Progressive systemic sclerosis with pancytopenia, polymyositis and complete heart block developing after D-penicillamine therapy: A case report.

A 55-year-old woman came to our hospital because of cutaneous sclerosis of the limbs in September 1996, and was diagnosed with scleroderma based on a skin biopsy. In August 1997, the cutaneous sclerosis became progressive (hemoglobin level, 4.3 g/dl; platelet count, 7 × 10⁹/l). The laboratory results were positive for the direct Coombs test, bone marrow aspiration showed a dry tap, and the bone marrow biopsy showed marked fatty marrow. Indium-111 bone marrow scintigraphy showed a markedly decreasing uptake. These findings indicated bone marrow hypoplasia associated with hemolytic anemia. After prednisolone therapy (60 mg) was initiated, the direct Coombs test became negative but the blood cell count did not increase. Then, 300 mg of cyclosporin was initiated and anemia and thrombocytopenia improved. The cyclosporin dosage was gradually decreased and the patient’s hematological condition was good, although the cutaneous sclerosis changed only a little. This is a rare and interesting case of a patient with scleroderma associated with bone marrow insufficiency and hemolysis who responded well to cyclosporin.

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Progressive systemic sclerosis with pancytopenia, polymyositis and complete heart block developing after D-penicillamine therapy: A case report.

Cited by 2 publications

References 11 publications

Postural changes, dysphagia, and systemic sclerosis

Postural changes, dysphagia, and systemic sclerosis

Scleroderma Associated with Anemia and Thrombocytopenia that Responded Well to Cyclosporin

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