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The authors report a case of pyoderma gangrenosum associated with a positive B27 pelvispondylitis. The search for a hemorrhagic rectocolitis, possible link between the two syndromes, revealed to be negative. Pelvispondylitis is an unusual complication of pyoderma gangrenosum. Review of the literature shows only one case of sacroiliitis, beside seronegative polyarthritis unrelated to HLA B27.
Four patients with pyoderma gangrenosum were treated with hyperbaric oxygen to prepare the wounds for skin grafting. Each wound responded to a course of daily hyperbaric oxygen with reduction of infection and increased capillary angiogenesis. During follow-up periods of 12 to 30 months, all wounds remained healed. Although the exact etiology of pyoderma gangrenosum is unknown, vasculitis with wound ischemia and infection are prominent components. Inspired oxygen partial pressures of 1100 to 1300 mmHg elevate wound oxygen tension despite relative ischemia. The impaired intracellular bacterial killing of hypoxic leukocytes is corrected during each day's 2-hour bolus of hyperbaric oxygen. Daily wound oxygenation increases collagen production by fibroblasts to support capillary angiogenesis.
Five cases of pyoderma gangrenosum occurring in a kindred are presented. Three of the cases occurred after abdominal surgery and tended to be confused with postoperative wound infections. Two cases occurred after superficial injury to the leg and were also thought to represent a peculiar form of cellulitis. None of the patients are known to have any of the underlying diseases usually associated with pyoderma gangrenosum. The cases are presented to alert the physician to this entity and to document the unusual familial occurrence.
Fourteen cases of pyoderma gangrenosum are reported. Twelve were associated with various diseases: ulcerative colitis, Crohn disease, colic tumor, rheumatoid arthritis, chronic myeloid leukemia, agammaglobulinemia, respiratory infections (2 cases) and post surgical intervention. In 2 cases pyoderma gangrenosum appeared to be primary. The clinical characteristics of the affection make it a very particular type of ulcer: pustular onset, frequent localization to other than leg, rapid eccentric spread, frequent associated morbidity (mainly digestive and hematologic). The pathogenesis is still unknown and the treatment is not well codified.
The typical lesion of pyoderma gangrenosum is a rapidly extending necrotic ulceration, often associated with a system affection, inflammatory colitis, hematologic affections, rheumatic disorders. In about 30% of cases it develops on the site of a sometimes minimal injury. In the two cases reported one developed in the region of a graft after excision of a basal cell epithelioma, the other in a peritoneal drainage scar after colon resection. In patients with pyoderma gangrenosum, initial discussion must involve possible causes of the cutaneous necrosis, but in the cases reported the clinical appearance and rapid progression of the lesion, and negative results of serial bacteriologic and mycologic examinations were in favor of this diagnosis. An accurate diagnosis is essential since general corticotherapy is usually necessary.
A 46-year-old man was diagnosed as having pyoderma gangrenosum after special stains and cultures from a skin biopsy specimen were reported as negative. Cutaneous sporotrichosis is usually diagnosed with relative ease on the basis of clinicopathologic features and prompt growth of the fungus in culture, although organisms are difficult to detect in tissue even with special stains. Identification of Sporothrix schenckii was delayed for three months in this patient because of unusual growth characteristics noted in the culture. The report of this patient's clinical course illustrates both the need to frequently reassess the diagnosis of pyoderma gangrenosum in treatment-resistant patients and the fact that S schenckii may be difficult to diagnose clinically and mycologically if the growth characteristics of the fungus are unusual.
Host defense mechanisms were studied in a patient with recurrent pyoderma of the scalp. Evaluation of the patient's inflammatory response demonstrated normal yeast phagocytosis, normal capillary tube migration, normal results from a nitroblue tetrazolium dye test, and significantly decreased neutrophil chemotactic response (NC). The impaired NC was associated with a heat labile plasma inhibitor. Chromatography of the patient's and of normal human plasma demonstrated three distinct protein peaks. Chemotactic inhibitory activity was found in the third peak of the patient's plasma but not in the control plasma. Normal in vitro NC was restored when greater than 40% normal human plasma was added to the column fractions that contained the inhibitor. Based on these findings, a subsequent exacerbation of the patient's pyoderma was treated with fresh frozen plasma, and dramatic clinical improvement occurred within 72 hours.
The case is presented of a patient with busulfan (Myleran) treated myeloid leukaemia, who developed bullous pyoderma gangrenosum. Skin symptoms appeared at the time when treatment was discontinued due to signs of bone marrow depression. The pyoderma disappeared following treatment with systemic steroid.
Three cases of pyoderma gangraenosum were associated with Takayasu arteritis (pulseless disease), characterized by disappearance of pulse, hypertension and/or aortic insufficiency. Another 11-year-old boy, suffering from a serious congenital heart malformation, presented skin lesions of pyoderma gangraenosum. In spite or rather low incidence of association of ulcerative colitis (10.4 p. 100), Japanese cases show frequent association of cardiovascular diseases, especially Takayasu arteritis (27.1 p. 100).
Two new cases of pyoderma gangrenosum with chronic myeloid leukemia are described and added to three others of the literature. Pyoderma gangrenosum is often bullous, painful and progresses quickly. It seems to have a bad prognosis in myeloid leukemia as in polycythemia vera, in which it appears lately as an evolution to myelofibrosis or acute leukemia transformation. Chemotactic disorders of polynuclear may be the main etiological factor in these cases.
Cefoperazone (CPZ), a new semisynthetic cephalosporin derivative, was administered in the treatment of pyoderma. The results are shown as follows. CPZ was given to 21 patients with pyoderma. In 21 patients, marked improvement was seen in 7, effectiveness in 10, slight improvement in 1 and ineffective in 3. Total clinical effectiveness rate was 81.0%. No serious side effects were observed.
Two patients with pyoderma gangrenosum and ulcerative colitis are described. To our knowledge this is the first report of such an association in Blacks. The unusual vesicopustular form of pyoderma gangrenosum was well demonstrated by one patient who also responded remarkably to clofazimine. The associations, pathological findings and therapy are briefly reviewed. Colectomy may ameliorate the skin lesions when conventional therapy is unsuccessful.
A review of our records disclosed eight children with pyoderma gangrenosum. All had associated inflammatory bowel disease, and four had arthritis. In three patients the bowel disease was severe and required resection of portions of the affected bowel. The cutaneous lesions had a chronic course (average duration, 2 1/2 years), and systemic therapy was required in seven cases. Previously reported cases of pyoderma gangrenosum in children are reviewed, and the management of this skin disorder in childhood is discussed.
Pyoderma gangrenosum is a relatively rare, destructive, inflammatory disease of unknown cause which may present as a purely cutaneous disorder or may be associated with an underlying internal disease (ulcerative colitis, Crohn's disease, rheumatoid arthritis, multiple myeloma, lymphoma and others). Four selected patients are described which reflect the clinical spectrum of this condition; these cases and a review of the literature serve as a background for analysis of pyoderma gangraenosum as an entity and a discussion of its pathogenesis.
A cases of Pyoderma vegetans attacking only the outer skin of the eyelid and leaving free the conjunctiva is reported. The diagnosis of this rare condition, belonging to the pemphigus diseases, depends on clinical course and especially on histological investigation. The aetiology of Pyoderma vegetans still remains unknown, an immun-pathological mechanism is discussed.
Pyoderma gangrenosum is a clinical diagnosis. The ulcer shows three absolutely distinctive morphologic features: a purple-red, raised, undermined border; an irregular base, and cribriform scarring at the periphery. Evaluation of a patient with pyoderma gangrenosum requires a systematic search for possible associated inflammatory bowel disease or other internal disease. Although a variety of local treatments are used, systemic steroids are almost always necessary.
The authors report one case of Pyoderma gangrenosum whose unusual cervical localization was particularly bothering. Besides, the patient, an 18-year-old girl, had clinical symptoms of Crohn's disease confirmed by radiological investigations and bowel pathology. A benign monoclonal gammapathy and an auto-immune haemolytic anaemia were also found. After four plasma exchanges Pyoderma healed rapidly, Crohn's disease stabilized and biological abnormalities disappeared. Comments and review of the literature are reported. It appears that such a case is quite exceptional, as far as clinical, biological data and therapy (plasma exchange) are concerned.
INTRODUCTION: The term "neutrophilic dermatosis" is normally used to cover a number of dermatoses characterized by their response to corticotherapy or to drugs effective on the neutrophilic polynuclear. Neutrophilic dermatosis was frequently associated with hematologic disease. We study the case of a neutrophilic dermatosis accompanied by a pulmonary involvement and then review the literature on the reported cases of neutrophilic dermatosis with extra-cutaneous localisations. CASE STUDY: A woman aged 54 displayed a pyoderma gangrenosum successively followed by a pustular dermatosis (related to subcorneal pustular dermatosis of Sneddon-Wilkinson) and by a pulmonary involvement not caused by any underlying infection. The laboratory results revealed a benign IgA gammaglobulin. The use of corticotherapy and a relay by dapsone led to the gradual disappearance of the cutaneous and pulmonary lesions. DISCUSSION: Pulmonary involvement have been observed in the Sweet syndrome and in the pyoderma gangrenosum. The neutrophilic lung is diagnosed once a tumorous or infectious cause has been eliminated and on the basis of the response to the cortisone treatment. CONCLUSION: The link between pulmonary involvement and neutrophilic dermatosis is sometimes mentioned but is in fact often difficult to confirm. The positive reaction to corticosteroids could be a diagnostic test.