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We report a woman with erythema palmare hereditarium, an anomaly not documented so far in the German literature. It is characterized by a bright erythema of the palms, usually persistent since birth. It is transmitted in an autosomal-recessive mode and has a benign course. In our patient it was possible to trace the palmar erythema over three generations.
OBJECTIVES: Diclofenac is a non-steroidal anti-inflammatory drug used for a variety of painful and inflammatory conditions. A new low-dose, topical-gel form of diclofenac sodium (diclofenac-Na) has been developed for pain relief and redness reduction after sunburn. The objective was to compare exposure to oral diclofenac-Na with the systemic exposure to diclofenac after application of the new topical diclofenac-Na 0.1% Emulgel gel (diclofenac-Na gel) to normal skin and to that with ultraviolet-induced erythema relative. METHODS: This study was an open, single-centre, three-period, non-randomised trial in 18 healthy Caucasian subjects. During the first period, 12.5 g gel (12.5 mg diclofenac-Na) was applied twice on a single day to normal skin. During the second period, a 25-mg diclofenac-Na, enteric-coated tablet was given orally three times in a single day. During the third period, the diclofenac-Na gel was applied, as in the first period, but during the early phase of an erythema induced by three times the ultraviolet minimal erythema dose, i.e. a first-degree sunburn associated with pain. During each period, venous blood samples were collected over 24 h and urine was collected over 72 h after first administration for the determination of diclofenac in plasma and urine and of 4'-OH-diclofenac in urine. RESULTS: The systemic exposure after topical application of 25 mg diclofenac-Na on sunburned skin was less than 3% that of 75 mg oral diclofenac-Na and was not increased to that measured on normal skin. CONCLUSION: The diclofenac-Na 0.1% Emulgel gel can be applied safely to sunburned skin (superficial sunburn, i.e. first degree) as well as to normal skin.
The available physical and biological broad-band radiometers designed to determine erythema-effective radiation do not show any response or over/underestimate the biologically effective radiation to a high extent in the ultraviolet (UV)A spectral region. The data presented in this paper demonstrate that the biological system used in this study is the first one to make possible measurements of erythema-effective radiation in the sun in the UVA and UVB spectral region. These measurements were performed with a spore-film filter system as well as with spectroradiometers. It was demonstrated that this biotechnological method could be used to determine exact values expressed as minimal erythemal dose (MED). The spore-film system was tested in various field campaigns performed in Germany and in Japan. The seasonal daily variation of UV radiation in Germany determined in the period November 1995 to December 1996 using the spore-film filter system in sunny conditions tallied well with model calculations. The daily dose in Germany measured with the spore-film system close to the summer solstice, in sunny conditions (20.45 MED), was approximately 20 times higher than the lowest value measured close to the winter solstice (0.82 MED), a result which was in accordance with model calculations. The data determined with the spore-film filter system in Sapporo and Naha, Japan, fitted to the erythema-weighted data calculated from spectroradiometric measurements (Brewer), even at low solar radiation angles in a solar spectrum with less UVB but significant UVA. The spore-film dosimeter values were about 103 +/- 8% of the integrated dose of the Brewer instrument. The standard deviation of the spore-film measurements obtained in Japan was 12.8%. The responsivity of the spore-film system towards longer wavelengths within the UVA spectrum was tested with the Okasaki Large Spectrograph with monochromatic radiation. At a wavelength of 365 nm--in a spectral region which is dominant in many tanning lamps and with minor importance for solar radiation in summer conditions--the tested spore-film system gave results that were close (112% compared to the calibration dose) to the calibration dose which was used for irradiation.
Behçet's (BD) is a systemic inflammatory disease with histological evidence for vasculitis. Leucocyte-leucocyte and leucocyte-endothelial cell interactions are critical in inflammatory reactions that are influenced by the expression, activation and shedding of adhesion molecules. We investigated the expression of some adhesion molecules (E- and L-selectin, VLA-4, ICAM-1, PECAM-1, VCAM-1 and CD18 and CD11c chains of beta-2 integrins) on endothelial and inflammatory cells by immunohistochemistry on cryostat sections of erythema nodosum lesions taken from 15 patients with BD and 12 patients with erythema nodosum of unknown cause. Hematoxylin-eosin stained sections of all specimens were also assessed. The major histopathological findings were perivascular mononuclear cell infiltration and secondary vasculitic changes with no difference between the two groups (P > 0.05). However, the frequency of thrombophlebitis was higher in BD (P < 0.001). Endothelial and inflammatory cell adhesion molecule expression did not show any significant difference between groups (P > 0.05). Although VCAM-1 expression and intensity on endothelial cells of BD patients seemed to be lower, this did not reach statistical significance (P = 0.056). We concluded that subcutaneous thrombophlebitis is an important feature of erythema nodosum like lesions in BD, which is almost impossible to understand by physical examination alone. Colchicine, which is known to have some influence on adhesion molecules, might have affected our results, as these showed no significant difference regarding adhesion molecules between the two groups.
UNLABELLED: Erythema necrolyticum migrans (ENM) usually presents as a cutaneous paraneoplastic phenomenon which is in most cases associated with a glucagon-producing tumour. Here it is for the first time described as a side-effect of glucagon treatment in persistent hyperinsulinaemic hypoglycaemia of infancy (PHHI). In both patients, the skin lesions disappeared after discontinuation of glucagon administration. In the first child the erythema resolved without scarring within 10 days after glucagon was substituted with other medication while in the second patient healing followed subtotal pancreatectomy which rendered glucagon infusion unnecessary. Initially the clinical resemblance to atopic dermatitis is prone to cause diagnostic errors, especially in this age group. CONCLUSION: erythema necrolyticum migrans should be considered as a differential diagnosis in patients who develop erythematosquamous skin lesions under glucagon treatment.
UNLABELLED: A review was made of the 36 paediatric patients in whom the diagnosis of erythema nodosum had been established between 1977 and 1996 at the Department of Paediatrics, University of Bern, Switzerland. Infectious diseases were associated with erythema nodosum in 20 (including 10 streptococcal infections) and non-infectious inflammatory diseases in 8 patients. None of the 36 patients had tuberculosis or had been exposed to sulphonamides, phenytoin or hormonal contraceptives. There were eight patients in whom either the associated disease was not diagnosed, or there was no other disease. CONCLUSION: Most cases of erythema nodosum are nowadays caused by non-mycobacterial infectious diseases or by non-infectious inflammatory diseases.
BACKGROUND: Controversy exists concerning the role of bile reflux and Helicobacter pylori ( H. pylori) infection in the development of inflammation of the gastric remnant after gastrectomy. This study was designed to investigate association of bile reflux and H. pylori infection or both with inflammatory changes in the gastric remnant. METHODS: A questionnaire on GI symptoms was returned by 200 gastrectomy patients, and 24-h bilirubin monitoring in the gastric remnant was performed on 55 patients with Bilitec 2000. Upper GI endoscopy evaluated reflux gastritis in the gastric remnant, and the presence of H. pylori infection and chronic, active inflammatory cellular infiltration in the biopsy specimens were examined microscopically with the updated Sydney system. RESULTS: No difference in the incidence of GI symptoms was observed among individual gastrectomy patients. Bile reflux was lower in patients who had undergone a gastrectomy with jejunal interposition, a pylorus-preserving gastrectomy, and a gastrectomy with Roux-Y anastomosis than those who had undergone a Billroth-II (B-II) anastomosis ( P < 0.05). Endoscopy showed positive correlation between mucosal erythema and bile reflux ( P < 0.001). No correlation was observed between the mucosal erythema and chronic and active inflammatory cellular infiltration. Infection of H. pylori correlated with chronic and active inflammatory cellular infiltration ( P < 0.001). Bile reflux did not correlate with the severity of chronic and active inflammatory cellular infiltration or H. pylori infection. CONCLUSIONS: Bile reflux into the gastric remnant was observed by Bilitec 2000. Mucosal erythema and chronic, active inflammatory cell infiltration in the gastric remnant after gastrectomy may be caused by bile reflux or H. pylori infection, respectively.
Simultaneous occurrence of Sweet's syndrome and erythema nodosum is very rare. We describe a case of a young male with a recent history of streptococcal infection who presented with concurrent Sweet's syndrome and erythema nodosum. Although the exact pathogenesis of these dermatoses is not yet clear, their similarities and simultaneous occurrence suggest a possible common underlying mechanism and may represent a continuum of reactive dermatoses. Evaluation of the role of cytokines in the etiopathogenesis of these conditions will be useful for further assessment and treatment of these conditions. Like the association of acanthosis nigricans and certain cancers and diabetes, Sweet's syndrome and erythema nodosum may be associated with certain malignancies, autoimmune disorders, or inflammatory bowel disease. Early recognition of these skin lesions can guide a search for underlying disorders. Patients with Sweet's syndrome should undergo an age-appropriate work-up for malignancy.
The etiology of erythema induratum, a rare disease of the skin in the United States but occasionally seen in natives of Asian countries, remains a source of debate. Its association with tuberculosis, although strongly suspected for more than one century, has not been clearly defined. We report a case of erythema induratum occurring in a young Chinese woman in the setting of active pulmonary tuberculosis. Both diseases promptly responded to antituberculous therapy. The diagnosis of erythema induratum should urge the clinician to search for a source of active tuberculosis, and treatment should be initiated accordingly.
As evidence of vascular involvement in classical erythema nodosum unassociated with Behçet's disease, the occurrence of endothelial cell necrosis based on the development of dark cell degeneration was observed by electron microscopy in the vasculature of the dermis and subcutaneous fat. The incidence of endothelial cell necrosis in all segments of the microcirculatory system was roughly 20% in three of 12 patients. The remaining nine patients showed a very low rate of necrosis, including two patients in whom endothelial cell necrosis was totally absent. The presence of necrotic endothelial cells that are forced out and eliminated by neighboring intact endothelial cells into the vascular lumen is closely associated with thrombus formation in the cutaneous vasculature. The incidence at which endothelial cell necrosis occurs in classical erythema nodosum is not always high; however, it is concluded that endothelial cell necrosis may play a significant role in the pathogenesis of erythema nodosum.
The folic acid antagonist, methotrexate, has many applications in the treatment of neoplastic disease. While methotrexate produces several well-recognized toxic effects, cutaneous reactions are rare. A patient who developed classical erythema multiforme while receiving low-dose methotrexate as treatment of nonmetastatic gestational trophoblastic neoplasia is presented. Erythema multiforme has been associated with a variety of pharmacologic agents. It typically presents as a pruritic papular dermatitis of the extensor surfaces of the extremities and may require multiple skin biopsies to establish the diagnosis. Spontaneous reversal usually occurs with discontinuation of therapy. Patients developing erythema multiforme related to antineoplastic agents should be switched to an alternate regimen.
OBJECTIVE: To investigate if the occurrence of subcutaneous fibrosis after radiotherapy in an unselected group of breast cancer patients is related to cellular radiosensitivity of skin fibroblasts as measured in a clonogenic assay. MATERIALS AND METHODS: An in vitro colony-forming assay of normal fibroblast radiosensitivity was applied to primary skin biopsies from 31 breast cancer patients who received post-mastectomy radiotherapy with large doses per fraction (2.7-3.9 Gy) more than 10 years earlier. Three clinical normal-tissue endpoints were assessed. Two late endpoints, subcutaneous fibrosis and telangiectasia, were evaluated in three treatment fields by a single experienced clinician. In addition, skin erythema had been assessed at the end of treatment by members of the staff and junior staff. From previous analyses of normal tissue response, individual clinical radiosensitivity could be assessed as "excess risk' of each of the three reactions. This was defined as the difference between the actual observed response in the patient and the expected response estimated from individual treatment characteristics in a linear quadratic (LQ) mixture model and, for the two late endpoints, with correction for the follow-up time. This clinical radioresponsiveness was compared with the in vitro radiosensitivity of the skin fibroblasts. To this end, the fractions of colony-forming cells after graded single doses were fitted by an LQ survival curve using non-linear regression from which the surviving fraction at 3.5 Gy (SF3.5) was estimated. Assessment at 3.5 Gy was chosen to reflect the fraction size during clinical radiotherapy. RESULTS: A statistically significant variability of in vitro radiosensitivity between patients could be detected for both SF2 (P = 0.0095) and SF3.5 (P = 0.0008). A significant correlation was observed between SF3.5 and excess risk of fibrosis (rs = -0.46, P = 0.009) while no association was found between fibroblast radiosensitivity and either the occurrence of severe skin telangiectasia or the acute endpoint skin erythema. CONCLUSION: These results suggest that variability in the occurrence of subcutaneous fibrosis, but not telangiectasia or erythema, after radiotherapy is partly accounted for by differences in cellular radiosensitivity of normal skin fibroblasts.
We report three cases of patients with erythema induratum of Bazin who showed extreme tuberculin hypersensitivity reactions and a good therapeutic response to isoniazid. In the first patient satellite lesions with epithelioid granuloma formation developed 3 weeks after injection of purified protein derivative. In the second patient a high fever developed 1 day after injection of purified protein derivative, and the third patient blistered at the injection site; the histopathologic pattern was similar to erythema multiforme. Immunohistologic study of the epithelioid granulomas demonstrated a predominance of Leu-1--bearing, Leu-3a--bearing, and human lymphocyte antigen-DR-expressing cells. We conclude that erythema induratum of Bazin represents a tuberculid caused by heightened delayed-type hypersensitivity to components of tubercle bacilli released from latent foci of infection.
Drug-induced, photosensitive erythema multiforme has not been reported, although drugs and sunlight are listed among precipitating factors in erythema multiforme. We describe a case of a drug-induced erythema multiforme-like eruption in a photodistribution that was reproduced by clinical challenge with the drug and sunlight. On contact with Rhus verniciflua, the Japanese lacquer tree, the patient had a flare of the eruption, which was limited to the areas previously exposed to sun. Immunohistochemical studies suggested that the keratinocytes in the skin that retain teh photoactivated substances may facilitate epidermal invasion of lymphocytes by persistent expression of intercellular adhesion molecule-1.
The case of a 57-year-old diabetic man with necrolytic migratory erythema in the absence of glucagonoma is reported. The clinical and pathologic features of his dermatitis and subsequent clinical course are compared with those of canine superficial necrolytic dermatitis, an unusual cutaneous necrotizing eruption of dogs that is identical histologically to necrolytic migratory erythema. In addition to a necrolytic dermatitis, both our patient and most dogs with superficial necrolytic dermatitis have diabetes mellitus and hepatic failure in the absence of glucagonoma. Thus hyperglucagonemia most likely is not a factor in the origin of the necrotizing dermatitis in this patient or in dogs. The role of hepatocellular dysfunction in the pathogenesis of necrolytic migratory erythema and superficial necrolytic dermatitis is considered.
The glucagonoma syndrome is characterized by elevated serum glucagon, a pancreatic alpha-cell tumor, anemia, hypoaminoacidemia, and necrolytic migratory erythema. Necrolytic migratory erythema may cause marked morbidity and is frequently misdiagnosed. A 42-year-old white woman with a 1 1/2-year history of refractory dermatitis (most severe on the lower extremities) had the glucagonoma syndrome. Her severe morbidity was markedly relieved with the administration of intravenous amino acids. This therapy was successful in controlling the necrolytic migratory erythema through recurrences after somatostatin (SMS 201-995), surgical debulking, and chemotherapy proved inadequate.
BACKGROUND: Recurrent annular erythema associated with the anti-Ro(SS-A) antibody response has recently been reported in Orientals. The association is assumed to represent a distinct clinical entity. OBJECTIVE: Our purpose was to extend knowledge on the immunogenetic spectrum of the disease. METHODS: Sixteen anti-Ro(SS-A)-positive Japanese patients with recurrent annular erythema and Sjögren's syndrome were studied. The standard complement-dependent microcytotoxicity assay was used to type the HLA-A, -B, -C, -DR, and -DQ, as well as the HLA-DRw52 and -DRw53 antigens. RESULTS: All 16 patients were positive for HLA-DRw52 antigens as compared with 52% of control subjects (p < 0.01 relative risk 14.8). No significant deviations were noted in the phenotype frequencies for HLA-A, -B, -C, and -DQ antigens. CONCLUSION: HLA-DRw52 is closely related to annular erythema in anti-Ro(SS-A)-positive Japanese patients with Sjögren's syndrome.
A 69-year-old man had erythema elevatum diutinum for several years before he developed IgA paraproteinemia and a limited form of Wegener's granulomatosis. This is the first report of an association between erythema elevatum diutinum and Wegener's granulomatosis. IgA paraproteinemia has been reported in association with erythema elevatum diutinum but not with Wegener's granulomatosis.