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Pyoderma gangrenosum appeared parastomally after proctocolectomy and ileostomy for ulcerative colitis in two patients. The cases are described and diagnosis and treatment are discussed.
Pyoderma gangrenosum is an ulcerative skin disorder usually associated with an underlying systemic disease. Head and neck involvement is rare, but possibly more common than once thought. The etiology of this disease is unclear, but may be related to an abnormal immunologic response. There are no pathognomonic histologic or laboratory findings; the diagnosis is made by the clinical appearance of the lesions and disease course. Treatment consists of immunosuppression and local wound care in addition to a search for and treatment of an underlying primary systemic disorder. A case report and review of the literature is presented with discussion of common head and neck manifestations, the differential diagnosis, and treatment alternatives.
The function of microphages has been studied in two patients with chronic pyoderma vegetans by in vitro determination of phagocytosis as well as chemotaxis. The results showed a striking decrease in the chemotactic activity of the neutrophil granulocytes, a reduced phagocytosis of Candida albicans and Staphylococcus aureus, and a weakness of the intracellular killing of these microorganisms. However, the NADH-dependent oxidase activity appeared to be intact. No defect was found in the specific cellular or humoral immune system in either patient.
Pyoderma gangrenosum is a serious skin disorder. About half of the cases are associated with systemic diseases such as inflammatory bowel disease or arthritis. Many management regimens have been recommended, but treatment must be individualized and must include concern for the physical and psychologic needs of the patient. This particularly stressful and uncertain lesion is a challenge to the physician.
The clinical, histologic, and immunofluorescent findings in 86 cases of pyoderma gangrenosum seen at the Mayo Clinic between 1970 and 1983 were reviewed. Males and females were affected in equal numbers. The most frequent site of lesions was the leg. Sixty-seven patients (78 per cent) had associated systemic disease, with arthritis and inflammatory bowel disease being commonest. Cutaneous histopathologic changes varied with the site of biopsy. Lymphocytic vasculitis was predominant in the zone of erythema peripheral to the area of ulceration, while neutrophilic infiltrate and abscess formation were more prominent centrally. In most cases studied, direct immunofluorescence showed immunoglobulins and complement deposited in and around superficial and deep dermal vessels.
The function of microphages has been studied in two patients with chronic pyoderma vegetans by in vitro determination of phagocytosis as well as chemotaxis. The results showed a striking decrease in the chemotactic activity of the neutrophil granulocytes, a reduced phagocytosis of Candida albicans and Staphylococcus aureus, and a weakness of the intracellular killing of these microorganisms. However, the NADH-dependent oxidase activity appeared to be intact. No defect was found in the specific cellular or humoral immune system in either patient.
We report on a 69-year-old woman suffering from an erosive pustular and crusted lesion of the scalp having persisted over years. There could not be found any external cause, and a large number of different physical and medicamental therapies failed. Prompt healing was achieved, however, after a low serum zinc level was corrected by a zinc preparation administered orally.-The dermatosis described closely resembles the so-called "erosive pustular dermatosis of the scalp". A search of the earlier dermatological literature supported our view that this is not a new disease, but rather a variant of pyoderma vegetans, a disease showing extremely variable clinical and histopathological features.
We report the successful trial of RePUVA in a patient who had suffered from chronic vegetating pyoderma for 5 years and who had responded poorly to the preceding treatments.
The authors examined 1132 patients with chronic ulcerative colitis (CUC) seen at the Cleveland Clinic to clarify the relationship between the clinical features of colitis and the incidence and severity of erythema nodosum (EN) and pyoderma gangrenosum (PG). There were 21 patients (2%) with EN and 21 (2%) with PG, both of which affected those with active and extensive colitis. CUC was inactive in only 10% of the EN group and 20% of the PG group; it was left-sided in 20% of the EN group and 15% of the PG group. The mean duration of CUC before the appearance of EN and PG was 5 and 10 years, respectively. Three patients had consulted a dermatologist for PG before a clinical diagnosis of CUC was made. EN and PG recurred in 20 and 33% of cases, respectively. EN appeared almost exclusively on the legs, while PG appeared on various areas of the skin, including the site of ileostomy in one patient. EN was controlled with adequate medical or surgical treatment of CUC, but PG was relatively refractory, requiring specific treatment of its own in 30% of patients. In one case each, EN and PG recurred after subtotal colectomy but had resolved completely after proctectomy. In one patient, PG developed for the first time 1 year after total colectomy. Less than half of EN patients and two-thirds of PG patients ultimately had to undergo colectomy because of bowel disease. No patient required colectomy because of skin lesions.
Butchers seldom file for workman's compensation for the occupational disease pyoderma. In the case presented here there is a clear temporal correspondence between the practice of the trade and the onset of the disease. It is suggested that the disease, contracted by humans from animals, be recognized under number 3107 of the BKVO (list of occupational diseases recognized by West German law). No factors were found outside the butcher's trade which could have contributed to the pyodermia.
Ten patients with pyoderma gangrenosum, seven female and three male, 25 to 94 years old, mainly with multiple lesions, have been treated with clofazimine, 100 mg three times daily. Associated disease was registered in three patients: diabetes mellitus, a previous adenocarcinoma of the colon treated by hemicolectomy, and pustulosis palmoplantaris. In a further patient, M-component was found in the serum. In seven cases the lesions were completely healed by two to five months of therapy, and in three cases the ulcers healed partially. Side effects were redness of the skin in seven cases and dryness of the skin in two patients. No hematological side effects occurred. The working mechanism is still obscure.
Crohn's ileocolitis manifested in 2 Black patients with the exceptionally rare complications of pyoderma gangrenosum and sclerosing cholangitis. It is emphasized that Crohn's disease is very rare in the Black population of sub-Saharan Africa, and, as far as we are aware, no patient with large-bowel involvement has hitherto been reported.
A 69-year-old man with recurrent eruptions of pyoderma gangrenosum for 4 years is described. The patient also suffered from paraproteinemia (increased IgA with M-component) and transient acantholytic dermatosis resembling pemphigus erythematosus. He had no intestinal symptoms.
Seven patients with pyoderma gangrenosum have been treated with clofazimine. In contrast to the beneficial effect described in other articles only one healed. (Received March 27, 1983.)
A 46-year-old man with pyoderma gangrenosum and IgA gammopathy is described. This patient had no other identifiable associated systemic disease. His condition was unresponsive to oral corticosteroids, sulfones, and sulfapyridine therapy. His course was complicated by chronic sinusitis and necrotizing pneumonia. He was treated with clofazimine.
Pyoderma gangrenosum (PG) is a characteristic ulcerative condition of unknown etiology. Four cases are reported of patients with PG who responded to minocycline hydrochloride therapy. Possible mechanisms for this apparently efficacious modality are discussed.
This is a report of a 24-year follow-up of a man now 33 years of age, who suffers almost continuously from severe inflammatory lesions of the lips, nose and eyelids, with increased susceptibility to respiratory infections since early childhood. The condition, previously described as "pyo-rhino-blepharo-stomatitis vegetans (McCarthy)", was treated with systemic corticosteroids and antimicrobial agents for years, but failed to improve until the immune status of the patient was checked after withdrawal of the steroids. T lymphocytes were found to be abnormal as to count in peripheral blood and various functional qualities determined in vivo and in vitro. For treatment, levamisole and thymopoietin pentapeptide (TP-5) were given. Subsequently each drug induced rapid and complete clearing of all lesions, but was followed by the recurrence of facial periorificial lesions after drug withdrawal. Change of the regimen by administering either inosiplex orally or commercial calf thymus extract parenterally, remained ineffective. During therapy with levamisole as well as TP-5, the number of T lymphocytes in peripheral blood normalized, yet impaired functions failed to improve. There was an elevated ratio of T-suppressor/T-inducer cells in blood using OKT antibodies. In vitro testing of different functions of polymorphonuclear leucocytes revealed normal results except for a slight decrease of chemotactic activity during levamisole. In view of the long clinical course, the mass of clinical and immunological data collected over decades, and the therapeutic results as a whole, the disease can be characterized as a peculiar type of pluriorificial pyoderma vegetans, caused by a distinct immunodeficiency of T lymphocytes.
Pyoderma gangraenosum (Pg) is regarded as an indicator of internal primary diseases of mainly auto-immunological character. Gammopathies as basic diseases are very often of the IgA-type. In contrast to the characteristic clinical picture we have observed unspecific, cellular traits. New investigations have reported immunological findings such as deposition of IgG and IgM as well as C3-complexes at the vascular walls. A decreased function of T-cells is described. Pg often develops in consequence of minor traumata. In these cases the disproportion between the primary lesion and the ulcerous process becomes obvious. By presentation of a typical case in addition to a literature survey, it is discussed whether the primary disease is responsible for this development. Because of the close relationship between Pg and the underlying immunopathology it is proposed that Pg - in analogy to the neoplastic cutaneous syndrome - be classified dipolar-like as a paraimmunological syndrome.