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PubMed2006

The relationship between neutrophilic dermatosis of the dorsal hands and sweet syndrome: report of 9 cases and comparison to atypical pyoderma gangrenosum.

BACKGROUND: Neutrophilic dermatoses are a collection of diseases with varying presentation unified by clinical and histologic features. Neutrophilic dermatosis of the dorsal hands is a recently described clinical entity and an evolving disease concept. Its relationship to acute febrile neutrophilic dermatosis (Sweet syndrome), pyoderma gangrenosum, and a primary vasculitis has been debated. OBSERVATIONS: We present 9 cases (8 women and 1 man) of neutrophilic dermatosis of the dorsal hands, all with consistent histologic features. Two cases had histologic evidence of vasculitis, and 3 had clinical extension of lesions onto the forearms. Most showed fever, leukocytosis, and/or elevated erythrocyte sedimentation rate. Individual cases were associated with leukemia, lung carcinoma, and inflammatory bowel disease. All 9 patients responded to systemic corticosteroid therapy, with additional response to dapsone, methotrexate, and potassium iodide therapies in several cases. Of the 9 patients, 5 showed complete resolution of their skin disease, whereas 4 required ongoing therapy. We assessed the 43 cases previously reported in the literature. CONCLUSION: The clinical presentation, laboratory data, histologic features, and response to corticosteroid therapy offer strong evidence that neutrophilic dermatosis of the dorsal hands is a localized variant of Sweet syndrome and is also identical to atypical pyoderma gangrenosum when that condition presents on the hands.

Adult↗
PubMed2000

Twenty cases of peristomal pyoderma gangrenosum: diagnostic implications and management.

HYPOTHESIS: Our experience with peristomal ulcers suggested that peristomal pyoderma gangrenosum (PPG) is an infrequent and usually unrecognized complication of inflammatory bowel disease. We hypothesized that a review of our experience with PPG would clarify the essentials of its diagnosis, evaluation, and treatment. DESIGN: A case series of 20 consecutive patients with PPG complicating inflammatory bowel disease were treated at our institution between 1986 and 1999. There were 15 women and 5 men. At the time of development of peristomal pyoderma, 10 of 20 patients had a diagnosis of Crohn disease (CD), while 9 had a diagnosis of ulcerative colitis (UC). One patient was diagnosed as having CD only after first developing PPG. MAIN OUTCOME MEASURE: Healing of PPG. INTERVENTIONS: All patients had failed local enterostomal care prior to referral. Debridements and/or stomal revisions were uniformly unsuccessful. Biopsies, when performed, did not provide clinically important information. Treatment was directed toward inflammatory bowel disease, with variable clinical responses to corticosteroids, metronidazole, cyclosporine, sulfasalazine, and infliximab. RESULTS: Ultimately, 13 patients had a diagnosis of CD. Of these patients, 12 (92%) of 13 developed PPG coincident with recurrent disease. Two patients had a remote history of proctocolectomy for UC and subsequent evaluation revealed CD. One patient developed PPG adjacent to a urinary Kock pouch after cystectomy; ultimately, a diagnosis of CD was made. No patients were lost to follow-up, but in 1 case of UC, no evaluation for latent CD was carried out. The final diagnosis was CD disease in 13 (65%) of 20 and UC in 7 (35%) of 20 patients. All PPG ulcers healed completely, within an average of 11.4 months (median, 8 months; range, 1-41 months). Ulcer resolution was achieved with medical therapy alone in 14 (70%) of 20 cases. Resection of active gastrointestinal CD resulted in healing in 5 (83%) of 6 cases. One case healed 2 months after conservative therapy only. CONCLUSIONS: This review of the largest reported series of PPG suggests the following: (1) PPG complicating inflammatory bowel disease is uncommon and often misdiagnosed by clinicians; (2) local wound care measures have little role in the healing of PPG; (3) PPG usually heralds active CD; (4) in patients with prior history of UC, PPG indicates CD until proven otherwise; (5) prolonged medical therapy (11 months), usually with immunosupression, is required for healing of PPG; and (6) if feasible, surgical resection of all active CD leads to the healing of PPG ulcers.

Adult↗
PubMed2000

Clinical features and treatment of peristomal pyoderma gangrenosum.

CONTEXT: Peristomal pyoderma gangrenosum (PPG), an unusual variant of pyoderma gangrenosum, has been reported almost exclusively in patients with inflammatory bowel disease (IBD) and is frequently misdiagnosed. OBJECTIVE: To better characterize the clinical manifestations, diagnosis, and management of PPG. DESIGN, SETTING, AND PATIENTS: Retrospective analysis of 7 patients with PPG observed in a university-affiliated community setting between 1988 and December 1999. MAIN OUTCOME MEASURES: Clinical and histopathologic features, associated disorders, and microbiologic findings. RESULTS: Two patients had Crohn disease, 2 had ulcerative colitis, and 3 had abdominal cancer. Five patients had at least 1 relapse of PPG after initial healing. Although 3 of 4 patients with IBD had active bowel disease, a parallel course with PPG occurred in only 1 patient. Both patients whose stoma was relocated developed an ulcer at the new site. Effective therapies included topical superpotent corticosteroids; intralesional injection of triamcinolone acetonide at the ulcer margin; topical cromolyn sodium; oral dapsone, prednisone, cyclosporine, mycophenolate mofetil; and intravenous infliximab. CONCLUSION: Our experiences demonstrate that although PPG has been most often reported in patients with IBD, it may occur in the absence of IBD. Biopsy of the skin lesion is not diagnostic but excludes other causes. Relocation of the stoma may be associated with a new ulceration and should be avoided. Trauma to the skin of a predisposed patient may elicit the pustules or ulcerations associated with pathergy. JAMA. 2000;284:1546-1548.

Adult↗
PubMed1984

Treatment of pyoderma gangrenosum with disodium cromoglycate.

The case of a 35-year-old black woman with a 10-year history of ulcerative colitis and a 3-year history of pyoderma gangrenosum is presented. The pyoderma gangrenosum was treated successfully with topical application of disodium cromoglycate.

Administration, Topical↗
PubMed2001

[Pyoderma gangrenosum: a rare cause of unexpected wound healing delay].

Pyoderma gangrenosum represents a rare but severe disturbance of wound healing following operative interventions. A case report of pyoderma gangrenosum after breast surgery is presented and the aetiology and the various options regarding investigations and treatment are discussed considering the current literature.

Anti-Inflammatory Agents↗
PubMed1996

[Successful topical administration of cyclosporin A in pyoderma gangraenosum].

Therapy of pyoderma gangrenosum can be complicated by side effects of the various systemic therapies, including corticosteroids, azathioprin and cyclosporin A. In this paper we report on a 49-year-old patient presenting with recalcitrant pyoderma gangrenosum who was treated successfully by topical application of cyclosporin A. Cyclosporin A serum levels were always in the subtherapeutic range, and no side effects were observed.

Administration, Topical↗
PubMed1999

[Therapy-resistant pyoderma gangrenosum--treatment with mycophenolate mofetil and cyclosporine A].

A 68-year old woman presented with ulcerations on the calves that had occurred spontaneously. The very painful lesions both clinically and histologically showed the characteristics of pyoderma gangrenosum. During hospitalization she was treated with corticosteroids (oral, i.v., topically), clofazimine, cyclophosphamide, intravenous immune globulin, cyclosporine (oral, local), dapsone, thalidomide and sodium cromoglycate (topically) without any benefit. Finally, when treated with mycophenolate mofetil (CellCept) (oral) and cyclosporine (oral), her skin lesions showed continuous improvement. The topical application of thrombocytic growth factors (cytokines) probably accelerated the granulation. Eight weeks after initiating this treatment the lesions could be covered with split thickness skin grafts. Our observation suggests that mycophenolate mofetil, a novel immunosuppressive agent which has thus far been used almost exclusively in transplantation medicine, may be an effective therapeutic modality in combination with cyclosporine A for the treatment of pyoderma gangrenosum.

Aged↗
PubMed2002

[CD8-positive, CD30-negative cutaneous T-cell lymphoma simulating pyoderma gangrenosum].

Ulcerated primary cutaneous lymphomas are not rare, but the clinical manifestation as a pyoderma gangrenosum look-alike is extraordinary. CD8-positive lymphomas are rare, unclassifiable tumours with variable prognosis. We report on a 49-year-old patient with a large ulcerated primary cutaneous lymphoma on the left chest wall presenting as pyoderma gangrenosum. With immunohistochemical staining, most lymphocytes were shown to be CD8-positive. The CD30 antigen was not expressed. After radiotherapy with complete skin irradiation, the lymphoma regressed completely. The patient has been free of relapse for 28 months so far.

CD8 Antigens↗
PubMed2002

[Pyoderma gangraenosum. Case report and comparison with necrotizing fasciitis].

The differential diagnosis of patients with ulcerative trachelophyma repeatedly causes difficulties. Particularly in view of the possibly fatal outcome, early differentiation between two clinical pictures is very important. Necrotizing fasciitis is often misdiagnosed or the diagnosis is delayed with a mortality rate of approximately 25-40%. It is characterized by local disintegration of the subcutaneous fascia and extensive gangrene of the skin. Sometimes it is not even possible to control the disease using combined antibiotics. Early surgical exploration is mandatory to stop progression of the disease. Pyoderma gangrenosum develops following an initial lesion with pustules and bullae in an ulceration with slight bleeding. Therapy consists of i.v. glucocorticoids (60-200 mg prednisolone/day) administered as early as possible. In complete contrast to the therapy for necrotizing fasciitis, surgical intervention should be strictly avoided in cases of pyoderma gangrenosum.

Adult↗
PubMed2002

Sensorineural hearing loss as an extra-intestinal manifestation of ulcerative colitis in an adolescent girl with pyoderma gangrenosum.

UNLABELLED: Inflammatory bowel disease is becoming increasingly frequent in children of all ages. In addition to the usual gastrointestinal stigmata of weight loss, anaemia, and rectal bleeding, children may exhibit prominent extra-intestinal manifestations such as joint symptoms, skin signs and some other auto-immune manifestations. Here we present a 15-year-old girl with ulcerative colitis in whom pyoderma gangrenosum and acute sensorineural hearing loss developed. CONCLUSION: Although pyoderma gangrenosum is well described with inflammatory bowel disease, sensorineural hearing loss a is very unusual finding, possibly of auto-immune aetiology. We recommend steroid or immunosuppressive therapy in such a patient.

Adolescent↗
PubMed1976

Selective immunoglobulin M (IgM) deficiency in two immunodeficient adults with recurrent staphylococcal pyoderma.

Two adult men with recurrent pyoderma due to Staphylococcus aureus and a selective deficiency of immunoglobulin M (IgM) antibody synthesis are described. An analysis of each patient's polymorphonuclear leukocyte chemotaxis, phagocytosis and killing of Staph. aureus, serum opsonizaiton of Staph. aureus, and serum and lymphocyte-mediated responses to antigenic stimulation was performed. Family studies revealed a possible autosomal dominant inheritance pattern with heterogenetic expression of various dysgammaglobulinemic states in each patient's first degree relatives. In vivo studies of delayed hypersensitivity and in vitro studies of polymorphonuclear leukocyte and lymphocyte function were normal. A defect in IgM, but not in IgG (immunoglobulin G), antibody synthesis to a number of antigens, and a mild decrease in serum opsonic activity to Staph. aureus correctable by heat inactivated normal human serum were found in each patient. In these patients, the recurrent staphulococcal pyoderma prompted an investigation of host defense mechanisms and revealed low to absent IgM levels and a defect in IgM antibody synthesis.

Adult↗
PubMed1995

Tissue immunoglobulin G subclasses observed in immune-mediated dermatopathy, deep pyoderma and hypersensitivity dermatitis in dogs.

A panel of monoclonal antibodies has been used to define three of the four subclasses of canine immunoglobulin G (IgG2, IgG3 and IgG4) in formalin-fixed tissues. These reagents, together with a polyclonal antiserum specific for the Fc region of canine IgG, were used in an immunohistochemical study of biopsies of skin from five normal dogs and from the lesions of canine pemphigus foliaceus in seven dogs, discoid lupus erythematosus in eight dogs, bullous pemphigoid in one dog, cutaneous drug eruption in two dogs, deep pyoderma in 13 dogs and hypersensitivity dermatitis in eight dogs. IgG autoantibody was identified in the skin of all the dogs with immune-mediated dermatoses by using the polyclonal reagent, and antibody of the IgG2 and/or IgG4 subclass was identified in the epidermis of three of the dogs with pemphigus foliaceus, two of those with discoid lupus erythematosus and in the dog with bullous pemphigoid. In all the lesions, the infiltrate of dermal plasma cells consisted of similar numbers of IgG2 and IgG4 bearing cells, with relatively few IgG3 positive cells. The total number of these cells was generally approximately the same as or greater than the number of cells labelled with the polyclonal reagent. There was no significant difference between the IgG-bearing plasma cell infiltrate in German shepherd dogs with deep pyoderma and that in dogs of other breeds. The infiltration of IgG2 and IgG4 bearing plasma cells into the skin of dogs with a range of cutaneous disorders was related to the selectively enhanced serum levels of these subclasses in the diseased dogs.

Animals↗
PubMed1991

Pyoderma gangrenosum associated with systemic lupus erythematosus: response to pulse steroid therapy.

Pyoderma gangrenosum in a 35-year-old woman with long-standing systemic lupus erythematosus was treated with pulse steroid therapy. Significant improvement in the ulceration and control of the lupus erythematosus occurred. The unusual association of these disorders is reviewed, and the efficacy of pulse therapy in the treatment of refractory cases of pyoderma gangrenosum is discussed.

Adult↗
PubMed1992

Pyoderma gangrenosum, polycythemia rubra vera, and the development of leukemia.

A patient with long-standing, well-controlled polycythemia rubra vera developed recurrent episodes of bullous pyoderma gangrenosum followed by the transformation of his hematologic disease into a rapidly progressive acute myeloid leukemia. This case, together with previously described patients, indicates that the appearance of bullous pyoderma gangrenosum in a patient with polycythemia rubra vera is often of ominous prognostic significance.

Humans↗
PubMed1992

Pyoderma gangrenosum-like ulcer in a patient with large granular lymphocytic leukemia.

Large granular lymphocytic leukemia refers to a clonal expansion of lymphocytes that have abundant cytoplasm and azurophilic granules. The disease is characterized clinically by chronic neutropenia and it may be associated with recurrent pyogenic infections. Except for these infections, cutaneous manifestations of this disease have not been well characterized. We describe a patient with large granular lymphocytic leukemia, which was confirmed by molecular genetics studies, who had a pyoderma gangrenosum-like ulcer on his leg. Results of an evaluation of the histologic characteristics and the leukocytic immunophenotype of a skin biopsy specimen from the ulcer demonstrated large granular lymphocytes within the blood vessels. Cutaneous ulceration may be a manifestation of large granular lymphocytic leukemia, and this disease should be considered when diagnosing patients with otherwise unexplained pyoderma gangrenosum-like ulcers of the skin.

Complement C3↗
PubMed1995

Pyoderma gangrenosum in an HIV-infected patient.

Pyoderma gangrenosum has previously been reported in four patients with HIV infection. We describe here the case of a fifth HIV-infected patient with pyoderma gangrenosum and review the characteristics of the four previously reported cases.

Adult↗