Cases reported "Pyoderma Gangrenosum"

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11/20. Simultaneous bilateral microvascular tissue transfer for soft-tissue defects of the lower extremities in pyoderma gangrenosum.

    pyoderma gangrenosum is a necrotizing skin disease of unknown etiology. The lesions are painful and occur mostly on the lower extremities, either single or multiple, but can appear in any region of the body. The authors report a case of pyoderma gangrenosum involving bilateral skin lesions over the anterior surface of the legs in a 13-year-old male patient. The lesions presented with ulcerative colitis, which is one of the most commonly associated diseases of pyoderma gangrenosum. After examination, systemic immunosuppressive treatment was administered and the progression of the disease was controlled. The lesions were treated with the simultaneous application of two free anterolateral thigh fasciocutaneous flaps after radical debridement. The procedure was successfully performed and no recurrence was observed. Although microvascular free tissue transfer cannot be performed in a standard fashion in pyoderma gangrenosum cases because of the risk of pathergy response, it should be considered as a surgical option in selected cases, otherwise difficult to manage, with the simultaneous application of appropriate systemic medical treatment.
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12/20. 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.
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13/20. Peculiarities of PAPA syndrome.

    OBJECTIVES: To describe a family from new zealand with the pyogenic arthritis, pyoderma gangrenosum and acne (PAPA) syndrome, an autoinflammatory, variably expressed, erosive destructive form of arthritis. methods: Information was gained through medical records and interviews of the affected patients and wider family. dna sequencing was performed at Seay Center for Musculoskeletal research texas Scottish Rite Hospital for Children. RESULTS: Five patients were affected over three generations with an E250Q mutation found on the CD2BP1 gene on chromosome 15. Common features included a severe, pauciarticular-onset, destructive peripheral arthritis, beginning at ages 5, 5, 2, 3 and 1(1/2) years. This combination marked cervical ankylosis (in two members), micrognathia and a more severe phenotype is unique. A third-generation family member treated early with DMARDs is following a less severe course. The skin involvement was variable, all with degrees of acne from puberty, though only one patient displayed pyoderma gangrenosum. A clear pattern of the arthritis switching off in adolescence and the triggering of skin disease was observed. CONCLUSIONS: Differing degrees of joint destruction, and cervical ankylosis in this family with the E250Q mutation demonstrate PAPA syndrome's variable expression. Further understanding of this rare condition and its pathway may allow better targeting of treatments, not just for families with this specific syndrome but also for other, more common, forms of arthritis.
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14/20. pyoderma gangrenosum treated with high-dose intravenous immunoglobulins: Two cases and review of the literature.

    pyoderma gangrenosum (PG) is a neutrophilic skin disease commonly treated with immunosuppressants. High-dose intravenous immunoglobulins are used to treat a range of inflammatory diseases, but we found only five reports of the use of high-dose intravenous immunoglobulins in the treatment of PG. We report on two patients with PG for whom immunosuppressants could not be prescribed and who were treated with high-dose intravenous immunoglobulins.Case 1 was a 58-year-old man who presented with a 6-year history of PG. He was initially treated with prednisone. The 20 mg/day dosage of prednisone could not be reduced and treatment had to be discontinued after 1 year because of serious adverse effects. minocycline treatment led to improvement but had to be discontinued after 6 years because of facial skin hyperpigmentation. Case 2 was a 66-year-old man who presented with a 3-year history of PG. Different therapeutic procedures for PG (prednisone, topical tacrolimus or betamethasone) had failed. High-dose intravenous immunoglobulins were administered monthly at a dose of 2 g/kg for 6 months. The treatment induced stabilisation of the disease and made it possible to reduce corticosteroid use in both patients. These cases show that high-dose intravenous immunoglobulins represent a therapeutic alternative for PG, but the efficacy of this treatment should be confirmed in further studies.
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15/20. Treatment of pyoderma gangrenosum with cultured keratinocyte autografts.

    BACKGROUND. pyoderma gangrenosum is an uncommon chronic skin disease characterized by rapidly enlarging cutaneous ulcers. immunosuppressive agents, such as corticosteroids, are the mainstay of therapy. OBJECTIVE. We describe a patient with pyoderma gangrenosum treated with cultured keratinocyte autografts for a full-thickness ulcer located on the dorsal and lateral aspects of the foot. methods. After stabilizing the ulcer with intralesional and systemic corticosteroids, the ulcer was debrided and cultured keratinocyte autografts were secured with nylon mesh. An outer dressing of gauze and elastic bandage was used. RESULTS. The patient had > 95% "take" of the grafts and the ulcer was fully healed in less than 1 month. The grafted area preserved the clinical phenotype of the palmar skin from which the original biopsy was taken. CONCLUSION. Cultured keratinocyte autografts can provide permanent wound coverage for patients on high doses of immunosuppressive medications.
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16/20. Peristomal pyoderma gangrenosum and inflammatory bowel disease.

    pyoderma gangrenosum (PG) is a debilitating skin disease most often associated with inflammatory bowel disease and is a reportedly rare cause of peristomal ulceration. The lesions of PG rapidly evolve from small, erythematous pustules to deep, painful, pyogenic ulcers within hours to days of onset. Although the behavior and the appearance of the lesions of peristomal PG are diagnostic, a lack of familiarity with PG often leads to misdiagnosis and inappropriate therapy. This study reports four cases of peristomal PG and discusses the 20 previously reported cases in patients with inflammatory bowel disease. Seventy-five percent of patients were female and 67% had Crohn's disease. All patients had colitis, including all of the patients with Crohn's disease, 82% of whom had additional perineal complications. The diagnosis of peristomal PG was based on clinical appearance alone in 83% of cases. The onset of peristomal PG ranged from 2 weeks to 3 years following ostomy. The response to medical therapy was variable. All cases (17 of 17) treated with high-dose corticosteroids and local wound care responded, but five cases required additional therapy. No patient was successfully treated with stoma revision. risk factors for the development of peristomal PG include Crohn's colitis, female gender, and perineal disease. While most patients respond well to systemic steroids and local wound care, up to one third of patients require long-term medical management.
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17/20. Topical treatment with 1% sodium cromoglycate in pyoderma gangrenosum.

    BACKGROUND: pyoderma gangrenosum is an uncommon skin disease usually treated with systemic drugs, most frequently corticosteroids. Isolated topical treatment is usually considered unsatisfactory. OBJECTIVE: To evaluate the effect of topical 1% sodium cromoglycate solution on pyoderma gangrenosum in 5 patients 25-30 years of age. methods: 5 patients, including 2 under systemic steroid treatment, hospitalized in our department from 1992 to 1993 because of pyoderma gangrenosum, were treated with topical 1% sodium cromoglycate solution. Systemic corticosteroids were further added in 2 patients whose initial improvement was inadequate. RESULTS: Initial improvement was noted in all 5 patients after 3-7 days of sodium cromoglycate treatment. Complete healing of the ulcers occurred within 5-8 weeks. CONCLUSIONS: Topical treatment with sodium cromoglycate can be effective as adjunctive or sole treatment in pyoderma gangrenosum.
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18/20. Familial pyoderma gangrenosum presenting in infancy.

    pyoderma gangrenosum (PG) is a rare, poorly understood skin disease that occurs in all age groups. Less than 0.4% of patients are infants and represent a diagnostic challenge as early lesions may resemble other skin disorders. Here we report for the first time three siblings affected with PG all presenting during infancy. Unlike the older age group, the ulcers spared the legs but involved the buttocks, thighs and perianal area in all the infants. Conclusion: This is the first reported family with PG affecting three siblings suggesting autosomal recessive inheritance. The diagnosis may be more difficult in infants due to absence of underlying associated disorders and the tendency of the lesions to appear in areas where infants frequently have other dermatoses. PG characteristically involves the buttocks, thighs and perianal area and spares the legs.
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19/20. A case of pyoderma gangrenosum stabilized with lymecycline, topical benzoyl peroxide and treated by autograft.

    pyoderma gangrenosum is a chronic inflammatory ulcerative skin disease of unknown etiology, often associated with various systemic disorders such as inflammatory bowel disease, rheumatoid arthritis, chronic active hepatitis, diabetes mellitus and hematologic malignancies. The ulcers are characterized by their undermined violaceous borders. The disease remains a therapeutic challenge. Corticosteroids are the mainstay of therapy; however, side effects from this treatment and recalcitrant pyoderma gangrenosum require therapeutic alternatives. We report the case of a large subacute pyoderma gangrenosum stabilized with lymecycline, topical benzoyl peroxide and successfully treated by an autograft. This observation supports the opinion that the risk of pathergy of a graft can be avoided by the stabilization of the disease.
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20/20. Corneal melting and scleromalacia perforans in a patient with pyoderma gangrenosum and acute myeloid leukemia.

    Postoperative endophthalmitis may present in an atypical fashion (absent or minimal anterior chamber reaction) in the presence of underlying immunosuppressive disorder. The authors describe an apparently healthy 58-year-old man who displayed endophthalmitis with minimal anterior chamber reaction following penetrating keratoplasty for granular corneal dystrophy with underlying acute myeloid leukemia. Scleromalacia perforans in association with pyoderma gangrenosum subsequently developed, leading to ciliary staphyloma and corneal melting. pyoderma gangrenosum is an uncommon, idiopathic skin disease that may also have ocular manifestations.
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