The efficacy of interferon-alpha (IFN) was reported in three patients with
congenital dyserythropoietic anaemia (CDA) type I. We describe two addition
al cases treated with IFN, which normalized the haemoglobin level in both p
atients with a dramatic decrease in the size of the spleen in one. Haemoglo
bin remained stable more than 6 months after discontinuation of treatment.
IFN induced more than 50% decrease in the number of BFU-E in both patients'
bone marrow cultures, suggesting an indirect effect of IFN on erythropoies
is in vivo. We conclude that a trial of IFN therapy should be considered in
patients with CDA type I.