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PubMed2005

[Neutrophilic dermatosis associated with propylthiouracil-induced p-ANCA (p-antineutrophil cytoplasmic antibodies)].

INTRODUCTION: We report on a patient who progressively developed polymorphic expressions of neutrophilic dermatosis (Sneddon-Wilkinson subcorneal pustulosis and pyoderma gangrenosum) associated with p-antineutrophil cytoplasmic antibodies (p-ANCA), while receiving propylthiouracil for hyperthyroidism. To our knowledge, such associations have never been published so far. CASE-REPORT: A 40 year-old woman was treated with propylthiouracil for Graves'disease. After 16 months of therapy, she noted flares of pustular lesions surrounded with erythematous halo mainly localized on the trunk. The lesions became chronic, and were not improved by potent topical corticosteroids. When first seen in our department in February 2003, the eruption was typical of Sneddon-Wilkinson subcorneal pustulosis. This diagnosis was confirmed by the histological examination of a skin biopsy of a pustule. One month later, she developed an inflammatory progressively ulcerative lesion on the right ankle, typical of pyoderma gangrenosum. The diagnosis was confirmed by the histological examination of a skin biopsy taken on the evolving border of the lesion and showed polynuclear neutrophilic infiltration without vasculitis. Direct immunofluorescence was negative. The presence of serum anti-myeloperoxydase p-ANCA was known for this patient since October 2002. No IgA monoclonal gammapathy was revealed on extensive biological check-up. Systemic oral corticosteroid therapy (1 mg/kg/day) dramatically improved skin lesions with complete healing within 8 weeks. DISCUSSION: Propylthiouracil is well known to induce the occurrence of ANCA in 20 to 64p. 100 of patients treated for Graves'disease. The mechanisms involved are badly recognized so far. Cutaneous vasculitis, glomerulonephritis and polychondritis may be clinically associated with those antibodies. Rare observations of neutrophilic dermatosis, mostly Sweet's syndrome, have been described in patients with propylthiouracil-induced ANCA. One case-report described a 44 year-old woman who developed pyoderma gangrenosum associated with propylthiouracil-induced p-ANCA. These manifestations usually appear within 2 years, as our patient. The data in the literature, allows us to report the polymorphic expressions of neutrophilic dermatosis in this patient with p-ANCA which could be related to propylthiouracil. Such association of Sneddon-Wilkinson subcorneal pustulosis and pyoderma gangrenosum with p-ANCA has never been described in this endocrinologic context so far. Furthermore we propose that neutrophilic dermatosis should be inscribed in the list of side effects induced by propylthiouracil therapy.

Adrenal Cortex Hormones↗
PubMed1994

[Pyoderma gangraenosum: differential diagnosis in ulcus cruris and postoperative exacerbating processes].

A case of pyoderma gangrenosum that was initially misdiagnosed as postthrombotic ulcers is described. Surgical debridement triggered postoperative exacerbation. Dermatologic examination was then initiated and the diagnosis pyoderma gangrenosum was established. No underlying illness was detectable. Immunosuppressive therapy stopped disease activity promptly and as previously reported low-dose cyclosporine was also effective in our exceptionally therapy resistant case. Pyoderma gangrenosum should be considered as a rare, but important differential diagnosis of leg ulcers and postoperative progressive exacerbation. Timely diagnosis based on clinical suspicion is pertinent for correct therapy and to avoid iatrogenic deterioration of the disease.

Diagnosis, Differential↗
PubMed2000

[Presentation of pyoderma gangraenosum in a dermatologic atlas of the early 19th century].

Pyoderma gangrenosum was first described in 1930 by Brunsting, Goeckermann and O'Leary. Nevertheless we found some illustrations in an atlas on dermatology, published by Marie-Nicolas Devergie in the first half of the 19th century, which appear to be pyoderma gangrenosum. In addition to discussions of typical syphilitic affections of the skin, Devergie's "Clinique de la Maladie Syphilitique" includes illustrations of gangrenous ulcers, which appeared unexpectedly after local and systemic therapy with mercury. Devergie interpreted those enlarging ulcers as a side effect of mercury therapy. Thus we were able to find evidence of pyoderma gangrenosum more than 100 years before first description in 1930. The etiology of this clinical picture is still unsettled. The favorite postulate has been a bacterial genesis which was the subject of numerous publications until the 1960s.

Atlases as Topic↗
PubMed2001

Pyodera gangrenosum after aortic valve replacement.

A 68-year-old male patient developed pyoderma gangrenosum after elective aortic valve replacement. Treatment with steroids and clofazimine was initiated, which resulted in rapid cessation of the necrotic process. Due to thoracic instability, stabilization of the sternum was performed followed by surgical debridement and atraumatic wound closure. Postoperatively, immunosuppression with cyclosporine A was added; the patient recovered completely. Pyoderma gangrenosum is a potential life-threatening complication that must be considered in patients with severely impaired wound healing after cardiac operation.

Aged↗
PubMed2004

The effect of infliximab on extraintestinal manifestations of Crohn's disease.

OBJECTIVE: The purpose of this open pilot study was to assess prospectively the effect of infliximab on extraintestinal manifestations in patients with active Crohn's disease refractory to conventional treatment. PATIENTS AND METHODS: Twenty-two consecutive patients with Crohn's disease and one with ulcerative colitis presenting at least one of the known extraintestinal manifestations of Crohn's disease participated in the study. All the patients had Crohn's disease activity index (CAI) scores above 2. Each patient received an intravenous infusion of infliximab at a dosage of 5 mg/kg. A thorough questionnaire was used reviewing the extraintestinal manifestations of Crohn's disease such as erythema nodosum, pyoderma gangrenosum, eye lesions, arthritis or arthralgia, sacroiliitis or inflammatory back pain, hepatic disease, hematologic manifestations (megaloblastic, iron deficiency or hemolytic anemia, thrombocytosis), thrombosis, and nephrolithiasis. Musculoskeletal complaints were evaluated using the parameters intensity of pain, duration of morning stiffness (in minutes), presence of inflammatory back pain, Schober's test of the lumbar region, chest expansion, and distance from occiput to wall. The clinical assessment was performed on the day of the infusion and 2 weeks later. RESULTS: Eleven out of 23 patients had arthralgia of inflammatory nature, three others had evidence of active synovitis on physical examination, and 11 reported inflammatory back pain. Four patients suffered from protracted pyoderma gangrenosum; three had resistant aphthous stomatitis. Eleven patients had more than one extraintestinal manifestation. All four with pyoderma gangrenosum demonstrated significant improvement of their ulcers after one course of infliximab, with complete resolution of the skin lesions in three of them after repeated infusions of infliximab. Aphthous stomatitis completely responded in all patients after a single infusion. Seven out of 11 patients with arthralgia and seven out of 11 with inflammatory back pain/sacroiliitis experienced benefit after treatment with infliximab and reported at least partial clinical improvement in duration of morning stiffness, tender joint count, and visual analogue scale for pain. Only one of three patients with frank arthritis demonstrated clear improvement, and two others failed to respond to infliximab treatment. CONCLUSION: These preliminary results are encouraging and suggest a promising role of infliximab in the treatment of extraintestinal symptoms of Crohn's disease.

Adult↗
PubMed1997

A distinctive vesiculopustular eruption associated with hepatobiliary disease.

BACKGROUND: Cutaneous manifestations of hepatobiliary disease may be a clue to the clinical diagnosis. We encountered a form of vesiculopustular eruption in five patients with hepatobiliary disease. A similar eruption has been described in the setting of ulcerative colitis, and has received the designation of vesiculopustular pyoderma gangrenosum. METHODS: Skin biopsy specimens were examined by conventional histopathologic methods and, in three cases, fresh tissue was available for direct immunofluorescence examination. A diagnosis of hepatobiliary disease was known in three of the patients and in the other two it was established by the skin biopsies. RESULTS: All skin biopsies of the five patients showed a neutrophilic dermatosis that had two morphologies. One was characterised by superficial neutrophilic dermolysis with subepidermal bulla formation and the other by a nonulcerating destructive suppurative folliculitis and perifollicular neutrophilic dermolysis. Both were frequently accompanied by a neutrophilic and lymphotic eccrine hidradermitis and a Sweet's syndrome-like vascular reaction. The lesions were held to represent a novel vesiculopustular eruption with features reminiscent of pyoderma gangrenosum. CONCLUSIONS: We propose the term vesiculopustular eruption of hepatobiliary disease for this distinctive clinicopathologic entity, which bears a strong histologic resemblance to pyoderma gangrenosum. Patients with a known hepatopathy who present with necrotizing vesicles and pustules may have such an eruption. The onset may be the first clue to or herald an exacerbation of underlying liver disease.

Adult↗
PubMed1990

Refractory parastomal ulcers: a multidisciplinary approach.

Chronic parastomal ulcers in patients with ileostomy or colostomy stomas are unusual. Previous reports have implicated infections, fistulas, recurrent inflammatory bowel disease (IBD), pyoderma gangrenosum, and trauma. Over the past 8 years we have evaluated 10 cases of such refractory parastomal ulcers that occurred at a mean of 11 years after stomal surgery. Eight patients had had an ileostomy for IBD while two had undergone colostomy for colon cancer. Five patients with IBD were diagnosed as having pyoderma gangrenosum ulcerations. They required systemic treatment for a mean of 25 weeks to effect ulcer healing. The other five patients had either parastomal ulcers on the basis of dermatoses (contact dermatitis, eczema, or bullous pemphigoid) or contact ulcers due to face-plate pressure and parastomal dermatitis. These patients received topical treatment with healing of ulcers in a mean of 4 weeks. We conclude that parastomal ulcers occurring in patients without IBD or IBD patients without classic pyoderma gangrenosum require early dermatologic evaluation as they respond relatively quickly to appropriate local therapy.

Chronic Disease↗
PubMedDate not supplied

Combination of massive mucinosis, dermatomyositis, pyoderma gangrenosum-like ulcer, bullae and fatal intestinal vasculopathy in a young female.

Cutaneous mucinosis secondary to autoimmune collagen vascular disease is well recognized, but manifestation as cellulitis-like massive cutaneous mucinosis preceding dermatomyositis is unusual. Here we report a 21-year-old Taiwanese woman with a large, rapid onset, painful erythematous, edematous plaque, which histopathologically revealed septal panniculitis with fat necrosis and massive mucin deposition. Incapacitated muscle weakness of proximal extremities, generalized edema, heliotrope erythema, and Gottron's papules developed in a short period of time with high titers of serum muscle enzyme. Serological titers of ANA, anti-dsDNA, anti-ENA panels, and erythrocyte sedimentation rate, however, all showed unremarkable results. Diagnosis of dermatomyositis was confirmed by electromyographic findings of myopathy. As the disease progressed, large, deep cutaneous ulceration and vesiculobullous lesions also developed. In spite of aggressive treatment, the patient died 9 months after the disease onset, probably due to the complication of gastrointestinal ischemia and perforation.

Adult↗
PubMed1999

[Extra-intestinal manifestations and complications in chronic active ulcerative colitis in childhood and adolescence].

HISTORY AND CLINICAL FINDINGS: A 18-year-old girl suffered from chronic active ulcerative colitis for ten years with growth retardation, primary amenorrhoea and osteopenia, so that total colectomy had been discussed as a possible treatment option. However, clinical remission was reached using a medication with budesonide (9 mg), mesalazine (3 g) and azathioprine (100 mg) when a painful ulcer occurred at the right lower leg. INVESTIGATIONS: The clinical examination revealed an ulcer of 7 cm diameter with a red-bluish margin at the right lower leg. The typical clinical features led to the diagnosis of pyoderma gangrenosum. TREATMENT AND COURSE: High doses of orally administered prednisolone induced the healing of the pyoderma gangrenosum within 2 months. After another 2 months systemic steroids were discontinued. Further laboratory tests confirmed secondary adrenal and ovarian failure (negative CRH-GnRH-test). Change of drug therapy to hydrocortisone (25 mg) in combination with mesalazine (3 g) and azathioprine (100 mg) improved the clinical course. Hydrocortisone was gradually reduced until CRH-GnRH-response returned to normal. CONCLUSION: Drug therapy with azathioprine can be regarded as a treatment option in pediatric patients with chronic active inflammatory bowel disease. It can be concluded from this case report that faced with controversial therapeutic options in adolescent patients the decision mainly depends on the clinical experience of the physician than on the results of controlled clinical trials.

Adolescent↗
PubMed1993

[Cyclosporin A therapy in pyoderma gangraenosum].

A 33-year-old woman with pyoderma gangrenosum was treated with cyclosporine A. Cyclosporine A is a valuable drug for the treatment of recalcitrant pyoderma gangrenosum. Its mechanism of action is discussed. The numerous side-effects require close monitoring of cyclosporine A blood levels and several other parameters.

Adult↗
PubMed2003

Superficial granulomatous pyoderma treated with intravenous immunoglobulin.

Superficial granulomatous pyoderma is a rare variant of pyoderma gangrenosum. The superficial ulceration and vegetative margins are clinically distinctive; the suppurative and granulomatous histology is characteristic, though not pathognomonic. The condition is said to be relatively benign and more responsive to treatment than classic pyoderma gangrenosum, though published evidence of this is not always convincing. We present a case of superficial granulomatous pyoderma that was unusually aggressive, but that underwent dramatic and lasting resolution after intravenous immunoglobulin therapy.

Aged↗
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↗
PubMed1992

[Cyclosporin A in the therapy of inflammatory dermatoses].

The experience reported in the literature with cyclosporin A (CyA) in the treatment of various inflammatory and autoimmune dermatological diseases is reviewed and compared with the authors' own experience of treating 36 patients presenting with psoriatic arthritis [8], generalized pustular psoriasis [2], palmoplantar pustular psoriasis [12], Behçet's disease [2], disseminated circumscribed scleroderma [2], acrodermatitis continua suppurativa [2], pemphigus vulgaris [1], lupus erythematosus [3], pyoderma gangrenosum [1], severe atopic eczema [2], and actinic reticuloid [1]. On the basis of the authors' own experience and the reported results, treatment with CyA appears to be primarily indicated in pyoderma gangrenosum, circumscribed scleroderma, psoriatic arthritis and acrodermatitis continua suppurativa. In diseases such as actinic reticuloid, Behçet's disease, localized and generalized pustular psoriasis, treatment with CyA leads to good results with an acceptable risk-benefit ratio. In our view, it is doubtful whether treatment with CyA alone is indicated in alopecia areata, lichen ruber, dermatomyositis, atopic eczema, systemic scleroderma, bullous diseases, and lupus erythematosus, and in the last two it should be given only in combination with systemic steroids. Literature reports provide no support for the use of CyA in ichthyosis vulgaris, pityriasis rubra pilaris, and cutaneous T-cell lymphomas. The risk-benefit ratio of CyA treatment should be carefully considered, especially in diseases that are not life-threatening.

Behcet Syndrome↗
PubMed1994

Leukocyte colony-stimulating factors. A review of associated neutrophilic dermatoses and vasculitides.

BACKGROUND: Hematopoietic colony-stimulating factors are a diverse group of cytokines now commercially available through recombinant technology. The colony-stimulating factors have proven utility in a wide spectrum of cytopenic states, and their use is now common-place. Unlike many cytokines, the colony-stimulating factors are usually well tolerated. We present a dramatic case of pyoderma gangrenosum arising during granulocyte colony-stimulating factor therapy. Additionally, the relevant literature regarding cutaneous complications of colony-stimulating factor therapy is summarized. OBSERVATIONS: Including our case, two reported patients developed pyoderma gangrenosum while under colony-stimulating factor therapy. Sweet's syndrome presented in an additional three patients, and necrotizing vasculitis was precipitated in three others. The eruptions consistently developed after 1 to 2 weeks of colony-stimulating factor therapy and were apparently unrelated to the underlying systemic illness. Although most reactions developed de novo, three were superimposed on a preexisting inflammatory process. CONCLUSIONS: Serious cutaneous adverse effects of colony-stimulating factors are distinctly rare but include neutrophilic dermatoses and necrotizing vasculitis. Upregulation of neutrophilic function and secondary release of cytokines may induce these complications. Early recognition and cessation of colony-stimulating factor therapy will limit the morbidity of these adverse events.

Adult↗
PubMed1983

Vesiculopustular eruption of ulcerative colitis.

Many cutaneous disorders that have been described in association with ulcerative colitis (UC), including certain pustular eruptions, probably represent early, evolving lesions of pyoderma gangrenosum (PG). Several nonspecific, often poorly delineated pustular eruptions apparently unrelated to pyoderma gangrenosum have also been reported to occur with ulcerative colitis. A patient had an evanescent, vesiculopustular eruption with a course paralleling that of his UC. Histologic examination of biopsy material from the skin revealed intraepidermal and subcorneal neutrophilic abscess formation and a mixed dermal, perivascular inflammatory cell infiltrate with notable sparing of the follicles. An IgG deposition in a bandlike pattern was identified at the dermoepidermal junction by direct immunofluorescence microscopy. This vesiculopustular eruption may represent a distinct entity or another case or variant of similar pustular eruptions previously described in association with UC.

Adult↗
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↗