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PMID: 7524085 Published · ppublish English Comparative Study Journal Article Research Support, U.S. Gov't, P.H.S.

Recombinant adeno-associated virus (rAAV)-mediated expression of a human gamma-globin gene in human progenitor-derived erythroid cells.

Miller JL, Donahue RE, Sellers SE, Samulski RJ, Young NS, Nienhuis AW

Abstract

Effective gene therapy for the severe hemoglobin (Hb) disorders, sickle-cell anemia and thalassemia, will require an efficient method to transfer, integrate, and express a globin gene in primary erythroid cells. To evaluate recombinant adeno-associated virus (rAAV) for this purpose, we constructed a rAAV vector encoding a human gamma-globin gene (pJM24/vHS432A gamma). Its 4725-nucleotide genome consists of two 180-bp AAV inverted terminal repeats flanking the core elements of hypersensitive sites 2, 3, and 4 from the locus control region of the beta-globin gene cluster, linked to a mutationally marked A gamma-globin gene (A gamma) containing native promoter and RNA processing signals. CD34+ human hematopoietic cells were exposed to rAAV particles at a multiplicity of infection of 500-1000 and cultured in semisolid medium containing several cytokines. A reverse transcriptase polymerase chain reaction assay distinguished mRNA signals derived from transduced and endogenous human gamma-globin genes. Twenty to 40% of human erythroid burst-forming unit-derived colonies expressed the rAAV-transduced A gamma-globin gene at levels 4-71% that of the endogenous gamma-globin genes. The HbF content of pooled control colonies was 26%, whereas HbF was 40% of the total in pooled colonies derived from rAAV transduced progenitors. These data establish that rAAV containing elements from the locus control region linked to a gamma-globin gene are capable of transferring and expressing that gene in primary human hematopoietic cells resulting in a substantial increase in HbF content.

MeSH Terms
Antigens, CD/analysis Antigens, CD34 Base Sequence Cell Line Cloning, Molecular DNA Primers Dependovirus/genetics Gene Expression Genetic Therapy/methods Genetic Vectors Globins/biosynthesis Hematopoietic Stem Cells/metabolism Humans Introns Molecular Sequence Data Plasmids Polymerase Chain Reaction Recombination, Genetic Restriction Mapping Transduction, Genetic Transfection
Chemicals
Antigens, CD Antigens, CD34 DNA Primers Globins
Authors & Affiliations
6 authors, click to expand affiliations / ORCID
Miller J L
National Heart, Lung and Blood Institute, Bethesda, MD 20892.
Donahue R E
Sellers S E
Samulski R J
Young N S
Nienhuis A W
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Article Info
Journal
Proceedings of the National Academy of Sciences of the United States of America
Abbr.
Proc Natl Acad Sci U S A
ISSN
0027-8424
Published
1994-10-11
Pages
10183-7
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC44982
Subset
IM
Grants
NHLBI NIH HHS · HL 48347-03 · United States
Corrections
ErratumIn
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