Cases reported "Erythema"

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1/24. Treatment of reticular erythematous mucinosis with a large dose of ultraviolet B radiation and steroid impregnated tape.

    To date, there are no successful treatments for reticular erythematous mucinosis (REM) other than antimalarial drugs such as chloroquine; these have many adverse side effects and are prohibited for use in japan. We report a case of REM improved by a large dose of ultraviolet B (UVB) radiation and a steroid impregnated tape. The large dose of UVB radiation improved the erythema after 5 weeks. Application of steroid impregnated tape to a lesion where a large dose of UVB had been given produced an additive clinical effect. UVB radiation and steroid impregnated tape are considered to be effective treatments for REM.
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2/24. erythema chronicum migrans.

    erythema chronicum migrans is characterized by an expanding erythematous band about an arthropod bite. While rare, it is an important disease to recognize because of its rapid response to even modest doses of antibiotics. This response and the association with arthropod bites suggest a microbial vector. Richettsial titers have been demonstrated in several cases but we were unable to show them in our patient, who we believe is the second reported case of erythema chronicum migrans incurred by an American without traveling abroad.
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3/24. Severe oral mucositis after therapeutic administration of [131I]MIBG in a child with neuroblastoma.

    OBJECTIVE: The purpose of this report is to document a newly encountered oral side effect of targeted radiotherapy with iodine 131-metaiodobenzylguanidine ([(131)I]MIBG) in the treatment of neuroblastoma. STUDY DESIGN: A 14-month-old girl was diagnosed with stage 4 neuroblastoma. After completion of chemotherapy, the tumor showed no signs of regression; treatment with 3700 MBq [(131)I]MIBG was therefore decided on, 8 months after diagnosis. RESULTS: Fourteen days after infusion of MIBG, severe oral mucositis was diagnosed, with a generalized erythema involving the mucous membranes of the hard and soft palate, buccal mucosa, and upper and lower lips. The gingiva exhibited a general linear erythema. CONCLUSIONS: Visualization of the salivary glands on [(123)I]MIBG images suggests that accumulation of radiolabeled MIBG in the salivary glands may be related to sympathetic innervation.
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4/24. Erythrokeratoderma variabilis without connexin 31 or connexin 30.3 gene mutation: immunohistological, ultrastructural and genetic studies.

    Erythrokeratoderma variabilis, characterized by migrating erythema and fixed keratotic plaques, is a rare congenital disorder which has recently been connected with connexin (Cx)30.3 or Cx31 gene mutations. We present a 9-month-old Japanese girl who exhibited the typical clinical features of the disease, but carried no Cx30.3 or Cx31 gene mutations. Histopathologically, regular acanthosis with hyperkeratosis and hypergranulosis was observed in her lesional skin. Upregulation of involucrin and loricrin expression, and a weak expression of Cx26 was immunohistochemically observed in the upper spinous and granular layers. Electron microscopy revealed no abnormality in the keratin filaments, cornified cell envelope or gap junctions. Direct sequencing revealed no pathogenetic mutations in the Cx26, Cx30.3, Cx31 or Cx31.1 genes in this patient. The results indicate that erythrokeratoderma variabilis is pathologically heterogeneous, and that abnormalities in keratinization other than Cx30.3 and 31 gene mutations may underlie some forms of this disease.
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5/24. Immunohistochemical investigations and introduction of new therapeutic strategies in scleromyxoedema: case report.

    BACKGROUND: Scleromyxoedema is a rare chronic skin disease of obscure origin, which may often be associated with severe internal co-morbidity. Even though different casuistic treatment modalities have been described, to date, curing still seems to be impossible. CASE PRESENTATION: We report a 44-year-old Caucasian female presenting with remarkable circumscribed, erythematous to skin-coloured, indurated skin eruptions at the forehead, arms, shoulders, legs and the gluteal region. Routine histology and alcian blue labelling confirmed a massive deposition of acid mucopolysaccharides. Immunohistochemical investigations revealed proliferating fibroblasts and a discrete lymphocytic infiltration as well as increased dermal expression of MIB-1 and anti-mastcell-tryptase cells. bone marrow biopsies confirmed a monoclonal gammopathy of undetermined significance without morphological characteristics of plasmocytoma; immunofixation unveiled the presence of IgG-kappa paraproteins. CONCLUSIONS: Taking all data into account, our patient exhibited a complex form of lichen mxyoedematosus, which could most likely be linked a variant of scleromyxoedema. Experimental treatment with methotrexate resulted in a stabilisation of clinical symptoms but no improvement after five months of therapy. A subsequent therapeutic attempt by the use of medium-dose ultraviolet A1 cold-light photomonotherapy led to a further stabilisation of clinical symptoms, but could not induce a sustained amelioration of skin condition.
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6/24. Hereditary mucoepithelial dysplasia: clinical, ultrastructural and genetic study of eight patients and literature review.

    BACKGROUND: Hereditary mucoepithelial dysplasia is a dominantly inherited disease, mainly characterized by chronic mucosal lesions associated with keratitis, non-scarring alopecia, keratosis pilaris and perineal intertrigo. Since the original report by Witkop, this condition has been considered to be a disorder of desmosome/gap junction formation, but there has been no ex vivo investigation of these components using genetic and immunolabelling techniques. OBJECTIVES: To perform light and immunoelectron microscopic studies, and partial genetic analysis on five patients in a family and three sporadic cases and to point out similarities of this rare disorder with chronic mucocutaneous candidiasis and other follicular keratosis syndromes, i.e. ichthyosis follicularis-alopecia-photophobia (IFAP), keratitis-ichthyosis-deafness (KID) and Siemens syndromes. methods: Biopsies from the involved oral mucosa and armpit skin of patient 1 were prepared for standard histopathology, electron microscopy and immunocytochemistry. Microsatellite genotyping was performed in three affected family members. Direct sequencing after polymerase chain reaction amplification of the entire coding region was performed. RESULTS: A 14-year-old male had recurrent keratitis, widespread keratosis pilaris, perineal intertrigo, hypotrichosis and oral mucosal involvement. A similar phenotype was noted in four members of his family and in three sporadic cases. Histological examination of oral mucosa and skin samples showed a psoriasiform pattern, dyskeratotic features and cytoplasmic vacuoles. Expression of connexins (Cx), desmosomal, adherens junction and cytoskeleton proteins (Cx 26, 32 and 43, desmogleins 1 and 2, plakoglobin, desmoplakins I-II, plakophilin 1, beta-catenin, E-cadherin, keratins, beta-tubulin, vimentin and actin) was normal. Ultrastructural studies showed a reduced number of desmosomes. Dyskeratotic cells exhibited internalized gap junctions, long filamentous inclusions reactive with antikeratin antibodies, and bundles of perinuclear fibres resembling clear tonofilaments. Genetic analysis in the studied family excluded the desmosomal cadherins in chromosome 18q12 as candidate genes. CONCLUSIONS: A diagnosis of hereditary mucoepithelial dysplasia should be strongly suggested by the triad of non-scarring alopecia, well-demarcated erythema of oral mucosa and psoriasiform perineal rash, after exclusion of the clinically related follicular keratosis syndromes. Defective expression of cytoskeleton elements and/or a modification of mechanisms regulating junction-cytoskeleton assembly may be primarily responsible for impaired epithelial cohesion.
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7/24. Acquired port-wine stain related to acoustic neuroma.

    Port-wine stains are frequently seen congenital vascular malformations consisting of ectatic dermal capillaries. Acquired port-wine stain that develops later in life is an uncommon vascular lesion that is morphologically identical to a congenital port-wine stain. In the majority of acquired port-wine stains, the aetiology is unknown, but trauma is an important causative factor. Other proposed aetiologies include chronic sun exposure, hormonal changes, frostbite injury, obstruction of the peritoneovenous shunt, herpes zoster infection, and cerebral arteriovenous malformation. Here we report the first case of a patient who had an acquired port-wine stain related to a solid brain tumour.
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8/24. Photodynamic therapy for the treatment of erythema, papules, pustules, and severe flushing consistent with rosacea.

    We report a case of a 45-year-old woman who presented with facial erythema, papules, pustules, and severe flushing consistent with rosacea. The patient had failed standard pharmacologic treatments. The patient's flushing was so severe that she had undergone an elective sympathectomy. She received 6 sessions of photodynamic therapy (PDT) with 5-aminolevulinic acid (ALA as Levulan Kerastick, Dusa Pharmaceuticals) given at 2-week intervals. Improvement was evident after the second treatment and was considered "excellent" after the sixth treatment. Improvement continued and no flares were observed 1 month after the final treatment.
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keywords = tick
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9/24. Erythema figuratum in septic babesiosis.

    babesiosis is a rare worldwide-distributed protozoal zoonosis caused by a haemoprotozoan of the genus Babesia, transmitted through bites of tick of the genus ixodes. The first demonstrated case of human babesiosis in the world was discovered in europe, in 1957. However, most of the cases were reported later in the north-east of the united states where babesia microti has been the cause of over 300 cases of human babesiosis since 1969. In europe, the most severe cases are observed in asplenic patients infected by a parasite of cattle, the Babesia divergens. Only two cases of babesiosis have been reported in spain. We present a case of erythema figuratum associated to septic babesiosis in a non-splenectomized man, which is currently the third case of babesiosis in spain.
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keywords = tick, bite
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10/24. Neurologic complications of erythema-migrans-disease in childhood--clinical aspects.

    Erythema-migrans-disease represents a tick-borne borreliosis with a high and widespread incidence according to first epidemiological surveys. The early symptom is a distinctive erythema migrans eventually followed by arthritis and cardiac involvement as well as neurologic complications in the form of lymphocytic meningoradiculitis Garin-Bujadoux-Bannwarth. In a one-year-period we observed 9 children who developed neurologic complications as the predominant feature of erythema-migrans-disease. Eight children suffered from a lymphocytic meningitis and/or a peripheral facial palsy and one child presented with a severe polyradiculoneuritis with complete transverse myelitis. In contrast to adults the clinical course of neurologic manifestations in children seems to be milder and shorter and not associated with the typical painful radiculitis. Acute Bell's palsy seems to be a rather common symptom, whereas other peripheral pareses of the mononeuritis multiplex type were not observed. With respect to the assumed frequency and because of the possibility of antibiotic therapy, erythema-migrans-disease should be ruled out in any case of so-called idiopathic facial palsy or aseptic meningitis, which are frequently encountered in children.
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