Use of Negative Pressure Wound Therapy to Successfully Treat Postoperative Pyoderma Gangrenosum Following Laparoscopic Surgery: A Case Report

PG is a rare skin disorder with an estimated annual incidence of 3 to 10 cases per million, with no predisposition by sex.¹ It is defined as a neutrophilic dermatosis, rather than an infectious or gangrenous skin disease. A link exists between PG and irritable bowel disease, rheumatoid arthritis, or hematological disorder in 50% to 75% of patients.¹ In 20% to 30% of affected patients, an exacerbation is preceded by trauma or surgery.^2,3 Because of the rarity and variable clinical presentation of PG, diagnosis is often delayed, which leads to a significantly prolonged healing process. The authors of the present case report believe that highlighting cases of PG can increase clinicians’ level of suspicion for the diagnosis and lead to better understanding and treatment of the disease.

A 75-year-old white male presented to the emergency department with suspected surgical site infection at the suprapubic trocar incision site through which laparoscopic appendectomy for acute appendicitis had been performed 2 weeks previously. His relevant medical history consisted of gout, multiple surgical interventions for anal fistula, benign prostatic hyperplasia, and primary myelofibrosis.

The patient presented to the emergency department because of fever, feeling unwell, and increasing redness at the suprapubic surgical wound. Upon inspection, a superficial necrotic skin lesion with surrounding redness was seen (Figure 1). The wound was quite tender to palpation. Blood work showed a mild inflammatory response, with normal leukocytosis of 7.4 × 10³/µL and C-reactive protein level of 49 mg/L.

The patient was admitted to the surgical ward, blood cultures were taken, and intravenous antibiotic treatment in the form of piperacillin/tazobactam was initiated. On day 1 of hospitalization, fever continued and a further increasing redness at the suprapubic wound was observed. Tigecycline was added to the antibiotic treatment regimen. On day 3, owing to poor evolution of the wound despite broad-spectrum antibiotic treatment, surgical debridement was performed, followed by initiation of NPWT. A black sponge was used, and suction was continued at −125 mm Hg. Histopathologic findings of surgical debridement showed signs of abscess formation with numerous polynuclear infiltrations and small bacterial colonies. Cultures of the specimen showed only epidermidis and S capitis.

The first bandage change 3 days later was witnessed by the surgical team, and the suspicion of PG or another skin disorder was mentioned (Figure 2). A full-thickness biopsy specimen from the edge of the wound was examined and showed ulcerations with polynuclear infiltration. Gram stain and Ziehl stain results were negative. The diagnosis by exclusion of PPG was confirmed according to criteria generated using a Delphi consensus exercise.⁴ NPWT was continued after physiologic cleansing of the wound and surrounding skin. Oral corticosteroid therapy in the form of prednisolone was initiated at a dose of 32 mg once daily, with dose reduction by 8 mg every 5 days. Corticosteroids were then continued at a dose of 4 mg once daily for the following 3 months. Broad-spectrum antibiotics were discontinued, and doxycycline 100 mg every 12 hours was initiated because of its immune-modelling properties. Cyclosporin was also initiated at a dose of 100 mg every 12 hours, with close drug dose monitoring.

The patient was discharged on day 11, and NPWT was continued in an outpatient setting. Bone marrow examinations showed no disease progression of his known myelofibrosis. Complete wound closure was noted approximately 2 months after admission (Figure 3). All medication was discontinued after 3 months after discharge.

Three weeks after cessation of medical therapy, the patient presented with a bullous lesion on the thigh (Figure 4). Local wound care was initiated by means of physiologic cleansing and application of a nonadhesive bandage. Prednisolone was initiated at a dose of 32 mg once daily, with a decrease in dose of 8 mg every 5 days followed by a maintenance dose of 4 mg thereafter. At the time of presentation with a bullous lesion, cyclosporin and doxycycline were both initiated at a dose of 100 mg every 12 hours. Rapid healing occurred over a 2-week period. All medication was discontinued after 1 month. At 1-year follow-up, no new lesions had been reported.

PG is a rare neutrophilic skin disorder characterized by a variable clinical spectrum. It is a diagnosis of exclusion, because no definitive laboratory or histopathologic testing is available. Extensive neutrophilic infiltrate in full-thickness biopsy is a nonspecific but major criterion for diagnosis, however.⁴ Thus, PG is often misdiagnosed, and treatment is predominantly based on expert opinion and small clinical trials. The exact pathophysiology of PG is not fully understood, but there is an interaction between genetics, neutrophil dysfunction, and pathologic inflammatory processes.⁵

In contrast, PPG has a less variable clinical course, because it predominantly presents as a necrotic ulcerative lesion with surrounding erythema. Tolchakjov et al retrospectively described an average time interval of 11 days.⁶ Females were more susceptible than males. The breast and abdomen were reported to be the most frequently affected regions.⁶ In a 2016 literature review, only 34% of patients had associated systemic disease, of which chronic hematological disorder was the most prevalent.⁷

The management of PG consists of diagnosis and treatment of a possible underlying cause, avoidance of triggers, adequate wound management, adequate pain management, and topical, systemic, or targeted immunomodulatory therapies. Local or systemic corticosteroids with dose tapering comprise the first-line therapy for treating patients with PG. Cyclosporin, which is a glucocorticoid-sparing immunosuppressive agent, is a first-line therapy for PG.⁸ Increasing evidence is emerging concerning biologics as therapeutic options for PG.⁵ Antibiotics are used in patients with PG only if superinfection is suspected. Wound care achieved by NPWT has previously been reported to be a valuable strategy,⁹ and the results of the present case study reinforce such findings. NPWT should be considered in appropriate cases. Multiple authors have reported that skin grafting combined with systemic immunosuppression has led to excellent results in select cases.⁹ Surgical debridement may be indicated to remove a large amount of necrotic tissue or if superinfection cannot be controlled by conservative therapy.⁹

Limited data are available regarding prevention of PPG. In a retrospective analysis, Xia et al³ stated that more invasive procedures and chronic PG are the most important risk factors for recurrence. They found prophylactic immunosuppression to be of no benefit.

The case reported herein highlights the misleading presentation of PPG. In the postoperative period, wound problems are most often associated with surgical site infections. However, prolonged antibiotic treatment may prolong healing times owing to delayed diagnosis. This case may serve as an example that even minimally invasive techniques such as laparoscopy may exacerbate PG. Surgeons must be aware of this skin condition, which may present as a common complication, but in fact necessitates a unique treatment approach.

The main limitation of this study is that it involves only 1 case. Thus, it is not possible to make general recommendations for the treatment of PPG.

PPG is a rare skin disorder, and diagnosis is often delayed. In the 75-year-old male patient with PPG and underlying myelofibrosis discussed in the present report, initial treatment with antibiotics and debridement failed. A clinically stable condition with no new lesions was achieved by means of NPWT combined with immunosuppressive therapy.

Early recognition and initiation of immunosuppressive therapy with local atraumatic wound care are important to avoid long healing times and complications in patients with PPG. NPWT results in rapid wound healing and should be considered as appropriate. Surgical debridement may be considered in cases of extensive necrosis.