Restoration of hemoglobin A synthesis in erythroid cells from peripheral blood of thalassemic patients

G Lacerra, H Sierakowska, C Carestia… - Proceedings of the …, 2000 - National Acad Sciences
G Lacerra, H Sierakowska, C Carestia, S Fucharoen, J Summerton, D Weller, R Kole
Proceedings of the National Academy of Sciences, 2000National Acad Sciences
Mononuclear cells from peripheral blood of thalassemic patients were treated with
morpholino oligonucleotides antisense to aberrant splice sites in mutant β-globin precursor
mRNAs (pre-mRNAs). The oligonucleotides restored correct splicing and translation of β-
globin mRNA, increasing the hemoglobin (Hb) A synthesis in erythroid cells from patients
with IVS2–654/βE, IVS2–745/IVS2–745, and IVS2–745/IVS2–1 genotypes. The maximal Hb
A level for repaired IVS2–745 mutation was≈ 30% of normal; Hb A was still detectable 9 …
Mononuclear cells from peripheral blood of thalassemic patients were treated with morpholino oligonucleotides antisense to aberrant splice sites in mutant β-globin precursor mRNAs (pre-mRNAs). The oligonucleotides restored correct splicing and translation of β-globin mRNA, increasing the hemoglobin (Hb) A synthesis in erythroid cells from patients with IVS2–654/βE, IVS2–745/IVS2–745, and IVS2–745/IVS2–1 genotypes. The maximal Hb A level for repaired IVS2–745 mutation was ≈30% of normal; Hb A was still detectable 9 days after a single treatment with oligonucleotide. Thus, expression of defective β-globin genes was repaired and significant level of Hb A was restored in a cell population that would be targeted in clinical applications of this approach.
National Acad Sciences