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The authors present three cases which created diagnostic problems. The etiology of these is, in the authors' experience, unique and not generally known. It is suggested that this etiology be considered whenever palatal erythema, ecchymoses, or petechiae are seen.
A survey of twenty-six cases of erythema multiforme was undertaken. All patients had oral lesions and seventeen had skin involvement, mainly on the hands. The average age was 32 years, and female patients predominated in the group. Nearly one third gave a history of a severe emotional disturbance prior to the attack, four patients developed lesions after penicillin therapy, and two presented initially with acute primary herpes simples stomatitis. Skin and mucosal biopsies were undertaken in some patients, and it was concluded that no pathognomonic features were identifiable; a subepithelial bulla, however, could occasionally help in the differential diagnosis. Other laboratory investigations, including a complete blood count, determination of herpesimplex titer, and urinalysis, were not helpful, except that the erythrocyte sedimentation rate was raised in sever cases. Possible etiologic factors were discussed in relation to the survey. It was thought that the cause is still obescure except in drug-induced cases and that the diagnosis is essentially a clinical one, since various laboratory investigations cannot be shown to produce consistent pathognomonic findings.
Patients with erythema multiforme (EM) often have chronic or recurring oral lesions that cause intense pain and interfere with a variety of functions including eating and speech. Previous studies suggest that levamisole restores to normal the function of phagocytes and T lymphocytes, and activates the inflammatory response. In our previous double-blind study 8 of 13 patients with EM had a decrease in severity and frequency of attacks. The purpose of this open prospective study was to evaluate short-term and long-term clinical efficacy of levamisole in patients with mucocutaneous EM. Thirty-nine patients with mucocutaneous EM seen in the Oral Medicine Clinic, School of Dentistry, University of California-San Francisco, comprised our study group. Levamisole was used alone in 17 patients or in combination with prednisone in 22 patients and was given as a single dose of 150 mg/day for 3 consecutive days. Thirty-one patients showed a complete response from levamisole (alone in 13 and in combination with prednisone in 18). Four showed a partial response of signs and symptoms, and four others had no benefits from levamisole whether alone or in combination. The most common side effects from levamisole were skin rash, tiredness, weakness, myalgia, taste change, and insomnia.
Erythema nodosum leprosum (ENL) is a serious complication of lepromatous leprosy, affecting skin and peripheral nerves in a large percentage of these patients, and is presumed to result from spontaneous immunologic changes. Its pathogenesis is poorly understood, although histopathologic features have suggested immune complex (IC)-mediated injury. Abundant circulating antibody is present but no convincing correlation has been established between circulating IC and ENL. We have examined cutaneous leprosy lesions in vivo using blisters induced by prolonged gentle suction to determine whether or not IC are demonstrable in lesions with or without ENL, using an IC assay based on monoclonal rheumatoid factor binding. We also examined whether antibodies involved in such IC are produced locally or reach the skin via the circulation. Surprisingly large quantities of IC were found in ENL lesions, and in some cases the quantities were significantly higher than in matching serum. Total IgG, IgA, and IgM in the skin were not higher than expected, however. Attempts to demonstrate increases in intracutaneous levels of specific anti-Mycobacterium leprae antibodies were unsuccessful. This is the first report of the demonstration of IC in suction blister fluid. The results indicate that large quantities of IC may be present in cutaneous leprosy lesions and are consistent with the hypothesis that they are formed in situ when circulating antibody combines with antigen in the skin. The nature of the antigen in these IC remains undefined.
Topically applied 16-vinylprostaglandins demonstrate the property of rapid transit through the skin and a profound effect on the cutaneous vasculature. At low concentrations in the guinea pig and rabbit. 15-deoxy-16-hydroxy-16-vinyl-PGE2 (DHV-PGE2) and its methyl ester (DHV-PGE2Me) elicit a distinct and persistent erythema which is restricted to the area of application and is not associated with a wheal. Skin temperatures are elevated for several hours following application. Accordingly, these compounds may have therapeutic utility in conditions where local blood flow is compromised or where an enhanced blood flow is desired . In the spontaneously hypertensive rat, topically applied DHV-PGE2 and DHV-PGE2Me produce a dramatic and persistent lowering of blood pressure. The maximal effects are comparable to those obtained wit equal oral or intravenous doses and are maintained for a longer period of time. Moreover, with the topical route, there is no prolongation in the time required for onset of action (3-5 minutes). It appears that while the skin presents only a minimal diffusion barrier to these compounds, a sufficient depot is maintained to give sustained release and prolonged duration. Transdermal delivery of 16-vinyl prostaglandins may offer a convenient means of achieving a clinical antihypertensive effect without the characteristic side effects generally associated with oral or intravascular prostaglandins.
Erythema multiforme (EM) is an inflammatory disorder of the skin, which may include mucous membrane involvement, that features target (iris) lesions. Mediators specifically involved in EM are not well characterized; its pathogenesis remains enigmatic. In this study, evidence for participation of kinins in the pathophysiology of inflammation in EM was investigated by assessing cleavage of high-molecular-weight kininogen (HMWK) in plasma. These data were compared with analyses of plasmas from patients with serum sickness, chronic idiopathic urticaria/angioedema, and from normal control subjects. Patients with EM demonstrated significant levels of circulating cleaved HMWK in plasma during active disease (p less than 0.01), which declined during remission/recovery. Plasmas from patients with EM obtained during active disease also demonstrated significant levels of 94 kd C1 inhibitor (p less than 0.01) and C1 inhibitor-kallikrein complexes. Patients with serum sickness and chronic idiopathic urticaria/angioedema did not demonstrate these findings and did not differ from normal control subjects (p = not significant). Although the kininogenase responsible for HMWK cleavage in EM has not been conclusively demonstrated, these findings suggest that HMWK cleavage resulted from activation of the contact system and that some of the manifestations of EM in selected patients may in part be accounted for by inflammatory and proinflammatory actions of kinins. Based on these preliminary findings, it will be important to establish whether or not HMWK cleavage in EM is a general finding in patients with this disorder. Further investigation is needed to characterize more clearly kininogenase activity and elucidate the possible role of kinin generation in EM.
Hyperpigmentation, pulmonary infiltration, and erythema nodosum occurred in a patient receiving long-term minocycline therapy. Bronchoalveolar lavage revealed both a neutrophilic and an eosinophilic alveolitis. The pulmonary and systemic symptoms promptly resolved after discontinuation of minocycline.
Chemotherapy-induced acral erythema has been described in association with several different chemotherapy regimens. We review the literature on this topic and suggest that different mechanisms may be responsible for the clinically heterogeneous array of acral changes.
A 57-year-old woman with a 6-year history of a dermatitis that evolved into typical necrolytic migratory erythema is reported. Four biopsy specimens were obtained in 5 years. The early lesions revealed superficial perivascular inflammation in the dermis, minor epidermal spongiosis, and scattered dyskeratotic cells in the upper epidermis. The differential diagnosis of this pattern of dyskeratotic dermatitis, particularly in a chronic eruption, should include consideration of hyperglucagonemia and the possibility of an associated pancreatic islet cell tumor.
Erythema gyratum reopens is a slowly expanding, mildly scaling dermatosis with a "wood-grain" pattern and is seen in patients with an underlying malignancy. To date only 49 cases have appeared in the literature, 41 of which (84%) were associated with a neoplasm, most commonly of the lung. Several patients also had pruritus, palmoplantar keratoderma, ichthyosis, vesiculobullous lesions, and/or eosinophilia. Histopathologic findings are nonspecific. The skin findings usually disappear with therapy for the underlying malignancy.
Two women with rheumatoid arthritis developed erythema annulare centrifugum while receiving gold thiomalate therapy. Both patients had similar clinical and histologic findings. Results of a lymphocyte stimulation test were positive in one patient but not in the other.
BACKGROUND: Erythema elevatum diutinum (EED) is a rare, chronic form of cutaneous vasculitis that can result in fibrosis. Four patients infected with the human immunodeficiency virus (HIV) had unusual nodular lesions of EED. OBJECTIVE: Our purpose was to document the occurrence of these lesions in HIV-infected patients and to characterize the histopathologic and immunophenotypic features of nodular EED. METHODS: Clinicopathologic studies were supplemented by special stains of biopsy specimens. RESULTS: EED may be a complication of HIV infection. Nodules of EED are composed of small aggregates of Mac-387+ spindled cells. Within the aggregates can be seen neutrophils, nuclear dust, and fibrin. The marked fibrosis of the lesions may account for the lack of response to dapsone. CONCLUSION: EED should be considered in the differential diagnosis of juxta-articular nodules in HIV-infected patients; its diagnosis can be confirmed by biopsy.
Erythema elevatum diutinum (EED) is a rare disease of unknown origin that belongs to the spectrum of leukocytoclastic vasculitis. Chronic lesions of EED contain lipid deposits, for which the term extracellular cholesterosis has been coined. We studied a typical case of EED with long-standing lesions. Findings of electron microscopic examination revealed a heavy, exclusively intracellular lipid deposition that consisted of lipid droplets, myelin figures, and rare cholesterol clefts within histiocytes but also within epidermal keratinocytes, mast cells, pericytes, and lymphocytes. These findings are in keeping with the results of previous ultrastructural studies and suggest that the term extracellular cholesterosis is a misnomer; intracellular lipidosis would more accurately describe the lipid deposition.
BACKGROUND: Necrolytic migratory erythema (NME) is an uncommon inflammatory dermatosis with a distinct clinical and histologic appearance. NME is usually associated with glucagonoma. Only a few cases of NME in the absence of glucagonoma have been previously reported. OBJECTIVE: We sought to understand further the pathogenesis of NME by analyzing data from three patients. METHODS: Three patients were examined both clinically and histopathologically. RESULTS: Each patient had an extensive erythematous scaling eruption in intertriginous, perioral, and acral areas, and a markedly red, smooth tongue. Skin biopsy specimens showed confluent parakeratosis, epidermal pallor, papillary edema, and a lymphohistiocytic infiltrate. Two patients had alcoholic liver disease and one had liver dysfunction as a result of hemochromatosis. Serum albumin level was depressed, and liver enzyme values were increased in all three patients. Glucagonoma was undetectable in these patients. CONCLUSION: In the absence of glucagonoma, hepatocellular dysfunction and hypoalbuminemia appear to be the most common factors associated with NME.
We performed immunohistologic studies on a 75-year-old white woman with erythema nodosum (EN) and a systemic lymphoma. A skin biopsy specimen from an EN lesion showed lobular and septal pannicular infiltration by atypical lymphocytes. The cutaneous lymphocytic infiltrate was composed of a monoclonal population of B cells with lambda light chains. Atypical lymphocytes were also seen in the peripheral blood, and flow cytometry showed a predominance of the same phenotype of B cells with lambda light chains. Chemotherapy for systemic B-cell lymphoma resulted in the simultaneous resolution of EN and the lymphoma. This is the first documentation of EN representing direct cutaneous invasion by a B-cell lymphoma.
A case of erythema infectiosum (fifth disease) is discussed. As it is a childhood illness that initially presents with a facial rash, the oral and maxillofacial surgeon will often be the first practitioner to diagnose this interesting, but benign, disease.
The author describes two patients with erythema ab igne, a condition which used to be common before the introduction of central heating. Sporadic examples of the condition are still seen and treated.
At the time of birth, the fetal ground substance is under the influence of maternal and placental hormones. Hormones are known to exert significant effect on ground substance. The ground substance viscosity that is ideal for intrauterine life is too viscous for the newborn. Sites of minor skin trauma develop swelling, inflammation and pustules. Dilution of tissue fluids causes formation of some of the mediators of inflammation including those responsible for intraepidermal pustule formation. The newborn responds to inflammation with eosinophilic granulocyte. The clinical lesions referred to as erythema toxicum neonatorum are known to localize at pressure sites. The skin lesions correct as maternal and placental hormone influence weakens. The entity occurs in 31 to 72% of white infants. The hormone induced viscosity changes may aid in better understanding some aspects of neonatal physiology; such as jaundice of the neonatal period, the eosinophilic granulocyte inflammatory response of the neonatal period and hyaline membrane disease.