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PubMedDate not supplied

Chronic recurrent multifocal osteomyelitis preceding pyoderma gangrenosum and occult ulcerative colitis in a pediatric patient.

Chronic recurrent multifocal osteomyelitis (CRMO) is a clinicopathologic entity of unknown origin mainly affecting children and young adults. It shares many clinical features with pyoderma gangrenosum (PG), an uncommon ulcerating inflammatory disorder of the dermis and underlying subcutaneous fat. Both conditions are chronic and relapsing, and have been associated with inflammatory bowel disease (IBD). The diagnoses are made by exclusion. Histology is nonspecific and cultures are negative. There has been only one previous report of both conditions occurring in the same patient. Here we describe the second case, a 12-year-old girl who developed pyoderma gangrenosum in direct continuity to an underlying CRMO lesion of the right anterior tibia. Occult ulcerative colitis (UC) was subsequently discovered.

Child↗
PubMed2004

Pouchitis-associated pyoderma gangrenosum following restorative proctocolectomy for ulcerative colitis.

A 43-year-old female with ulcerative colitis treated by proctocolectomy and ileal pouch--anal anastomosis developed acute pouchitis. Although no extra-intestinal manifestations were present before the surgical procedure, pyoderma gangrenosum developed concomitantly with the appearance of acute pouchitis. Both conditions completely resolved with oral metronidazole only. This is the first report of acute pouchitis-related pyoderma gangrenosum. This association suggests that pouchitis may represent a recurrent ulcerative colitis-like syndrome within the novel environment of the pouch.

Acute Disease↗
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↗
PubMed2001

Sporotrichosis masquerading as pyoderma gangrenosum: case report and review of 19 cases of sporotrichosis.

We present the case of a 59-year-old woman who had large ulcerations on her right leg that were diagnosed initially as pyoderma gangrenosum and treated with three immunosuppressive agents (cyclosporin, prednisone and azathioprine) for 6 months. Results of a biopsy at 6 months showed numerous cigar-shaped bodies consistent with Sporothrix schenckii; identification was confirmed by tissue culture. A retrospective review was performed for all cases diagnosed as sporotrichosis from tissue culture or biopsy specimens at the Mayo Clinic. Nineteen cases were identified. The present case was the only one in which fungal organisms were visible on histological examination. The present case emphasizes the importance of making a definitive histological diagnosis in unusual ulcer cases or in suspected cases of pyoderma gangrenosum before the initiation of immunosuppressive therapy. The large number of cigar-shaped bodies in the tissue is a rare finding in sporothrix infection and has been reported in only two cases previously.

Biopsy, Needle↗
PubMed1995

Cutaneous ulceration resembling pyoderma gangrenosum in the primary antiphospholipid syndrome: a report of two additional cases and review of the literature.

The antiphospholipid antibody syndrome has been associated with various cutaneous manifestations, the most common of which may be leg ulceration. Cutaneous ulcers with clinical features suggestive of pyoderma gangrenosum have been reported in this setting only rarely. We report here two additional cases of the association between antiphospholipid antibody syndrome and pyoderma gangrenosum-like skin lesions. These patients suffered from the primary antiphospholipid syndrome. We suggest that clinicians assay for antiphospholipid antibodies when a clinical diagnosis of pyoderma gangrenosum is entertained.

Adult↗
PubMed1981

Idiopathic pyoderma gangrenosum and impaired lymphocyte function: failure of azathioprine and corticosteroid therapy.

A 58-year-old man with chronic "idiopathic' pyoderma gangrenosum, recurrent secondary staphylococcal infection, nail candidiasis, markedly impaired lymphocyte function and a serum blocking factor is described. Despite an initially favourable response to immunosuppressive therapy, the patient later relapsed whilst on high dose systemic corticosteroids and azathioprine. Immune function remains abnormal 3 years cessation of azathioprine. Attempted immunostimulation with Corynebacterium parvum proved unsuccessful. A striking but transient increase in lymphocyte function followed parenteral iron therapy for an unexplained iron-deficiency anaemia. Gradual reduction in prednisone dosage to 5 mg daily has coincided with a period of clinical resolution and modest improvement in lymphocyte reactivity. Prolonged immunosuppressive therapy may be inadvisable in chronic idiopathic pyoderma gangrenosum associated with defective cellular immunity.

Azathioprine↗
PubMed1976

Pyoderma gangrenosum associated with active chronic hepatitis: report of two cases.

In two patients, active chronic hepatitis was complicated by the development of pyoderma gangrenosum. The favorable response of the pyodermatous lesions to azathioprine therapy suggests that this drug may be of value in treating this disorder in patients for whom corticosteroid therapy produces no benefit or is contraindicated because of side effects. Azathioprine, too, is a potentially toxic drug and may need to be discontinued. Possible causal relationships between pyoderma gangrenosum and active chronic hepatitis are discussed.

Adult↗
PubMed1989

Pyoderma gangrenosum in an allogeneic bone marrow transplant recipient.

We present the case of a 15-year-old boy who developed facial pyoderma gangrenosum following an allogeneic bone marrow transplantation for the treatment of a 'blast' crisis developing in the course of chronic myelogenous leukaemia. The lesion appeared 7 months before any evidence of relapse. The discussion is focused on both the presentation of pyoderma gangrenosum associated with myelo-proliferative disorders and its pathogenesis via the underlying immunosuppression.

Adolescent↗
PubMed1980

Splenic infarction and tissue accumulation of crystals associated with the use of clofazimine (Lamprene; B663) in the treatment of pyoderma gangrenosum.

A male patient 68 years, suffering from pyoderma gangrenosum which was resistant to conventional treatment, received clofazimine 400 mg daily for 5 months, then reducing to 300 mg daily for the next 6 months. Eleven months after starting the drug, he was admitted to hospital with severe abdominal pain, laparotomy revealing infarction of the spleen, with violaceous congestion of the small bowel. The spleen was removed and post-operative recovery was satisfactory. Histopathological examination of the spleen (removed at operation) and of tissue from a duodenal biopsy (taken postoperatively) showed large numbers of striations and outlines suggestive of crystal deposition. Mesenteric lymph node revealed a massive accumulation of crystals in cortical and medullary sinuses. The findings emphasize that clofazamine should not be used in high dosage over prolonged periods of time, except under close clinical and laboratory supervision, and for conditions not amenable to other drugs.

Aged↗
PubMed1997

Bilateral peripheral ulcerative keratitis associated with pyoderma gangrenosum.

PURPOSE: A 37-year-old Hispanic man with a history of chronic myelogenous leukemia was first seen with fever and preseptal cellulitis of this right orbit after a tooth extraction. METHODS: The patient subsequently developed bilateral, severe peripheral ulcerative keratitis. He was treated with systemic antibiotics for a presumed underlying infectious cause. Several painful, necrotizing skin lesions developed over his face, trunk, and extremities. RESULTS: Repeated skin biopsies of the necrotizing lesions were consistent with pyoderma gangrenosum. Both the skin lesions and peripheral ulcerative keratitis responded dramatically to systemic prednisone. CONCLUSIONS: Pyoderma gangrenosum should be included in the differential diagnosis of peripheral ulcerative keratitis.

Adult↗
PubMedDate not supplied

Pyoderma gangrenosum associated with the secondary antiphospholipid syndrome.

A 64-year-old woman with an 11-year history of systemic lupus erythematosus and amputation of her left lower leg as a consequence of arterial embolism, presented with two large, non-healing ulcers on her right shank. Pyoderma gangrenosum associated with secondary antiphospholipid syndrome was diagnosed based upon the typical clinical features and increased antibodies to cardiolipin. Although an aggressive therapy with corticosteroids and cyclosporine was started, her condition continued to worsen. She finally died as a result of sepsis. We discuss the difficulties in diagnosis and therapy of SLE combined with the antiphospholipid syndrome and pyoderma gangrenosum.

Adrenal Cortex Hormones↗
PubMed1989

[Pyoderma gangrenosum and IgA gammopathy. Association with atrophic gastritis].

Pyoderma gangrenosum (PG) is a rare condition remarkable for its association with particular diseases, notably haemopathies and gastrointestinal diseases. As regards haemopathies, the associations most frequently encountered are with myeloid malignancies and monoclonal dysglobulinaemia. The association of PG with mainly inflammatory digestive tract diseases is also classical. The lack of publications concerning gastric atrophy and the dual haematological and gastric pathology which characterizes our case have prompted us to report it. A 60-year-old woman without significant history was admitted for PG on both knees, following vesiculo-bullous lesions. Laboratory examinations detected a normochromic anaemia tending to be macrocytic, a marked inflammatory syndrome and a monoclonal lambda light chain IgA peak at protein immunoelectrophoresis. Bone marrow biopsy, skeletal radiography and a search for Bence-Jones proteinuria were normal or negative. Colonoscopy showed no abnormality, but fibroscopy of the upper digestive tract revealed a severe gastric atrophy en plaques. Serum vitamin B12 level was moderately low, but there was no other sign of pernicious anaemia. After one month treatment with systemic corticosteroids, healing was obtained under replacement vitamin therapy. PG recurred a few months later; serum vitamin B12 level was normal, and the lesions healed after systemic corticosteroid treatment. In non-myelomatous dysglobulinaemia IgA is frequently found and there is no light chain predominance. PG often precedes dysglobulinaemia. Evolution towards a true myeloma seems to be exceptional. In a recent publication, 17 cases of association between PG and myeloma were mentioned, the IgA type being most common. Protein electrophoresis is indispensable in patients with PG. Five cases of congenital hypogammaglobulinaemia have been recorded, including three with IgA deficiency.(ABSTRACT TRUNCATED AT 250 WORDS)

Agammaglobulinemia↗
PubMed1979

Herpes simplex virus isolation from pyoderma gangrenosum lesions in a patient with chronic lymphatic leukemia.

Herpes simplex virus type 2 was isolated and identified from the vesicular border of pyoderma gangrenosum lesions on the genital region of a patient with chronic lymphatic leukemia. Dramatic relief of pain as well as quick disappearance of the vesicular margin of the lesions and of the inflammatory halo around them occurred as a result of local treatment with a solution of zinc acetate. A careful search for a viral agent should be done in every case of pyoderma gangrenosum occurring in a patient with a hematological malignancy or/and impaired immunity, especially if the lesions are on the face or in the genital region.

Acetates↗
PubMed1995

Pyoderma gangrenosum with systemic involvement.

We report a case of a patient with pyoderma gangrenosum associated with multisystemic manifestations. The patient showed liver dysfunction, respiratory failure, and aseptic meningeal reaction. Corticosteroid therapy was efficient in treating both the skin lesions and the other systemic disorders.

Adult↗
PubMed1992

[Peristomal pyoderma gangrenosum after colectomy for Crohn disease. Successful medical treatment].

A case of Pyoderma gangrenosum with two different abdominal sites in a female patient suffering from colonic Crohn's disease is presented. Local trauma on the midline scar of an incisional hernia and around the stoma were the possible triggering factors. Despite the major abdominal wall defect and an infected parietal collection, steroid therapy was very effective without the need for surgery.

Aged↗
PubMedDate not supplied

Anti-tumor necrosis factor alpha monoclonal antibody (infliximab) for the treatment of Pyoderma gangrenosum associated with Crohn's disease.

Here we report a case of Pyoderma gangrenosum (PG) associated with Crohn's disease successfully treated with infliximab. The efficacy of this drug in many inflammatory diseases has already been reported, but its use in PG has only been seen in very few cases. Our study confirms that this therapy is a valid alternative solution for treating PG, which is often unresponsive to conventional therapies.

Antibodies, Monoclonal↗
PubMed2003

Pyoderma gangrenosum: a review.

Since its first description in 1930, the pathogenesis of pyoderma gangrenosum (PG) has remained obscure even as an ever-widening array of systemic diseases has been described in association with it. The histopathologic distinction of PG from other ulcerative processes with dermal neutrophilia is challenging and at times impossible. In consequence, when confronted with a biopsy from such a lesion, the pathologist has an obligation to obtain a full and detailed clinical history. In short, as a diagnosis of PG does not hinge exclusively upon the biopsy findings in isolation from other studies, a solid knowledge of the clinical features, the systemic disease associations and the differential diagnosis will help the pathologist to avoid diagnostic pitfalls or the generation of a report which is non-contributory to patient care. In this review, we describe in detail the different clinicopathologic forms of PG, summarize the diseases associated with this process in the literature and in our experience, and briefly review the treatment options.

Adult↗
PubMed2004

Acute pustulosis of the legs in diverticulitis with sigmoid stenosis: an overlap between bowel-associated dermatosis-arthritis syndrome and pustular pyoderma gangrenosum.

BACKGROUND: Bowel-associated dermatosis-arthritis syndrome denotes the occurrence of diarrhoea with arthritis and skin lesions related to bowel disease with or without bowel bypass. In this condition, the histological finding of cutaneous aseptic neutrophilic cell infiltrate is non-specific and common to a wide spectrum of neutrophilic dermatoses, including pyoderma gangrenosum. OBSERVATION: We describe a 78-year-old woman with fever, abdominal discomfort and arthralgias, who developed grouped pustular lesions on her shins with histologically spongiform pustule formation. Aetiological assessment disclosed diverticular disease with sigmoid stenosis. CONCLUSION: Although clinical and histological features in our case fit the diagnosis of bowel-associated dermatosis-arthritis syndrome, they may also correspond to a pustular variant of pyoderma gangrenosum. Our observation raises the question of the nosological classification of bowel-associated dermatosis-arthritis syndrome within the spectrum of neutrophilic diseases.

Acute Disease↗