| Literature DB >> 11861306 |
Melissa A Baxter1, Robert F Wynn, Jonathan A Deakin, Ilaria Bellantuono, Kirsten G Edington, Alan Cooper, Guy T N Besley, Heather J Church, J Ed Wraith, Trevor F Carr, Leslie J Fairbairn.
Abstract
We have investigated the utility of bone marrow-derived mesenchymal stem cells (MSCs) as targets for gene therapy of the autosomal recessive disorder mucopolysaccharidosis type IH (MPS-IH, Hurler syndrome). Cultures of MSCs were initially exposed to a green fluorescent protein-expressing retrovirus. Green fluorescent protein-positive cells maintained their proliferative and differentiation capacity. Next we used a vector encoding alpha-L-iduronidase (IDUA), the enzyme that is defective in MPS-IH. Following transduction, MPS-IH MSCs expressed high levels of IDUA and secreted supernormal levels of this enzyme into the extracellular medium. Exogenous IDUA expression led to a normalization of glycosaminoglycan storage in MPS-IH cells, as evidenced by a dramatic decrease in the amount of (35)SO(4) sequestered within the heparan sulfate and dermatan sulfate compartments of these cells. Finally, gene-modified MSCs were able to cross-correct the enzyme defect in untransduced MPS-IH fibroblasts via protein transfer.Entities:
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Year: 2002 PMID: 11861306 DOI: 10.1182/blood.v99.5.1857
Source DB: PubMed Journal: Blood ISSN: 0006-4971 Impact factor: 22.113