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  • Title: Clinical and hematological response to hydroxyurea in a patient with Hb Lepore/beta-thalassemia.
    Author: Rigano P, Manfré L, La Galla R, Renda D, Renda MC, Calabrese A, Calzolari R, Maggio A.
    Journal: Hemoglobin; 1997 May; 21(3):219-26. PubMed ID: 9140718.
    Abstract:
    The possibility of increasing Hb F in vivo using drugs like 5-azacytidine, hydroxyurea, and butyrate has been established. However, in many cases this does not entail an increase in total hemoglobin. We report on a patient with Hb Lepore/beta-thalassemia being treated with hydroxyurea (30 mg/Kg/day) because of the presence of erythroid extramedullary masses with severe neurological abnormalities. During therapy the patient showed a remarkable improvement in neurological signs due to the reduction in extra-medullary masses, a significant increase in both total hemoglobin (from 5.8 to 9.7 g/dl) and Hb F (from 4.9 g/dl to 9.1 g/dl). The marked improvement in hemoglobin level in our patient with Hb Lepore/beta-thalassemia suggests gamma-globin gene activation due to the DNA structure determined by the crossover event.
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