Hemiarthroplasty in a patient with femoral neck fracture and pyoderma gangrenosum: a case report and review of the literature
Pyoderma gangrenosum is a rare ulcerating skin disease of unknown etiology, making its coincidence with orthopedic trauma a rare challenge. Patients are at risk of progression of the existing lesions and development of new lesions upon skin injury when surgical procedures are performed. To our knowledge, this is the first report in the literature of disease unrelated surgery during active pyoderma gangrenosum.
We present a case of femoral neck fracture in a Caucasian patient with concurrent pyoderma gangrenosum localized in the axilla. Hemiarthroplasty was safely performed after disease activity was reduced with systemic corticosteroids. Tissue-protective wound closure was used together with perioperative corticosteroids and antibiotics. No signs of pyoderma gangrenosum developed at the surgical wound site, and the axillary lesions showed constant improvement until healing with scar tissue.
In our patient, the preoperative steroid treatment, perioperative antibiotics, and soft tissue protective surgical technique led to successful management of this rare coincidence.
KeywordsPyoderma gangrenosum Hemiarthroplasty Orthopedics Trauma surgery Pathergy
The necessity to perform disease-unrelated surgery during active pyoderma gangrenosum (PG) is rare, putting surgeons in the difficult position of making decisions without underlying evidence. PG is a rare skin disease with unknown etiology. Brunsting et al. described the condition in 1930 and related it to staphylococci, but later research showed an autoimmune pathology to be more likely [1, 2]. In approximately 50% of cases, PG is related to trauma, surgery, malignancy, systemic inflammatory disease, and inflammatory bowel diseases such as ulcerative colitis and Crohn’s disease . The pathergy phenomenon describes the development of additional lesions or progression of existing ones during active PG when skin integrity is interrupted . It has been reported to occur at sites of intravenous lines, injections, and surgical wounds . Pathergy puts the patient at risk for complications, resulting in recommendations not to perform any surgery on PG lesions; other authors recommend surgical management of PG ulcers in a less active period of the disease [6, 7, 8]. Systemic corticosteroids, especially prednisolone, and immunosuppressants such as cyclosporine A are the first-line medical therapies for PG. Other options include various local treatments, hyperbaric oxygen therapy, or debridement and skin grafting. Literature suggests that surgical treatment of PG ulcers can be successful after an initial phase of anti-inflammatory treatment [9, 10]. Based on the case of a patient with PG but no active lesions, general recommendations for their wound management, focusing mainly on reduced skin puncturing, have been published . To our knowledge, there is no study or case report to guide a surgeon’s decision whether and how to operate on a PG-unrelated condition such as orthopedic trauma in patients with coincident active PG.
Regular follow-up with clinical and x-ray examinations at our department showed no development of PG lesions at the surgical site (Fig. 2d). At final follow-up in our department 7 months after injury, the patient presented pain-free with unlimited range of motion of the right hip and full mobility. The PG lesions had healed with scar tissue, and she has had no further disease activity since her initial diagnosis of PG.
This unique case of orthopedic trauma during active PG suggests that disease-unrelated surgery using a soft tissue protective technique, preoperative immunosuppression, and perioperative antibiotics can be performed successfully. Due to the rarity of the disease, no larger studies on PG and either related or unrelated surgery are available, but the general rationale for operations during the course of PG is based on the principle of avoiding pathergy.
Case studies have shown that surgical management of PG lesions without prior medical treatment led to worsening of the ulcers. In most of these reported cases, the PG was misdiagnosed as wound infection and treated with antibiotics and surgery alone. Antibiotics consistently showed no effect, and surgery resulted in worsening of the lesions when no appropriate PG treatment was initiated [6, 12]. Some authors even reported cases of lethal complications due to pathergy after surgical treatment of ulcers . Evidence for the effectiveness of soft tissue protective surgery in PG is described in the case report by Long et al., who compared two surgical wound closure techniques in a patient with a medical history of PG. The 61-year-old woman underwent bilateral hip arthroplasty, but unlike our patient, she had no PG lesions at the time of surgery and received low-dose prednisolone as a maintenance treatment. The first arthroplasty was performed using interrupted silk sutures to close the skin and led to multiple ulcers at the suture entry/exit sites. Therefore, the authors used continuous subcutaneous Dexon® sutures and Steri-Strips® for skin closure of the second arthroplasty. After this surgery, only two smaller and less florid ulcers occurred. On the basis of this experience, the authors concluded that soft tissue protective surgery reduces the risk for pathergy in patients with PG . She et al. described a case of avascular necrosis of the femoral head and a femoral neck fracture in a patient with a history of PG. They successfully performed total hip arthroplasty with perioperative corticosteroid therapy and did not describe any specific soft tissue protective management .
Several reports have shown surgical management of PG lesions such as debridement and skin grafting to be successful if performed in a less active phase of the disease after initial treatment with immunosuppressants [7, 14]. Niezgoda et al. reported a case where ulceration could not be stopped by corticosteroids alone, but after application of hyperbaric oxygen therapy, the disease progression was halted, and debridement with consecutive split skin grafting could be performed successfully. The authors were the first to describe postoperative use of vacuum-assisted closure that supported the complete healing of the lesion .
It must be considered that long-term exposure to corticosteroids or other immunosuppressive drugs puts patients at risk of developing life-threatening infections and other complications. Case reports have shown that the average treatment time can be reduced by surgical management of PG ulcers . Summarizing other authors’ experiences, it can be inferred that surgery for PG lesions can reduce the duration of the disease without high risk for complications as long as the following principles are applied: ongoing immunosuppression, reduced disease activity, and soft tissue protective surgery.
The current concept for development of PG is of autoimmune etiology. Several cases in the literature did show bacterial growth on ulcers, but overall PG lesions are usually aseptic. This and the responsiveness to immune-suppressive therapy suggest that the theory of PG being caused by bacteria is obsolete [16, 17].
The role of antibiotics in PG treatment seems to be the prevention of complicating superinfections, particularly during systemic immunosuppression. Thus, most authors of PG case series use antibiotics in addition to anti-inflammatory drugs.
Adhering to the current recommendations in the literature regarding surgery for PG lesions and the case report of Long et al. , we were able to perform disease-unrelated surgery free of complications in our described patient. The retrospective study of Xia et al.  reported only a 15.1% occurrence of PG lesions after surgical procedures; thus, the PG-free course of our arthroplasty surgery cannot be attributed solely to our treatment regimen.
Due to the rarity of this constellation, recommendations based on high-level evidence cannot be made for orthopedic trauma treatment during active PG. In our patient’s case, the use of pre- and perioperative steroids, combined with perioperative antibiotics and a soft tissue protective surgical technique, provided a successful strategy for the treatment of this rare challenge in orthopedic trauma surgery. This case report should encourage other surgeons facing this challenge not to avoid necessary operative treatment and improve patients’ quality of life by adhering to the described principles.
All authors participated in the acquisition and analysis of the data as well as the preparation of the manuscript. All authors read and approved the final manuscript.
There was no funding for the case report.
Ethics approval and consent to participate
This case report was approved by the local ethics committee of the Medical University of Vienna (no. 1445/2014).
Consent for publication
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
The authors declare that they have no competing interests.
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