pain control and position maintenance, and NIV for airway problems have complementary effects.

There are no systemically conducted studies on the selection of appropriate sedatives in NIV [12], and there are no guidelines or formulae for situations requiring sedation, so it is a clinically judged and implemented decision. Generally, it can be a hindrance during the application of NIV because it is necessary to closely monitor the patient for decreased consciousness. However, in a North American and European multicenter study to increase patient compliance, sedatives were used in 85% of cases and analgesics in 94% of cases [12,14]. Respiratory inhibitory effects from sedative use must be considered. Regarding the sedative to be used in the application of NIV, dexmedetomidine and ketamine appear to have the most appropriate pharmacological profile, followed by propofol and remifentanil, but there are no studies related to the specific drug or type of drug preferred for NIV, so the use of sedation in NIV seems mostly empirical and is probably unstructured, with recent studies showing favorable outcomes with dexmedetomidine [12,14].

There were a total of 9 cases where aseptic dressing was performed in the ICU, 7 of which were performed by applying NIV under light sedation. The average time required when aseptic dressing was performed during NIV was 57 minutes. The initial stage took a relatively long time due to the patient’s extensive wound and trial and error in the process of applying NIV. However, the patient’s wound gradually improved, and in addition, the operation preparation time such as optimization of the application of NIV and lithotomy position was shortened. The total operation time required was reduced to 30 to 45 minutes, resulting in 15 to 30 minutes less than the average operation time. Furthermore, by optimizing the manpower required for surgery, it was possible to operate efficiently in the ICU with a total of 4 staff (1 intensivist as a scrub nurse, 1 intensivist providing sedation and airway management, 1 surgeon, and 1 assistant). If the operation was performed in the operating room, at least a surgeon, anesthesiologist, assistant, scrub nurse, and a moving person to the operating room may be required. Furthermore, the arrangement of anesthesiology and other department medical personnel is difficult from day to day depending on the overall operation schedule and requires more manpower compared with the initial operation. There was no significant difference in the time required in the operating room and the time required in the ICU. A shortened operation time reduces the risk of applying NIV in patients with altered consciousness due to sedation. There have been no patient safety issues, and there are also benefits in terms of medical resources through efficient manpower management.

This is not the first time NIV has been used for invasive procedures. There have been published cases of an esophagogastroduodenoscopy performed under sedation with NIV to prevent sleep apnea, which can occur during sedation endoscopy [19,20]. However, this is limited to endoscopy and has been used in a very limited number of cases for surgery and surgical procedures.

Surgical procedures performed under light sedation in the ICU are not limited to NPWT. Sedation is expected to be a positive consideration for patients requiring periodic invasive surgical procedures who are burdened by repeated general anesthesia.

This case also illustrated certain limitations in application. Firstly, the medical staff, including ICU nurses, must be well trained. Without adequate training and familiarity with the use of NIV, they may not be able to respond appropriately in critical moments. Secondly, there should be a doctor performing the procedure, and a specialist controlling the sedation and the NIV. If constant monitoring is not possible, general anesthesia should be strongly considered. However, despite these limitations, we believe that this case report is significant in that the number of general anesthesia sessions were significantly reduced using NIV. Therefore, we believe that this should be further investigated.