Cases reported "Scleroderma, Localized"

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1/10. Grzybowski's generalized eruptive keratocanthomas: a case report.

    A 47-year old woman with Grzybowski's generalised eruptive keratoacanthomas is described. There was no history of skin disease in her family, except for an uncle's basal cell carcinoma. From 1995 she developed multiple lesions of various size, ranging from hundreds of small follicular lesions to large typical keratoacanthomas up to 5 cm in diameter, scleroderma-like facial skin and marked ectropion. Histological examination of small and large skin lesions was typical of keratoacanthoma, and no human papillomavirus was detected by polymerase chain reaction. Oral treatment with acitretin had no effect. Both cyclophosphamide and methotrexate therapy were refused by the patient despite the progressive course of the disease. Blepharoplastic surgery had some effect on eye symptoms. The etiology of this rare disease is unknown, but is probably related to some genetic defect.
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2/10. Bullous lesions in scleroderma.

    BACKGROUND: The occurrence of bullous lesions in localized or systemic scleroderma is rare. Three histologic patterns have been reported: lichen sclerosus et atrophicus-like, lymphangiectatic blisters and autoimmune blistering diseases. OBJECTIVE: To investigate the frequency, clinical, and immunopathologic features of patients with scleroderma and bullous eruptions and to review the literature regarding this rare condition. methods: A retrospective study of 53 cases of scleroderma (localized, generalized, and systemic) in the dermatology and rheumatology clinics at one institution over an 8-year span. Clinical, serologic, and immunopathologic findings were analyzed in four cases. RESULTS: Four of 53 patients exhibited bullous lesions in association with scleroderma. The first case illustrates lymphangioma-like clinical and pathologic presentation. The second case demonstrates bullous lichen sclerosus et atrophicus-like pattern. The other two cases exemplify a superimposed autoimmune skin disease, epidermolysis bullosa acquisita and penicillamine induced pemphigus foliaceus after treatment for systemic scleroderma. CONCLUSIONS: Of the 53 original patients, we have described four cases of bullous scleroderma (7.5%) Illustrating several pathogenetic mechanisms of bulla formation. inflammatory (lichen sclerosus et atrophicus), fibrotic/obstructive (lymphangiomatous), autoimmune (epidermolysis bullosa acquisita), and pemphigus foliaceus. The final case illustrates bullae as a complication of therapy for the underlying scleroderma.
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3/10. 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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4/10. Linear scleroderma en coup de sabre with associated neurologic abnormalities.

    Linear scleroderma represents a unique form of localized scleroderma that primarily affects the pediatric population, with 67% of patients diagnosed before 18 years of age. When linear scleroderma occurs on the head, it is referred to as linear scleroderma en coup de sabre, given the resemblance of the skin lesions to the stroke of a sabre. Here we describe 3 pediatric patients with linear scleroderma en coup de sabre who presented with neurologic abnormalities before or concurrent with the diagnosis of their skin disease. Our patients' cases highlight the underrecognized relationship between neurologic complications and linear scleroderma en coup de sabre and illustrate the importance of a thorough skin examination in patients with unexplained neurologic disease.
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5/10. Localized morphoea associated with oral submucous fibrosis.

    Localized morphoea is a disease characterized by localized sclerosis of the skin. The disease has been associated with several skin diseases. The case documented here is the first of its kind in which localized plaque type morphoea was seen in a patient with long standing oral submucous fibrosis.
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6/10. 20-MHz B-mode ultrasound in monitoring the course of localized scleroderma (morphea).

    Ultrasonographic methods have recently provided us with the means for objective and non-invasive monitoring of the dynamics of chronic skin diseases. We examined 34 patients with localized scleroderma (morphea) using a 20-MHz B-mode ultrasound scanner (DUB 20, Taberna pro Medicum, Luneburg). In patients with plaque-type and linear band-type localized scleroderma intraindividual comparison of sclerotic skin with corresponding areas of healthy skin showed thickening of the corium. The increase in corium thickness was between 2% and 251%. The extent of the difference in corium thickness between sclerotic and healthy skin depended on the location-originally thin skin showed a greater degree of sclerosis. We also frequently found enhanced reflexes in the lower corium and hyperechoic, widened bands of connective tissue traversing the subcutaneous fatty tissue from the corium-subcutis border in the direction of the muscle fascia. 20 patients were examined several times in the course of one year. In nine patients we found ultrasonographic evidence of regression (decrease in thickness 26%) and in nine the ultrasound examination showed progression (increase in thickness 28%). 20-MHz B-mode ultrasound imaging is a suitable non-invasive method for monitoring the course and treatment of localized scleroderma. Its routine use is strongly recommended.
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7/10. antibodies to the borrelia burgdorferi flagellum in patients with scleroderma, granuloma annulare and porphyria cutanea tarda.

    It is generally accepted that cutaneous Lyme borreliosis comprises erythema chronicum migrans, lymphadenosis benigna cutis, and acrodermatitis chronica atrophicans. In recent years the tick-borne spirochete borrelia burgdorferi has been associated with a number of other cutaneous disorders. We therefore investigated sera from 175 patients with localized scleroderma (morphea) (n = 64), systemic sclerosis (n = 74), granuloma annulare (n = 16) and porphyria cutanea tarda (n = 21) with the new, highly sensitive and specific borrelia burgdorferi flagellum ELISA assay. As controls (n = 297) served normal healthy volunteers and patients with other skin diseases. It was found that the distribution of individual antibody values and the median antibody levels were identical in controls and in patients with scleroderma, granuloma annulare and porphyria cutanea tarda. These data do not support the hypothesis of an etiological association between borrelia burgdorferi infection and scleroderma, granuloma annulare or porphyria cutanea tarda.
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8/10. Hypodermitis sclerodermiformis. Successful treatment with ultrasound.

    We report herein the successful use of ultrasound therapy in the treatment of hypodermitis sclerodermiformis (HS). It is a skin disease characterized by well-circumscribed, chronic, painful and tender, single or multiple, board-like, indurated, sharply bordered lesions, occurring on the legs of patients with venous insufficiency. Hypodermitis sclerodermiformis is a little-known clinical entity, often undiagnosed or misdiagnosed as phlebitis, cellulitis, or stasis dermatitis. It merits clinical recognition because of the gratifying and unique response it has to treatment with ultrasound therapy.
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9/10. Concurrent localized scleroderma and discoid lupus erythematosus. Cutaneous 'mixed' or 'overlap' syndrome.

    Four patients with concurrent, chronic, progessive, localized scleroderma and discoid lupus erythematosus were studied; the condition originated as linear scleroderma in three of them. Three of the four patients were young females at the onset of the first skin disease. Dermatopathologic study confirmed the scleroderma and lupus erythematosus (LE). Direct immunofluorescence showed a positive band test in three cases. Unusual serological results included a positive LE clot test in three cases, a positive extractable nuclear antigen test in one case, and a negative antinuclear antibody test on repeated occasions in all four cases. Rare cutaneous disease similar to systemic, "mixed," or "overlap" connective tissue disease exists and offers an opportunity to study unusual immunologic and pathological events in both scleroderma and LE.
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10/10. Coexistence of vitiligo and morphea: a case report and review of the literature.

    A patient with both vitiligo and morphea is described. These skin diseases rarely appear concurrently in the same patient, despite the fact that both are thought to an autoimmune etiology. Similar cases that have been reported in the literature are reviewed and discussed.
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