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PubMed2002

Erythema annulare centrifugum: report of a case with neonatal onset.

Darier's erythema annulare centrifugum (EAC) is often associated with infectious, autoimmune or neoplastic disease, nevertheless, most cases of EAC remain unexplained. We report a case of EAC with neonatal onset and a clinical course of over 20 years. The patient presented from the 6th day of life with erythematous papules that enlarged centrifugally to form figurate, annular or policyclic plaques involving the entire cutaneous surface. The clinical picture had a chronic course, disappearing only during unrelated febrile episodes. Histologic examination revealed a normal epidermis and a dense perivascular lymphohistiocytic infiltrate and numerous eosinophils in the superficial and reticular dermis. Today the patient is 24 years old and his lesions are still unchanged. Recently, clinical evaluation revealed a diffuse sderodermic induration. A second biopsy showed a diffuse thickening of collagen fibers in the dermis. To our knowledge, this is the second report of EAC with neonatal onset and such a long and peculiar clinical course.

Age of Onset↗
PubMed2003

Erythema multiforme ID reaction in atypical dermatophytosis: a case report.

BACKGROUND: Erythema multiforme (EM) is an uncommon mucocutaneous disorder, characterized by widespread erythematous-oedematous targetoid lesions, considered as a reaction pattern to many different stimuli. OBSERVATION: Presentation is made of a classical EM eruption concomitant to an inflammatory, atypical dermatophytosis of the ala nasi, observed in a 37-year-old male at the Dermatology Department of Cagliari, Italy. METHODS: Laboratory investigations revealed normal complete blood count and biochemistry profile. A direct microscopic examination with 20% potassium hydroxide of the nasal skin scales and of the vibrissae indicated the presence of dermatophyte hyphae and spores invading the rhinothrix. Culture confirmed a Trycophyton mentagrophytes infection. Specific determination of antiviral immunity in the blood (TORCH) was negative for recent infections, while the cell-mediated response, investigated with a delayed skin multitest, showed a marked positivity for the tricophyton antigen. Systemic therapy with terbinafine (250 mg/day) led to complete clinical and mycological recovery of the atypical ringworm in 30 days. Multiforme lesions rapidly improved and completely disappeared in 7 days. CONCLUSIONS: ID reaction to a distant focus of dermatophyte infection very rarely presents as EM. The presented case fits all the essential criteria required for the diagnosis of an ID reaction. The atypical clinical presentation of the ringworm infection, with marked involvement of the intranasal hairs, seems to have induced an inflammatory allergic response, similar to that evoked by sycosis.

Adult↗
PubMed2003

Erythema annulare centrifugum-type psoriasis: a particular variant of acute-eruptive psoriasis.

A case of erythema annulare centrifugum-type psoriasis in a 58-year-old white woman with a history of chronic plaque psoriasis is described. Initial failure of antipsoriatic treatments and an untypical histology complicated the diagnosis. After several trials acitretin at a low maintenance dose combined with oral fish oil and topical calcitriol led to sustained long-term remission. The spectrum of clinical differentiation of this rare disease, the histological characteristics and its nosological classification are discussed. It is suggested that this dermatosis represents a variant of acute psoriasis, rather than a variant of pustular psoriasis.

Biopsy, Needle↗
PubMed1998

Erythema multiforme associated human autoantibodies against desmoplakin I and II: biochemical characterization and passive transfer studies into newborn mice.

The demonstration of circulating autoantibodies directed against the constitutive desmosomal plaque proteins desmoplakin (dp) I and II in mucocutaneous lesions in a subset of patients with erythema multiforme major, suggests that humoral immune mechanisms may play a role in the pathogenesis of this severe skin disease. In this study we identified a specific peptide sequence--YSYSYS--representing an antigenic binding site for the human autoantibodies. This epitope is localized at the extreme carboxy terminal domain of dp thought to be responsible for the assembly of keratin filaments with desmosomes. To test the possibility whether these antibodies may exert any pathologic effects in vivo, human autoantibodies were affinity purified on a corresponding synthetic peptide matrix and peptide-specific antibodies were raised in rabbits. After repeated subcutaneous injections into newborn mice, affinity-purified human autoantibodies and anti-peptide rabbit IgG were detected on desmosomal plaques of keratinocytes overlying the injection site. Histologic and electron microscopic examinations showed hydropic degeneration of basal and suprabasal keratinocytes, dyskeratosis, signs of suprabasal acantholysis, and keratin filaments detached from the desmosomal plaques clumping around the nucleus. We demonstrate that autoantibodies are directed to an epitope within a dp domain crucial for the interaction of keratin filaments with desmosomes, and, when injected subcutaneously into newborn mice, produce pathologic changes. These findings imply that autoantibodies to dp could impair the function of desmosome-keratin filament complexes suggesting a pathogenic role in vivo.

Animals↗
PubMed2003

Prevention of immunosuppression by sunscreens in humans is unrelated to protection from erythema and dependent on protection from ultraviolet a in the face of constant ultraviolet B protection.

Sunscreens have been advocated as an important means of preventing skin cancer. Ultraviolet radiation induced immunosuppression is recognized as an important event in skin cancer development, yet the effectiveness of sunscreens in protecting the human immune system from ultraviolet radiation (i.e. ultraviolet radiation) is still unclear. The only currently accepted method of sunscreen rating is the sun protection factor system based on the prevention of erythema. We determined immune protection factors for six commercially available sunscreens using a nickel contact hypersensitivity model in humans. Both sun protection factor and immune protection factor testing was performed using the same solar simulated ultraviolet radiation source and dose-responses were used to determine endpoints both with and without sunscreens. We found that the immune protection factor did not correlate with the sun protection factor; however, immune protection factor was significantly correlated to the ultraviolet A protective capability of the sunscreens, indicating that sunscreen protection from ultraviolet A is important for the prevention of ultraviolet immunosuppression, when there is constant ultraviolet B protection. We recommend that sunscreens should be rated against their immune protective capability to provide a better indication of their ability to protect against skin cancer.

Adolescent↗
PubMedDate not supplied

Kikuchi disease with facial rash and erythema multiforme.

Kikuchi disease, or histiocytic necrotizing lymphadenitis, is a benign illness characterized by fever and cervical lymphadenopathy predominantly in young women. It has a self-limiting course. Skin changes occur in about one-third of patients and are of a nonspecific nature. A case of Kikuchi disease with cutaneous involvement in the guise of erythema multiforme and a facial rash is reported. The differential diagnosis with systemic lupus erythematosus and Rowell syndrome is discussed.

Biopsy↗
PubMedDate not supplied

Erythrokeratodermia variabilis with erythema gyratum repens-like lesions.

A large pedigree with erythrokeratodermia variabilis (EKV) and erythema gyratum repens-like lesions is described. Clinical, laboratory, and histologic findings of this family are presented. The differential diagnoses of the following dermatoses with an erythematous and a hyperkeratotic component are discussed: erythrokeratodermia variabilis (Mendes da Costa), progressive symmetric erythrokeratoderma (Gottron), loricrin keratoderma, erythrokeratoderma en cocardes (Degos), Netherton syndrome, keratitis-ichthyosis-deafness (KID) syndrome, erythrokeratolysis hiemalis (Oudtshoorn disease), and nonbullous congenital ichthyosiform erythroderma.

Adult↗
PubMedDate not supplied

AQP1 and AQP3, psoriasin, and nitric oxide synthases 1-3 are inflammatory mediators in erythema toxicum neonatorum.

Erythema toxicum neonatorum is a common, inflammatory skin reaction in healthy newborn infants characterized by an accumulation of activated immune cells in the lesions. Its etiology and physiologic significance are still unclear. The purpose of this study was to extend the search for possible inflammatory mediators of the rash. We performed immunohistochemistry on punch biopsy cryosections from lesions of four, 1-day-old infants and from four matched controls without rash, using antibodies against the water channel proteins aquaporin-1 (AQP1) and aquaporin-3 (AQP3), psoriasin, and the nitric oxide synthase (NOS) enzymes, neuronal NOS (nNOS), inducible NOS (iNOS), and endothelial NOS (eNOS). All sections from the lesions showed a dense, nodular cellular infiltrate located near the hair follicle. The vessels in the dermis showed a high incidence of AQP1 and eNOS. Strong staining for AQP1, AQP3, and psoriasin, as well as nNOS, iNOS, and eNOS were seen in the entire epidermal layer. The infiltrate in the dermis contained numerous cells expressing AQP1, AQP3, nNOS, iNOS, and eNOS. Double immunofluorescence staining showed that AQP3 was located in CD1a-expressing Langerhans cells and other dendritic cells in the dermis, as well as in CD14-expressing macrophages, CD15-expressing neutrophils, and EG2-expressing eosinophils surrounding the hair follicle. Our findings show that AQP1 and AQP3, psoriasin, and NOSs are involved in the activation of the skin immune system at birth.

Aquaporin 1↗
PubMedDate not supplied

Erythema dyschromicum perstans in prepubertal children.

Erythema dyschromicum perstans (EDP) is a rare disorder of pigmentation that is most common in Hispanic patients. In adults, EDP has a slow onset and is unlikely to resolve spontaneously. The etiology and clinical course in children is poorly defined. Physical examinations, chart reviews, and telephone interviews were performed for eight pediatric patients with EDP who were followed at Children's Memorial Hospital in Chicago between 1990 and 1998. All the patients available for long-term follow-up (five of the eight) experienced complete clearance without recurrence in an average of 2.5 years. In all of our patients, the onset was noted from July to December. The administration of aminopenicillins was coincident with the development of EDP in two of the patients. Review of the English-language literature reveals that 25 prepubertal children have previously been reported. Including our patients, 69% of prepubertal children with EDP experienced resolution. We concluded that the clinical course of childhood (prepubertal) EDP differs from that of adult EDP, and it is more likely to resolve within 2-3 years.

Age of Onset↗
PubMed2003

Extracorporeal monocyte granulocytapheresis was effective for a patient of erythema nodosum concomitant with ulcerative colitis.

We report an erythema nodosum (EN) patient whose condition became apparent during the clinical course of ulcerative colitis (UC). The patient relapsed frequently in spite of taking a high dose adrenocortical steroid during his morbidity period of UC. Monocyte-granulocytapheresis (M-GCAP) was combined with 5-aminosalicylic acid 2250 mg/day peroral and once a day of steroid enema. Monocyte-granulocytapheresis was performed once a week for 5 weeks, and succeeded in inducing clinical remission for both UC and EN. The immunological and clinical connections between UC and EN have never been fully elucidated. In this case, because the symptoms of UC and EN revealed parallel improvement after his inflammatory reaction had been brought under control by combining M-GCAP therapy, we hypothesize that the onset of EN appeared as a result of the patient's long-term, treatment-resistant immuno-disturbance, which first appeared as symptoms of UC. Immunomodulative effects induced by M-GCAP might help to control other chronic non-specific inflammations not concerned with targeted organ(s).

Adult↗
PubMedDate not supplied

Erythema multiforme following the infusion of hydroxyethyl starch.

Approximately 18 hours after attempted leukocytapheresis, erythema multiforme developed in a normal 28-year-old woman. About 50 milliliters of 6 percent hydroxyethyl starch (HES) had been infused before the procedure was stopped because of difficulty with access to the veins of her other arm. Although she had donated platelets several times, this was her first leukocytapheresis and first exposure to HES. Preapheresis steroids had not been given.

Adult↗
PubMed1998

POEMS syndrome, steroid-dependent diabetes mellitus, erythema elevatum diutinum, and rheumatoid arthritis as extramedullary manifestations of plasma cell dyscrasia.

POEMS syndrome is a rare synopsis of different multisystemic disorders (polyneuropathy, organomegaly, endocrinopathy, monoclonal gammapathy, and skin lesions) associated with plasma cell dyscrasia. We herein report the atypical case of a 44-year-old white man presenting with glomerulopathy, POEMS syndrome, and erythema elevatum diutinum with a few-year history of non-insulin-dependent diabetes mellitus (NIDDM) and seronegative rheumatoid arthritis (RA) as early manifestations of IgAlambda multiple myeloma. The prescription of 1 mg/kg/day prednisone improved the patient's features dramatically. Skin lesions improved by the association of glucocorticoids and plasma exchange, recurred when plasmapheresis ceased, and remitted when plasma exchange was reintroduced. NIDDM requiring insulinotherapy recurred when corticoids were discontinued and remitted when prednisone was reintroduced. However, prednisone and plasmapheresis had no effect on polyneuropathy, M-paraprotein, and plasma cell dyscrasia in our patient, who developed indolent multiple myeloma a few years later. We thus concluded that POEMS syndrome, steroid-dependent diabetes mellitus, rheumatoid arthritis, RA, and skin vasculitis in our patient were triggered by plasma cell dyscrasia.

Adult↗
PubMed2002

Uveitis and erythema nodosum in inflammatory bowel disease: clinical features and the role of HLA genes.

BACKGROUND & AIMS: There are few systematic studies on the natural history or immunogenetic associations of erythema nodosum (EN) or ocular inflammation in inflammatory bowel disease (IBD), but they are reportedly more common in patients with other extraintestinal manifestations (EIMs), particularly arthritis. Immunogenetic associations have previously been described in IBD arthritis and in EN associated with sarcoidosis. This study examined the clinical features and HLA-B, DR, and tumor necrosis factor alpha (TNF-alpha) associations of ocular inflammation and EN and their clinical and immunogenetic relationship to arthritis in IBD. METHODS: Details of EN and ocular inflammation were gathered by case-note review and questionnaire in 976 ulcerative colitis patients and 483 Crohn's patients. Sequence-specific PCR typing for polymorphisms in HLA-B, DR, and TNF-alpha was performed in 39 EN and 40 ocular patients. Results were compared with 490 IBD controls without EIMs, 38 patients with type 1 and 31 with type 2 peripheral arthritis, and 16 AS patients. RESULTS: EN and ocular inflammation were more common in women, were associated with IBD relapse, and recurred in approximately 30% of patients. They occurred more commonly with arthritis and AS than expected by chance. Ocular inflammation was strongly associated with HLA-B*27, B*58, and HLA-DRB1*0103. There is a weak association between EN and HLA-B*15 but a strong association with the -1031 TNF-alpha. CONCLUSIONS: EN, uveitis, and arthritis associated with IBD occur together commonly. They are associated with genes in the HLA region, and linkage disequilibrium between these genes may account for the clinical picture of overlapping but independent clinical manifestations.

Erythema Nodosum↗
PubMed2003

[Exudative erythema multiforme with transition to a toxic epidermal necrolysis after taking aceclofenac (Beofenac)].

HISTORY AND CLINICAL FINDINGS: A 75-year-old woman took aceclofenac (100 mg/day) for 15 days for long-standing arthritis. Starting with erythema of the face she developed multiple target lesions with central bullae on her neck, chest, back, including the palmoplantar region. In several areas the lesions became confluent and showed wide detachment. The mucous membranes of the eyes, mouth and genitalia also showed marked inflammation, most prominent were large erosions in the mouth. The clinical course culminated in an overall skin loss of about 15 % of the total body surface area. DIAGNOSIS, TREATMENT AND COURSE: Treatment with topical and systemic corticosteroids as well as antiseptics resulted in complete clearing of the cutaneous lesions within 4 weeks. Various analgetics, particularly the closely related diclofenac, were tolerated without any side-effects after oral intake. Re-exposition with aceclofenac was rejected due to safety reasons. CONCLUSION: This is the first report of a potentially life-threatening adverse cutaneous drug reaction after taking with aceclofenac.

Aged↗
PubMed2004

[Erythema multiforme caused by carbamazepine].

The antiepileptic carbamazepine can reduce aggressive behaviour and can be used as a mood stabilizer. We present a case report of a patient suffering from borderline personality disorder who was treated with carbamazepine and developed erythema multiforme.

Anticonvulsants↗
PubMed1985

[Meningopolyneuritis (Garin-Bujadoux, Bannwarth) erythema chronicum migrans disease of the nervous system transmitted by ticks].

The clinical spectrum of this remarkable non-syphilitic spirochaetosis (spirochaeta infection) of the nervous system is described in light of 53 further cases with reference to our earlier description of 47 cases. As the etiological diagnosis is not possible in all cases the knowledge of clinical courses is especially important. Males are affected almost twice as often as women. The disease is especially prevalent in the sixth decade. Usually, the syndrome begins in the warm season with exquisite pain and other sensory irritations in combination with marked malaise. Initial signs during the winter as well as painless clinical courses were also noted. About one half of the patients remembered contact with arthropodes or a chronic migrating erythema. In most cases (95%), asymmetrical peripheral signs followed the painful stage. More than two thirds of the patients showed affection of cranial nerves, usually facial palsy. Twenty percent demonstrated paresis of the extremities alone, the combination of involvement of cranial nerves and extremities was noted in one third of the cases. The sensory signs were usually only mild. Occasionally, the neurological impairment was limited to isolated sensory impairment. In contrast to the closely related Lyme disease, central nervous affection as well as cardiac and arthritic involvement is rare: 34 percent demonstrated mild psychopathological impairment; in one case a Babinski-phenomenon was elicited. Knee joint pain and gonarthritis were found in one case each. Even though meningeal signs were usually absent, the CSF showed alterations suggestive of chronic lymphocytic meningitis in all cases. At the same time, local synthesis of all three immune globulins was documented, especially of IgM (85%).

Adolescent↗
PubMed1985

[Progressive Borrelia encephalomyelitis. Chronic manifestation of erythema chronicum migrans disease of the nervous system].

A 54-year-old housewife became ill with transient diplopia followed by facial paralysis, tremor of the right hand and a progressively worsening disturbance in gait. Four years after the first appearance of the disease bilateral hypacusia, paralysis of the right hypoglossal nerve, dysarthria, severe spastic tetraparesis with ataxia, urinary retention and an organically induced character change were present. Lumbar puncture fluid contained 51/mm3 lymphocytes and 1164 mg/dl protein. Immunoglobulin A, G and M, synthesised locally, could be detected in cisternal fluid. The serum had a high antibody titre against Ixodes ricinus-Borrelia antigen and the same antibodies, formed locally, were present in the lumbar fluid. The neurological deficiencies improved markedly under parenteral penicillin treatment and the antibody titre decreased. The detection of Borrelian antibodies, especially those synthesised locally in the CSF, enabled an aetiological diagnosis to be made in seven other patients aged 18-47 years in whom the disease had been present for 1 to 8 years. These patients likewise presented symptoms characteristic of a disseminated disease of the CNS, with spastic pareses and evidence in the spinal fluid of chronic inflammation and disruption of CSF barriers in particular. This progressive encephalomyelitis differs from the common and spontaneously healing meningo-polyneuritis (Garin-Bujadoux-Bannworth), the usual manifestation of erythema chronicum migrans of the nervous system, in its progressive nature, its invasion of the CNS and the possible long lasting severe damage when not specifically treated.

Adolescent↗