Cases reported "Scleroderma, Localized"

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1/425. Repair of a large "coup de sabre" with soft-tissue expansion and artificial bone graft.

    The authors present a case of a "coup de sabre"--a linear form of scleroderma--in an 18-year-old woman treated by means of an expanded forehead and scalp flap, a hydroxyapatite implant, and an autologous iliac bone graft. Hydroxyapatite was implanted to augment a depressive bony deformity. The nasal deformity was repaired with an expanded forehead flap and an autologous iliac bone graft. This is the first reported case in which a deformity of scleroderma was treated with a combination of an expanded skin flap and a hydroxyapatite implant. ( info)

2/425. "Centripetal flagellate erythema": a cutaneous manifestation associated with dermatomyositis.

    We describe 3 patients with dermatomyositis who presented with flagellate erythema. This cutaneous eruption is characterized by erythematous linear lesions on the trunk and proximal extremities. Histologic examination of this eruption in one of our cases revealed an interface dermatitis. review of the literature and records of 183 patients with connective tissue diseases from our institution has shown that this peculiar eruption has been reported only in dermatomyositis. Because of the location of this eruption, we encourage the use of the term "centripetal flagellate erythema" to distinguish this entity from other linear eruptions seen in patients with connective tissue diseases. ( info)

3/425. dermatofibrosarcoma protuberans: an early non-protuberant phase of the tumour.

    A 40-year-old female patient presented with a flat scar like plaque on the upper chest. The patient's perception of subtle change in the lesion was of paramount importance in the decision to biopsy this lesion of innocuous clinical appearance. Histopathological findings were those of dermatofibrosarcoma protuberans. This case illustrates the early clinical features of the tumour and a recommendation for taking a biopsy of any scar-like plaque where there is no clear history of preceding trauma. ( info)

4/425. Localized scleroderma associated with progressing ischemic stroke.

    We present a 73 year-old Japanese woman with localized scleroderma involving the right side of the scalp accompanied by continuous tingling pain, who developed insidiously progressive left hemiparesis. In magnetic resonance imaging of the brain, an infarct first appeared in the watershed region of the right middle cerebral artery territory and subsequently extended to deep white matter accompanied by scattered hemorrhages. Focal stenosis in the M2 portion of the right middle cerebral artery was revealed on magnetic resonance angiography, and the distal vessels were only shown faintly. A biopsy specimen from the sclerotic scalp lesion showed obvious thickening of vessel walls and mild mononuclear cell infiltration. We believe that the progressing ischemic stroke was caused by hemodynamic disturbances from localized sclerotic obstruction of the middle cerebral artery, with an autoimmune pathogenesis. ( info)

5/425. Coexisting morphoea and granuloma annulare-are the conditions related?

    granuloma annulare and localized morphoea are both well described in the dermatological literature. We now present two patients with both of these diseases, a comorbidity rarely described. A possible pathogenic relationship is discussed. ( info)

6/425. Progressive facial hemiatrophy (Parry-Romberg syndrome).

    Three cases of progressive facial hemiatrophy are presented. In all three cases there is evidence of localised scleroderma or morphea in association with the facial hemiatrophy. This would seem to support the contention that the two disorders are closely related. In two cases, ocular complications are prominent and in one Raynaud's phenomena provide clear asymmetrical dilatation of the lateral ventricle suggest that there may be a central rather than a peripheral cause for the sympathetic overactivity. ( info)

7/425. pulmonary edema during acute infusion of epoprostenol in a patient with pulmonary hypertension and limited scleroderma.

    epoprostenol (prostacyclin) is currently approved for treatment of primary pulmonary hypertension; however, it is being evaluated in other forms of pulmonary hypertension, particularly scleroderma. Side effects associated with this medication are usually minor; serious complications are most often due to the delivery system required for continuous infusion. We describe a life threatening side effect of acute epoprostenol infusion (pulmonary edema) in a patient with pulmonary hypertension associated with limited scleroderma and discuss its management and potential etiology. This is the first case where epoprostenol has been successfully reinstituted. ( info)

8/425. radiation-induced localized scleroderma in breast cancer patients.

    radiation-induced scleroderma in breast cancer patients appears to occur in approximately one out of every 500 patients. We report four cases that developed within 3 months of conservative breast surgery and postoperative radiation treatment. The reaction was contained entirely within the treatment field and demonstrated the typical features of this condition where the breast becomes erythematous, violaceous, indurated, retracted, and progressively pigmented. The breast tends to soften and become more comfortable over 1-4 years; however, significant induration, retraction and pigmentary changes remain. There appears to be no predictive factors. radiation-induced scleroderma must be differentiated from cellulitis and recurrent breast cancer. ( info)

9/425. Isolation of borrelia afzelii from circumscribed scleroderma.

    A 45-year-old man presented with circumscribed scleroderma (CS) on the extremities. histology of lesional skin showed the typical manifestations of scleroderma including a perivascular and interstitial infiltrate of lymphocytes and plasma cells; in one of the biopsies spirochaetes could be detected. Despite treatment with penicillin, progression of CS was observed and spirochaetes were isolated from skin cultures obtained from active scleroderma lesions. These spirochaetes were identified as borrelia afzelii by sodium dodecyl sulphate-polyacrylamide gel electrophoresis of outer surface proteins and polymerase chain reaction (PCR) analysis of their chromosome. After two courses with ceftriaxone the lesions stopped expanding and sclerosis of the skin was diminished. At this time cultures for spirochaetes and PCR of lesional skin for borrelia afzelii dna remained negative. The pathogenetic role of borrelia afzelii in the development of CS is discussed. ( info)

10/425. Effect of penicillin g on corium thickness in linear morphea of childhood: An analysis using ultrasound technique.

    Linear morphea is the most common form of localized scleroderma in children. The pathogenesis of this disorder is unknown. We report a child with a 6-year history of linear morphea in whom intravenous administration of 5 MU aqueous penicillin g three times a day for 10 consecutive days caused reduction in the thickness of the corium as demonstrated by ultrasound measurements. ( info)
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