Pyoderma gangrenosum: a challenging complication of bilateral mastopexy

Abstract

A case of pyoderma gangrenosum progressively developing after bilateral mastopexy at the surgical site is described. The described case was successfully treated with corticosteroids, the application of the dermal regeneration template Integra^® and autologous skin grafts. This approach was able to save the patient's life and to generate a high‐quality aesthetical outcome. The article reported the case, reviewed the literature of pyoderma gangrenosum related to mastopexy or augmentation mammoplasty and discussed the use of a dermal regeneration template to optimise aesthetical results after reconstructive surgery.

Introduction

Pyoderma gangrenosum presents a diagnostic challenge during the postoperative phase because of the similarities with postoperative wound infections and its rarity of appearance (1). Therapeutically, rapid and aggressive intensive care interventions seem to have tissue and lifesaving effects. We report a case of extensive pyoderma gangrenosum after bilateral mastopexy. The clinical course as well as the unique therapeutic approach are discussed.

Case report

A 41‐year‐old Caucasian female patient, with unremarkable previous medical history, was transferred to our hospital with ulcerative wounds involving both postoperative mastopexy sites for hyperbaric oxygen therapy for, as then perceived, a rapidly progressive postoperative infection with tissue loss. Two weeks before arrival, our patient underwent a successful bilateral mastopexy. Three days postoperatively, the surgical wound sites became red and painful with rapidly progressive tissue breakdown, perilesional oedema and inflammation, as well as foul discharge and purple‐blue margins (Figure 1). The patient presented with high fever, chills, anorexia and vomiting. At that time, the patient was treated with broad‐spectrum antibiotics, without any positive effect on the progression. The wounds were then surgically explored and cleansed. The computerised tomographic scan and magnetic resonance imaging taken after arrival at our hospital revealed air collections at the left pectoralis muscle, fluid in the pleural space, oedema of the pectoralis muscles and retrosternal fluid accumulation (Figure 2). Bilateral pleural drainage was installed. Because of respiratory insufficiency, the patient was intubated and mechanically ventilated. Although no obvious bacterial infection could be demonstrated on bacterial culture, the patient was transferred to our hospital for hyperbaric oxygen therapy for mixed soft‐tissue infection. Multiple bacterial cultures in the following days remained sterile. A skin biopsy demonstrated a neutrophilic inflammation of an oedematous dermis with folliculitis without proven bacterial infection on Gram and Ziehl–Neelsen staining and without mycosis on Grocott staining and no signs of vasculitis on immunofluorescence. Because of the rapid evolution, the negative bacterial cultures, the atypical pathological findings and the typical clinical appearance of relatively indolent ulcers with extensive necrosis around the edges of the lesions, pyoderma gangrenosum was kept as diagnosis (Figures 3a,b). At that moment, laboratory results showed C‐reactive protein values with a maximum of 41·9 mg/dl (normal value < 0·5  mg/dl), a leucocytosis level of 32·3 × 10⁹/l (normal value 4–10 × 10⁹/l) with absolute neutrophilia, a fibrinogen level of 558 mg/dl (normal value 200–400 mg/dl) and a d‐Dimer concentration of 3114 ng/ml (normal value 0–500 ng/ml). With pyoderma gangrenosum in mind, the patient received high doses of methylprednisolone (40 mg three times a day intravenously), fluconazole (400 mg/day intravenously) and piperacillin/tazobactam (4 × 4 g/day intravenously). On a daily basis, the wound was carefully managed using saline as wound cleanser, an alginate (Algisite^®; Smith & Nephew NV, Brussels, Belgium), Melolin^® (Smith & Nephew NV) as secondary dressing and Cavilon^® Spray (3M, Diegem, Belgium), a no‐sting barrier film to protect the wound edges from maceration. Although no infection was diagnosed, hyperbaric oxygen therapy (2·8 ATA for 60 minutes) was provided daily. To prevent the development of osteoporosis, the patient received calciferol. Although the patient recovered significantly using this treatment plan, with rapid decrease in inflammatory parameters, the wound surface was too large for wound healing by secondary intention. The multidisciplinary team faced a problem because even the smallest surgical stress could induce reoccurrence. The team decided to use Integra^® (Johnson & Johnson Medical, Skipton, UK), a dermal regeneration template that act as a bilayer matrix that provides a scaffold for dermal regeneration (Figure 4). The corticosteroids were tapered over 2 months.

Figure 1.

Early presentation of pyoderma gangrenosum (5 days postoperatively).

Figure 2.

Early computerised tomographic scan of the thoracic region showing widespread skin necrosis in the region of the left breast, diffuse swelling of the pectoralis muscles with two small air collections as well as mediastinal oedema.

Figure 3.

(a) Pathological investigation of biopsy revealing ulcerated skin with a central nidus of necrotising acute follicular inflammation and formation of abscesses. Absence of primary vasculitis. (b) Dramatic evolution of pyoderma gangrenosum (14 days postoperatively).

Figure 4.

Application of Integra^®.

Accessory investigations for comorbidities of pyoderma gangrenosum showed no signs for inflammatory bowel disease, arthritis, monoclonal gammopathy, human immunodeficiency virus infection, malaria, liver disease, malignancy and other serologic disturbances except for an increased Ca‐125. Gynaecological follow‐up demonstrated no abnormal findings. Twelve months thereafter, a complete healing is present with almost a perfect restoration of the aesthetical appearance (Figure 5).

Figure 5.

Complete healing (12 months postoperatively).

Discussion

Pyoderma gangrenosum was first described by Brunsting and coworkers in 1930 (2). Although originally thought to be a bacterial disease, it is probably not infectious in nature. Of the four main clinical variants (ulcerative, pustular, bullous and vegetative), the ulcerative form is by far the most prevalent (1, 3, 4). Up to 50% of pyoderma gangrenosum patients have associated diseases, but it seems that if the cause or association is not found in the first 6 months, it is unlikely to be found later. Consequently, an important step during clinical assessment is the search for comorbidities associated with pyoderma gangrenosum (5). In our patient, no comorbidities were found.

Treatment of pyoderma gangrenosum incorporates both local and systemic drugs to control the inflammatory process. The most consistent results are reported with systemic corticoids and cyclosporin, and these effective, but toxic, modalities can be employed when the severity of the disease justifies the risks. Systemic corticoids are preferred to gain rapid control of the inflammatory state and the fast spreading and tissue‐destructing disease. Most cases respond to doses of 1–2 mg/kg/day prednisolone given orally. Pulse corticoid treatment (1 g of methylprednisolone once daily, for 3–5 days) may generate a quicker response. The adverse effects of steroid therapy should be recognised and treated. The response to cyclosporin (5–10 mg/kg/day) seems to be fairly consistent. Drugs like dapsone (after having excluded glucose‐6‐phosphate dehydrogenase and methemoglobin reductase deficiency), tacrolimus, thalidomide, azathioprine, methotrexate and others have been used successfully.

Since Winter and, later Hinman, good clinical practice consists of moist wound healing techniques (6, 7). Moreover, the right balance towards bacterial bioburden and inflammatory state should be found in order for the wound to walk through the different phases of healing. In the prediagnostic phase of such a case, when there is still doubt about wound infection, local and systemic antibacterial drugs seem the safest approach. In a certain perspective, hyperbaric oxygen therapy might be adjustable during this phase or even later 8, 9, 10, 11, 12). As fast as the diagnosis is accepted, following the concepts of good clinical wound care, dressings should be used that keep the wound at least moist, warm and free of infection. In our case, a calcium alginate dressing was used because of its moisture retention and haemostatic features Even the smallest expression of local trauma can induce a recurrence. If surgery is necessary or requested for reconstruction, the patient should be started on systemic steroids the week before surgery. An alternative plan would be to use a lower dose of steroids (0·5 mg/kg/day) and supplement this with a steroid sparing dose of sulfapyradene (500 mg up to 1 g/three times a day). Alternatively, dapsone at 100 mg/day can be provided.

An extensive literature search reveals at least 12 cases of pyoderma gangrenosum after mastopexy or augmentation mammoplasty as summarised in Table 1. As shown, a wide diversity of treatment options was used resulting in a variable residual scarring. ‘Classical’ comorbidities for pyoderma gangrenosum were only observed in a minority of these cases. The role of tissue‐engineered skin substitutes in burn surgery and in the treatment of chronic wounds is constantly evolving. Integra^®, the collagen‐based, artificial skin, is a safe and effective modality in severely burned patient with a marked decrease in the length of stay 13, 14, 15). This is the first report of a case of pyoderma gangrenosum treated with the dermal analogue Integra^®. We believe that our case demonstrates that using tissue‐engineered products might reduce scar formation in severe cases of pyoderma gangrenosum.

Table 1.

Overview of all reported cases of pyoderma gangrenosum after mastopexy

Flügel et al. (16)	27	Bilateral mammoplasty for fibroadenoma;	Antibiotics, methylprednisolone,	Healing	No
		7 days postoperatively, skin necrosis, fever	latissimus muscle flap, split skin grafts
Gruhl et al. (17)	23	Reduction mammoplasty; 9 days postoperatively,	Povidone iodine, penicillin,	Healing	No
		skin necrosis, no fat necrosis	methylprednisolone
Grau Salvat et al. (18)	27	Reduction mammoplasty;	Gentamycine ointment, oral antibiotics,	Healing with residual scarring	No
		4 days postoperatively, pustulous ulcers, undermined	cyclosporin, methylprednisolone
		borders, high fever, perilesional edema
Gudi et al. (19)	34	Mammoplasty; 2 weeks postoperatively	Topical steroids (clobetasol propionate)	Healing with scarring	Controlled hypothyroidism,
					antinuclear antibody positive
					Antiparietal cell positive
Berry et al. (20)	54	Breast reduction; 4 days postoperatively	Oral and intravenous (IV) antibiotics, cyclosporin	Epithelialisation after 6 weeks	No
Goncalves et al. (21)	41	Reduction mammoplasty;	IV antibiotics, methylprednisolone,	Healing	Thyroidectomy
		7 days postoperatively	Skin grafts		(Similar history
					of the patient's sister)
Lifchez et al. (22)	40	Reduction mammoplasty	Hydrogel, vancomycin, imipenem,	Healing	Hypothyroidism, neurogenic
			fluconazole, hyperbaric oxygen steroids		bladder, depression
			vitamin A, split thickness
			skin grafts
Shofer et al. (23)	24	Reduction mammoplasty (Bostwig);	IV and topical antibiotics/cyclosporin	Healed after 22 weeks	Antinuclear antibody titre 1:320
		5 days postoperatively
MacKenzie et al. (24)	51	Breast reconstruction with latissimus	IV antibiotics, cyclosporin	Completely healed	No
		dorsi muscle flap + prosthesis for
		adenocarcinoma of the left breast;
		4 days postoperatively
Sotillo‐Gago et al. (25)	43	Augmentation mammoplasty with silicone prosthesis;	Topical and oral antibiotics, cyclosporin,	Complete healing with	Ulcerative colitis diagnosed one
		5 days postoperatively, pustulous ulcers, raised edges		residual scarring within 45 days	month later
Clugston et al. (26)	37	Reduction mammoplasty	Intralesional triamcinolone	Complete resolution	Unknown
Van Poucke et al.	41	Augmentation mammoplasty	IV antibiotics, methylprednisolone,	Complete resolution	No
(current study)			hyperbaric oxygen, Integra, skin grafts

Acknowledgements

The authors unanimously declare no commercial interests in products used in this paper.