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A case of pyoderma gangrenosum (PG) in a 14-year-old boy with acute myelogenous leukemia (AML) is described. The onset of pyoderma gangrenosum coincided with the relapse of AML. The lesions responded dramatically to treatment with oral prednisone despite the persistence of leukemia. Pyoderma gangrenosum should be included in the differential diagnosis of any nodular, pustular, or necrotic cutaneous eruption in children with leukemia.
Sixty-three patients with pyoderma gangrenosum were seen and studied at the Mayo Clinic from 1971 to 1980. Biopsies from the erythematous border or necrotic edge of the pyoderma gangrenosum lesions usually demonstrated a characteristic pathogenic morphologic evolution. The early lesions revealed mild to moderate perivascular lymphocytic infiltrate associated with endothelial swelling. The fully developed lesions demonstrated necrosis in addition to a dense lymphocytic infiltration surrounding as well as involving the blood vessels. Extravasation of erythrocytes and thrombosis sometimes were seen. Ulceration, infarction, and abscess formation were found in the later stages of evolution. Direct immunofluorescence results were positive in the blood vessels of 36 of 65 (55%) specimens. IgM, C3, and fibrin were found in the papillary and reticular dermal vessels. IgG and IgA were only occasionally present. Pyoderma gangrenosum appears to be a reactive process that is manifested as a vasculitis. Biopsy material from the advancing active erythematous border has early characteristic dermatopathologic findings of lymphocytic vasculitis. Cutaneous vascular immune deposits suggest an immune pathogenesis of either an immune complex disease or lymphocytotoxic reaction.
Prospective studies of streptococcal pyoderma were carried out among black children enrolled in Project Headstart centers in Holmes County, Miss. Sera collected from 28 of these children in early October were tested for opsonic antibodies to one of two prevalent skin strains of group A streptococci isolated from them on one or more occasions over the preceding 3 months. The two streptococcal strains (A and B) belong to M-types previously unrecognized. Ten subjects (36%) had antibody to their homologous serotypes detectable by the indirect bactericidal test: this included 6 of 10 subjects infected with strain B but only 4 of 18 infected with strain A (P < 0.05). Of 17 children who had strains A or B isolated from skin lesions only, 12% developed type-specific antibodies (TSA) against the infecting serotype. In contrast, 11 subjects had these strains isolated from throat cultures (either with or without associated pyoderma), and 72% had detectable TSA (P < 0.01). There was no demonstrable relationship between the development of antibodies to streptococcal extracellular products or to non-type-specific cellular antigens and the development of TSA. These results demonstrate that type-specific immune responses do occur following infection with pyoderma streptococci. The frequency with which such antibodies develop is variable and appears related to a number of factors, including the immunologic properties of the infecting strain and the site of bacterial colonization. Pharyngeal carriage may represent an important mechanism for development of acquired immunity to skin strains of group A streptococci.
Pyoderma gangrenosum is a rare occurrence in patients with haematological malignancy. This characteristic but nonspecific inflammatory process with skin destruction occurred in 4 patients with myelodysplasia, in one with acute leukaemic transformation of myelofibrosis, and in de novo acute myeloblastic leukaemia in another. Clinically, the cutaneous lesion in these patients differed from that associated with inflammatory bowel disease, arthritis, or the idiopathic type of pyoderma gangrenosum by having the vesiculo-bullous borders. Histopathological differences were also evident since more superficial layers of the skin were involved in the ulceration than typically encountered in patients with non-malignant systemic disease. Despite the less penetrating nature of this variant, treatment of the pyoderma gangrenosum is unsatisfactory and in the absence of effective therapy for the underlying disease, healing occurred only in the patient with acute leukaemia who achieved complete remission in response to chemotherapy.
Pyoderma gangrenosum is a clinical condition often associated with internal disorders and immunologic abnormalities. The diagnosis is deduced solely from clinical examination because no histologic or laboratory changes are pathognomonic. The lesions are quite painful and can be particularly disabling. Laryngeal involvement should elicit concerns for protection of the airway. This case demonstrated involvement of the oral cavity, nose, and larynx--as well as other skin sites of the body--with pyoderma gangrenosum. To our knowledge, this is the first reported case of intranasal and laryngeal pyoderma gangrenosum. Early treatment of any associated internal disorders and aggressive treatment with corticosteroids usually result in adequate control of this problem.
A 61 year old patient suffered from pyoderma gangraenosum combined with IgG-paraproteinemia and plasmocytoma. In recent years several papers mentioned the existence of paraproteinemia in patients with pyoderma gangraenosum. Until now only in two cases the association with plasmocytoma were reported. This is the third case of pyoderma gangraenosum combined with plasmocytoma.
Pyoderma gangrenosum is an uncommon skin disorder characterised by deep ulcers surrounded by a violaceous over-hanging edge. Although in many instances there is no clear association with any underlying disease, pyoderma gangrenosum has been described in ulcerative colitis, Crohn's disease, polyarthritis, diabetes mellitus and myeloma. Pyoderma gangrenosum may also be seen as a rare manifestation of myeloproliferative disease including leukaemia. In children, as in our case, it may be the presenting feature.
A 67-year-old woman suffered from an ovarian carcinoma with lymph nodes metastasis. During 3 years, she was treated with alkylating agents (Melphalan). At the end of therapy, no recurrence was observed. Two years later, she developed concomitantly pyoderma gangrenosum and acute myelomonocytic leukaemia. Death occurred rapidly. The association between pyoderma gangrenosum and acute leukaemia is discussed in the light of 16 cases previously reported in the literature. In this case, an induction of leukaemia by cytostatic drugs seems likely. The authors conclude that pyoderma gangrenosum may be considered as a cutaneous signs of acute leukaemia.
A 52-year-old woman who had had ulcerative colitis for more than ten years experienced a third relapse of pyoderma gangrenosum of her lower extremities. She was treated with topical disodium cromoglycate. The dosage of salicylazosulfapyridine and steroids she was taking, and by which the ulcerative colitis, but not the pyoderma gangrenosum could be controlled, was not increased. Because pyoderma gangrenosum is often very difficult to treat and may require an aggressive approach using drugs that can cause serious side effects, we believe that a topical therapy is worth trying.
Although topical antibiotics have been considered less than effective agents in the treatment of "impetigo," recent experience suggests that topical therapy has a place as primary treatment of pyoderma and impetigo. This bacteriologically controlled, randomized study compared the safety and efficacy of mupirocin with oral erythromycin in the treatment of pyoderma and impetigo. A total of 29 mupirocin-treated and 30 erythromycin-treated patients completed the study. None of the mupirocin-treated patients reported adverse experiences compared with 4 erythromycin-treated patients who reported 6 adverse experiences. The mupirocin-treated group had a significantly higher benefit:risk ratio than the erythromycin-treated group as measured by the investigator's global evaluation (P = 0.01). Both treatments eradicated 100% of the two most common pathogens, Staphylococcus aureus and Streptococcus pyogenes. Results from this study demonstrate that mupirocin is as effective as systemic erythromycin ethylsuccinate for treatment of pyoderma and impetigo.
Pyoderma gangrenosum is an uncommon skin lesion often associated with autoimmune diseases. A clear association between leukemia and pyoderma gangrenosum in adults has been established. Two cases of pyoderma gangrenosum in children with leukemia in whom it may be an initial finding are presented.
A 36-yr-old patient suffering from chronic ulcerative colitis complicated by severe pyoderma gangrenosum of the leg is presented. Pyoderma gangrenosum rapidly progressed and failed to respond to a combined treatment of bed rest, prednisone, sulfasalazine, povidone-iodine, compresses, and debridement. Pyoderma gangrenosum responded and disappeared with no recurrence during the following months after the combined treatment of methylprednisolone, dapsone, and zinc sulfate.
The remote results of combined therapy of 300 patients with chronic pyoderma were estimated by the data of the 5-year catamneses. The patients were treated with the use of erycycline, a combined drug consisting of two antibiotics made in the USSR. Stable clinical recovery within 5 years was stated in 78.8 per cent of the patients. Significant improvement (single pustules 2-3 years after discontinuation of the treatment) was registered in 13.7 per cent of the patients. Improvement (insignificant relapses within the first 2 years) was stated in 6.3 per cent of the patients. No effect of the combined therapy was stated in 1.2 per cent of the patients. It was shown that the use of erycycline in the combined therapy of the patients with chronic pyoderma provided its high efficacy, favourable immediate and remote results, decreasing the treatment period at the average by 3.8 days and decreasing the number of relapses. It also provided economy. The analysis of the catamneses promoted critical and objective estimation of the treatment results and stability of the therapeutic remission thus facilitating solution of the problem of treatment and prophylaxis of chronic pyoderma.
Since Sweet's initial description of eight patients, the concept of acute febrile neutrophilic dermatosis has evolved and changed. We have expanded it to include patients with bullous pyoderma gangrenosum, bowel-bypass syndrome with or without the bypass, the vesiculopustular eruption or ulcerative colitis, and possibly even typical pyoderma gangrenosum. A variant of acute febrile neutrophilic dermatosis in which acute myeloid leukemia is present has been reported and seems identical to bullous pyoderma gangrenosum. Although no clear pathogenesis has been demonstrated, studies of the bowel-bypass syndrome have implicated immune complex disease. Therapy with prednisone is usually effective, but numerous other anti-inflammatory agents have been used effectively.
Two patients with pyoderma gangrenosum have responded remarkably well to treatment with Clofazimine (Lamprène). The first patient, a 68-year old women suffered from pyoderma gangrenosum of the buttock and left leg and on the incision scar for cancer of the breast. Laboratory findings showed monoclonal dysglobulinemia (alpha 2-kappa 2). A daily dose of 300 mg of Clofazimine resulted in complete healing with ten days. The second patient was a 24-year old women suffering from ulcerative colitis and a rapidly progressing pyoderma gangrenosum of the left leg. The lesions was completely healed after two weeks of Clofazimine therapy. The dosage was 200 mg daily and was increased to 400 mg daily. Our cases showed decreased cellular immunity and their phagocytic activity was variable.
Pyoderma gangrenosum is an ulcerative skin disorder with typical clinical characteristics. Histologic and laboratory findings are nonspecific. Pyoderma gangrenosum is associated with internal disorders including inflammatory bowel disease, paraproteinemias, leukemias, and arthritis. The pathogenesis of pyoderma gangrenosum is unknown, although a partial defect of cell-mediated immunity may exist. Treatment includes bedrest, local care, sulfonamides, sulfones, and corticosteroids.
Pyoderma gangrenosum has been associated with myelogenous leukemia and plasma cell dyscrasia. When associated with leukemia, pyoderma gangrenosum often has a distinctive clinical presentation with an advancing bullous margin. The pathogenesis of this disorder is unknown, although defective immune mechanisms may be operative. The occurrence of pyoderma gangrenosum and agnogenic myeloid metaplasia in the same patient has now been reported sufficiently to make it a recognized association.
A gammaAchi--paraproteinemia was found in a 73 year-old female patient with pyoderma gangraenosum. Since the paraporteins hitherto observed in pyoderma gangraenosum predominantly belong to the IgA class, a relationship may exist between pyoderma gangraenosum and this type of paraproteinemia.