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PubMed1997

Malignant pyodermas revisited.

The concept of malignant pyoderma (MP) has created controversy since its origin. The distinction of this disease from pyoderma gangrenosum was based on clinical criteria and response to treatment. Herein we discuss our current ideas on this entity and its possible relationship to Wegener's granulomatosis (WG). Follow-up data from the three original cases of MP are reported, as well as additional clinical and laboratory data from cases subsequently thought to represent MP. Many of these cases have similar clinical features such as facial and periauricular ulceration and occasionally signs or symptoms of WG, including positive titers of antineutrophil cytoplasmic antibodies (with a diffuse cytoplasmic staining pattern) (cANCA). MP represents a distinctive clinical disorder and may be a dermal manifestation of WG. Some cases of MP may represent pyoderma gangrenosum or other undefined systemic illnesses. Such cases of WG can be distinguished on the basis of clinical, histopathologic, and laboratory evidence including cANCA titers. MP should no longer be used as a final clinical diagnosis.

Adult↗
PubMed2002

Ocular and extracutaneous involvement in pyoderma gangrenosum.

PURPOSE: To report an unusual presentation of pyoderma gangrenosum with ocular and extracutaneous involvement. DESIGN: A single, interventional case report. METHODS: A 28-year-old female presented with nodular scleritis in the right eye and left orbital inflammation lasting 1 year. The patient initially responded to systemic steroids, but then ocular inflammation became more severe, associated with onset of fever, myalgias, and elevation of the erythrocyte sedimentation rate, accompanied by formation of multiple cutaneous abscesses. MAIN OUTCOME MEASURES: Ocular and systemic disease control. RESULTS: The diagnosis of pyoderma gangrenosum was made 1 year after the occurrence of ocular symptomatology, when the patient presented with ocular and skin lesions and abscesses in the spleen and liver. CONCLUSIONS: Pyoderma gangrenosum is a rare, necrotizing, noninfectious ulcerating skin disease. Ocular and extracutaneous involvement rarely occur, but the disease should be included in the differential diagnosis of scleritis and orbital inflammation.

Abscess↗
PubMed2003

Pyoderma gangrenosum in a patient with collagenous colitis.

The association between pyoderma gangrenosum and inflammatory bowel disease is well established. Collagenous colitis is one of the microscopic colitides, which are thought to be on the same spectrum of disease as Crohn's disease and ulcerative colitis. Before this study there had been no reported cases of pyoderma gangrenosum in the setting of collagenous colitis. We report a case of a 79-year-old woman with symptomatic collagenous colitis and refractory pyoderma gangrenosum.

Administration, Oral↗
PubMed1988

Pyoderma gangrenosum: a review.

Pyoderma gangrenosum is a poorly understood disease characterized by exacerbations and remissions of morphologically unique skin ulcers. It frequently is thought to be the cutaneous manifestation of an underlying systemic disease. In this review of pyoderma gangrenosum, the characteristics of the disease are described and a differential diagnosis is formulated. Associations with systemic diseases are made through a review of the literature. The pathophysiology of pyoderma gangrenosum and probable causes are considered, and special consideration is given to the immunologic mechanisms that may be operative in the disease. Finally the currently available therapeutic alternatives are reviewed.

Colitis, Ulcerative↗
PubMed1988

Cyclosporine therapy for pyoderma gangrenosum associated with sclerosing cholangitis and ulcerative colitis.

Malignant pyoderma of the right ear developed in a 58-year-old woman, and it spread insidiously over the entire right side of the face. Pyoderma gangrenosum was diagnosed on the basis of clinical and histologic findings and the patient's history of ulcerative colitis and sclerosing cholangitis. After successive therapeutic failures with a score of conventional approaches over an 18-month period, oral cyclosporine, 10 mg/kg/day, was started. Significant healing of the skin was noted within 1 month. There was an associated remission of the patient's cholangitis and inflammatory bowel disease. The dosage of cyclosporine was progressively tapered and then discontinued after 7 months, when healing was complete. No relapse occurred in the subsequent 14 months, and there was an associated remission of the patient's cholangitis and inflammatory bowel disease. Cyclosporine merits serious attention for treatment of both pyoderma gangrenosum and sclerosing cholangitis.

Administration, Oral↗
PubMed1996

Pyoderma gangrenosum severely affecting both hands.

We report a case of pyoderma gangrenosum affecting both hands simultaneously. Pyoderma gangrenosum affecting the hands is an extremely rare condition which may result in considerable tissue destruction. Management includes immunosuppressant therapy, treatment of associated medical conditions and minimal surgical intervention. Despite a high maintenance dose of corticosteroid and adequate control of coexisting ulcerative colitis and rheumatoid arthritis, tissue destruction in the hands spread rapidly in our patient. The key to the patient's dramatic improvement was the tissue biopsy suggesting pyoderma gangrenosum and the subsequent treatment with the cytotoxic immunosuppressant, azathioprine.

Amputation, Surgical↗
PubMed1988

A case study of pyoderma gangrenosum.

Pyoderma gangrenosum is a rare neutrophilic dermatosis seen initially as painful pustules or bullae on the skin that rapidly ulcerate and have a characteristic raised, purplish areola surrounding them. It is often associated with inflammatory bowel disease and less commonly with other systemic diseases, such as myelofibrosis, rheumatoid arthritis, and chronic active hepatitis. Pyoderma gangrenosum involving the hand is a rare entity, with only two previous cases reported in the literature. This article describes a patient with myelofibrosis in whom pyoderma gangrenosum of the hand developed after she underwent splenectomy.

Aged↗
PubMed1998

Pyoderma gangrenosum producing saddle nose deformity.

Pyoderma gangrenosum affecting the nose is rare and this may lead to diagnostic confusion because of the large differential diagnosis. As diagnosis is made, largely, on the basis of exclusion the treatment of pyoderma gangrenosum may be unduly delayed. The condition is often disfiguring, particularly following inappropriate surgical intervention, and early diagnosis is therefore important. We present a case of pyoderma gangrenosum managed initially in the community with minor surgery and resulting in the rare complication of saddle nose deformity.

Aged↗
PubMed2000

Interleukin-8 overexpression is present in pyoderma gangrenosum ulcers and leads to ulcer formation in human skin xenografts.

Interleukin-8 (IL-8) is a potent chemotactic polypeptide for neutrophils. However, the role of this cytokine during inflammation remains unclear. Skin specimens from patients with pyoderma gangrenosum demonstrated IL-8 overexpression in skin ulcers, which suggests a role for IL-8 in the development of the disease. We therefore constructed a recombinant adenovirus expressing the complementary deoxyribonucleic acid encoding human IL-8 (IL-8/Ad5) that induces a 2000-fold increase in IL-8 expression of infected human fibroblasts in vitro. Human skin engrafted to severe combined immunodeficiency mice and then injected with the recombinant virus demonstrated erythema, an intense perivascular infiltration of neutrophils, and extravasation of erythrocytes after 8 hours. By 12 hours after injection, neutrophils had accumulated beneath the epidermis, which then necrotized, and one or more ulcers that remained for approximately 2 weeks were observed. Clinically and histologically, the ulcers resembled pyoderma gangrenosum. These clinical and experimental findings suggest an etiologic role of IL-8 in the pathogenesis of pyoderma gangrenosum.

Animals↗
PubMed1999

Pyoderma gangrenosum with secondary pyarthrosis following propylthiouracil.

The association of pyoderma gangrenosum and arthritic symptoms is well documented. We present a rarely reported variant of this in a 44-year-old woman with pyoderma gangrenosum and bilateral large purulent effusions of her knees. She had no evidence of underlying rheumatoid arthritis or a specific seronegative spondyloarthropathy. Of note she had a history of Graves' disease for which she had been treated with propylthiouracil for 3 years and on investigation at this presentation had a markedly elevated perinuclear antineutrophil cytoplasm antibody (P-ANCA) level with specificities for IgM myeloperoxidase, IgG elastase and IgG lactoferrin. We believe this patient had pyoderma gangrenosum with secondary sterile pyarthrosis and a P-ANCA precipitated by propylthiouracil.

Adult↗
PubMed2001

Pyoderma gangrenosum of the hand: a case series and review of the literature.

Pyoderma gangrenosum is a rapidly developing cutaneous ulcer that is rare in the hand and may mimic a fulminating infection. The correct treatment is nonsurgical; surgery may actually incite a pathergic response worsening the condition. Misdiagnosis results in inappropriate treatment, unnecessary surgery, and even amputations. The records of 7 patients with pyoderma gangrenosum of the hand treated at 2 large academic medical centers between 1992 and 1999 were reviewed. There were 4 men and 3 women with an average age of 58 years (range, 32-81 years). Five patients had bilateral involvement. Minor trauma preceded the appearance of the lesions. An associated systemic disease was found in all patients with ulcerative colitis being the most common (3 patients). All patients initially had a misdiagnosis of infection. Thirteen misdiagnoses (range, 1-3/patient) resulted in 16 unnecessary surgeries (mean, 2.2/patient) including 4 amputations and 2 failed skin grafts. No surgical procedure resulted in clinical improvement. All cultures were negative. Several physicians (mean, 5; range, 3-7) examined each patient before the final diagnosis of pyoderma gangrenosum, which was made by, or in conjunction with, a dermatologist in all cases. The average time to clinical improvement after correct medical treatment was initiated was 5 days.

Adult↗
PubMed2000

Recurrence of pyoderma gangrenosum within a chronic wound following microvascular free-tissue transfer.

The authors present a 29-year-old woman with a chronic foot wound that failed to heal, despite extensive medical and surgical therapy. The diagnosis of pyoderma gangrenosum was ultimately made, and the patient was started on systemic cyclosporine therapy. In the absence of apparent active disease, surgical debridement and microvascular free flap reconstruction were performed to achieve wound closure. Six weeks postoperatively, recurrence of the pyoderma gangrenosum was identified in the free flap, resulting in partial, superficial, flap necrosis. Laboratory evaluation at that time demonstrated subtherapeutic cyclosporine levels. Once the cyclosporine level was increased to the therapeutic range, the wound healed, and the microvascular free flap was salvaged. Because of the relative lack of precision in both the clinical and pathologic determination of acuity level, as well as the tendency toward pathergy, surgical treatment of any form poses many potential risks for these patients. For this reason, surgery should serve only as an adjunct to medical therapy, which remains the mainstay for treatment of pyoderma gangrenosum.

Adult↗
PubMed2000

Cavitating pulmonary infiltrate in an adolescent with pyoderma gangrenosum: a rarely recognized extracutaneous manifestation of a neutrophilic dermatosis.

Neutrophilic dermatoses such as pyoderma gangrenosum are characterized by sterile, neutrophilic cutaneous infiltrates. Extracutaneous neutrophilic infiltrates can occur, primarily in the joints, lungs, heart, central nervous system, gastrointestinal tract, and eyes. Pulmonary disease is the most frequently reported extracutaneous manifestation of pyoderma gangrenosum and is characterized by patchy infiltrates or interstitial pneumonitis. We describe an adolescent with typical pyoderma gangrenosum who presented with cavitary pneumonia and responded completely to oral corticosteroids. In patients with inflammatory ulcers, extracutaneous neutrophilic disease should be considered, once an infectious process has been excluded.

Adolescent↗
PubMed2001

Pyoderma gangrenosum associated with biphenotypic acute leukemia.

Pyoderma gangrenosum is a neutrophilic dermatosis that may be associated with myeloid malignancies. Less information is available about the association of pyoderma gangrenosum with lymphoid malignancies. We present, to our knowledge, the first case of pyoderma gangrenosum associated with biphenotypic acute leukemia wherein the malignant cells show a phenotype specific for myelogenic and lymphocytic leukemia. Histopathologic examination revealed rather nonspecific features without involvement of leukemic cells in the skin lesions. Treatment with systemic steroids was followed by characteristically rapid healing of the skin lesion.

Female↗
PubMed1987

Pyoderma gangrenosum in an elderly woman. Course marked by extensive morbidity.

A case of extensive medical and functional morbidity in an elderly patient during treatment of pyoderma gangrenosum is described. Pyoderma gangrenosum is an uncommon cause of ulcerative skin lesions often involving areas of skin trauma about the lower extremity. Because of the possibility of underlying disease and the special treatment requirements, the condition must be differentiated from other, more common causes of skin ulceration. Treatment includes local care, systemic corticosteroid therapy, and control of any underlying disease. Pyoderma gangrenosum serves as a model for the interactions of various types of morbidity in the healthcare of the elderly.

Administration, Oral↗
PubMed2001

The use of high-dose immunoglobulin in the treatment of pyoderma gangrenosum.

BACKGROUND: Immunosuppressive medications such as corticosteroids and cyclosporin are the most commonly employed therapies in pyoderma gangrenosum. We describe a patient with multiple ulcers of pyoderma gangrenosum on the lower extremities in whom immunosuppressive therapy caused serious side effects and had to be discontinued but who was subsequently treated successfully with high dose intravenous immunoglobulin (IVIG). METHODS: IVIG was given intravenously at a dose of 400 mg/kg per day for 5 consecutive days. After 1 week there was an arrest in the progression of the ulcers and a marked reduction in pain. Two weeks later clinical improvement of the ulcers was observed. Subsequently, IVIG was given at a dose of 1 g/kg per day for 2 consecutive days. RESULTS: The treatment induced a dramatic clinical improvement of one ulcer and healing of the others. Side effects were minimal and well tolerated, and consisted of chills and a slight fever, which resolved with the administration of acetaminophen. CONCLUSION: We feel that IVIG can be used in patients with pyoderma gangrenosum in whom conventional therapies are ineffective or produce serious side effects.

Aged↗
PubMed1983

T helper-suppressor cell imbalance in pyoderma gangrenosum, with relapsing polychondritis and corneal keratolysis.

We found decreased T helper/inducer and increased T suppressor/cytotoxic cells in a 45-year-old woman with pyoderma gangrenosum. Serum immunoglobulin levels were normal, suggesting that these T suppressor cells did not function primarily to regulate antibody synthesis. The patient had diminished cutaneous delayed hypersensitivity responses, reacting to only one of six antigens tested, but responded in vitro to three of three antigens. Because of their various regulatory functions, excess T suppressor cells, or a lack of T helper cells, could be a common factor underlying many of the humoral and cell-mediated immune derangements, as well as the neutrophil abnormalities, that have been found in pyoderma gangrenosum. Our patient also had relapsing polychondritis and corneal keratolysis, consistent with the systemic nature of the disorder. The T-cell imbalance persisted even as the ulcer improved. Since monoclonal antibodies against T cell subpopulations are readily available, measuring these cell types as part of the immunological workup of patients with pyoderma gangrenosum might yield valuable clues concerning the pathogenesis of this disorder.

Corneal Ulcer↗