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A case of pyoderma vegetans (PYV) and hidradenitis suppurativa (HS) is described. Our patient had a previous history of perifolliculitis capitis abscedens et suffodiens and acne conglobata. Direct immunofluorescence findings ruled out pemphigus vegetans and suggested a potential pathogenic mechanism.
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.
Two patients with refractory pyoderma gangrenosum (PG) were treated with oral Tripterygium wilfordii multiglycoside (TWG). TWG is a Chinese medicine extracted from a medicinal herb, Tripterygium wilfordii Hook F, and has potent anti-inflammatory and immunosuppressive effects. The effect of TWG on PG was demonstrated by clinical findings. Improvement of the lesions occurred within two weeks, and the ulcers healed about a month. Mild side effects such as gastrointestinal disturbances were observed in both patients. These side effects were patient-acceptable, and there was no need to stop the treatment. Transient elevation of serum ALT was observed in one patient; the serum ALT returned completely to normal after the discontinuation of TWG. These results suggest that TWG may be an effective alternative for refractory PG and that careful monitoring of liver function during TWG treatment is necessary.
We present here a case of pyoderma gangrenosum (PG) on the stump of an amputated leg. The patient was a 69-year-old woman who had both of her legs amputated due to acute arterial occlusion. An ulcer first appeared nine years later, after which point it continued to fluctuate in size. Complications included regional blood flow disorder at the amputated stump, diabetes, and secondary infection. Despite various therapies, the ulcer exacerbated, and hypoproteinemia, increased CRP, and fever were confirmed. The patient was diagnosed as having PG based on her clinical symptoms and because the ulcer did not respond to various therapies. The ulcer improved significantly in response to administration of 40 mg/day of prednisolone, and complete epithelialization was later achieved. Given the presence of multiple complications, it was extremely difficult to confirm PG. Therefore, it is important for physicians to consider PG as one of the causes of intractable ulcers.
We report a rare case of squamous cell carcinoma developing from fistules of chronic perianal pyoderma in a 49-year-old Japanese man. He first noticed an abscess and nodule on his buttocks and perianal area 21 year previously (at the age of 28); the fistules formed later. These fistules were surgically removed, and an artificial anus was constructed 14 years ago (at the age of 35) in our hospital, when a histopathological examination revealed no malignant changes. However, he was recently admitted to our hospital with arterial bleeding from the ulcer of the buttock. On admission, the histological diagnosis of the ulcer was well differentiated squamous cell carcinoma. Wide local excision of the ulcer and scar tissue, including the sacrum, was performed. The defect was covered with a left latissimus dorsi flap and skin graft. He received radiation therapy after the operation. However, he died of cachexia and pneumonia. This case indicated that the CPP would better have been treated with wide excision before the development of SCC. Therefore, we recommend careful follow-up of patients affected by CPP and repeated biopsies of the lesion, particularly when the condition is severe, longstanding, and extensive. We discussed the term "CPP" and reviewed 22 cases of SCC arising in CPP reported in the Japanese literature.
Pyoderma gangrenosum developed in a man with a five-year history of paroxysmal nocturnal hemoglobinuria and monoclonal gammopathy. He had multiple walnut sized ulcers on his back and extremities, plasma IgM-k type M-protein and low erythrocytic CD55 expression. This is an extremely rare association. However, clonal expansion of plasma cells and chimeric expression of hematopoietic cell glycosylphosphatidylinositol (GPI)-anchored proteins may represent somatic mutations of hematopoietic stem cells in PG as well as PNH. PNH is based on abnormalities in the GPI-anchor formation on various hematopoietic and non-hematopoietic cells. Since the GPI-anchored proteins have pleiotropic functions in complement mediated cell lysis, leukocyte motility, and coagulation systems, the present case may indicate the possible involvement of a GPI-anchored protein abnormality in the pathogenesis of PG.
We present here a case of chronic perianal pyoderma (CPP), who was treated with a novel skin graft. The patient was a 28-year-old male who had suffered from painful abscesses and nodules on his buttocks for over 10 years. Although the abscesses and nodules were initially restricted to one buttock, they gradually spread over both buttocks. He visited our hospital in July of 2000. He was clinically diagnosed as CPP and treated with oral doses of antibiotics. When admitted to our hospital in September of 2000, an anal fistula was also found. The operations for CPP and anal fistula were simultaneously performed. After the anal fistula treatment, the lesions of CPP were totally resected. In this procedure, we removed the epidermis and upper dermis from the excised CPP lesions and grafted them on the defects of the excised lesion. There has been no recurrence of the CPP. This operation procedure, which we call the "recycled skin graft method" is less invasive, because a donor site for the skin graft is unnecessary. We considered it to be as one of the effective treatments for CPP.
A case of pyoderma gangrenosum which occurred in a patient with non-Hodgkin's lymphoma is described. The lesion started in the foot where dye was introduced for a lymphangiogram.
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.
The successful treatment of patients with pyoderma gangrenosum by means of the local application of sodium cromoglycate solution is reported. One of the patients also had chronic active hepatitis and a background history of ulcerative colitis. Possible mechanisms underlying the apparent success of this form of treatment are discussed.
A 50-year-old man with recurrent life-threatening sepsis and a cutaneous condition resembling pyoderma gangrenosum, was found to have a defect of neutrophil phagocytic function. Phagocytosis could be enhanced by corticosteroids, both in vitro in a dose-dependent manner, and in vivo, when it was accompanied by rapid clinical improvement. Studies with steroid hormones and immuno-stimulatory drugs are described.
Malignant pyoderma is a rare, chronic, destructive ulcerating skin disease of unknown cause that affects young adults, usually males. In the seven previously reported cases, the lesions were distributed mainly about the head and neck region, and some were associated with neurological disturbances. The disease is progressive but responds to high doses of systemically administered steroids. We here describe an additional case and a follow-up of a previously reported case that was managed successfully with corticosteroid and cyclophosphamide therapy.
The previously unrecorded association of superficial bullous pyoderma and IgG-producing multiple myeloma is described. A rapid response of the cutaneous manifestations was achieved by treatment of the malignant paraproteinaemia.
A 49-year-old woman with a 14-year history of pyoderma gangrenosum unresponsive to a variety of systemic and topical treatments was given oral cyclosporin A (CyA), a potent immunosuppressive agent widely used in organ transplantation for the prophylaxis of acute graft-versus-host disease. Improvement and subsequent healing of the lesions was evident within 3 weeks of starting treatment and has continued for the duration of treatment (12 months).
A 19-year-old man with severe acne developed extensive scalp folliculitis and later superficial pyoderma gangrenosum following treatment with isotretinoin. A cyclical neutropenia was noted and bone marrow findings suggested myelodysplasia. We believe that isotretinoin was implicated in the development of overt symptoms in this patient whose haematological condition was previously asymptomatic.
A 78-year-old Caucasian woman developed Sweet's syndrome which progressed over 3 weeks to pyoderma gangrenosum and subcorneal pustule formation. In spite of treatment the patient died and post-mortem examination revealed cryptogenic cirrhosis which could have explained the spectrum of neutrophilic skin disease observed in this patient.
We wish to report the progress of a patient with pyoderma faciale and Crohn's disease. The patient is interesting in that on two occasions the relapse in her skin condition coincided with the introduction of non-steroidal anti-inflammatory drugs. Therapy with isotretinoin was effective and well tolerated.
The successful use of cyclosporin A (CSA) in organ transplantation is now well established. In recent years its usefulness has extended to the treatment of cutaneous autoimmune disorders, including pyoderma gangrenosum (P.G.). We report two further cases of recalcitrant P.G., both associated with rheumatoid arthritis (R.A.) which responded to low dose CSA.