Adenoviral-mediated correction of methylmalonyl-CoA mutase deficiency in murine fibroblasts and human hepatocytes
2007

Gene Therapy for Methylmalonic Acidemia

publication Evidence: high

Author Information

Author(s): Chandler Randy J, Tsai Matthew S, Dorko Kenneth, Sloan Jennifer, Korson Mark, Freeman Richard, Strom Stephen, Venditti Charles P

Primary Institution: National Human Genome Research Institute, National Institutes of Health

Hypothesis

Can adenoviral-mediated gene delivery correct methylmalonyl-CoA mutase deficiency in human and murine cells?

Conclusion

Adenoviral-mediated gene delivery can effectively correct the enzymatic defect in methylmalonic acidemia in both murine models and human patients.

Supporting Evidence

  • The adenoviral vector restored propionate metabolism in both murine and human cells.
  • Infected human hepatocytes showed a significant increase in enzyme expression.
  • The study demonstrated the potential for hepatocyte-directed gene therapy in treating methylmalonic acidemia.

Takeaway

Scientists used a virus to fix a problem in cells from people with a rare disease, showing it could help treat them in the future.

Methodology

Adenoviral vectors were used to deliver the methylmalonyl-CoA mutase gene to murine fibroblasts and human hepatocytes, followed by enzymatic and expression studies to assess correction.

Limitations

The study primarily used murine models and human cells from a single patient, which may limit generalizability.

Participant Demographics

Human hepatocytes were derived from a 5-year-old boy with methylmalonic acidemia.

Digital Object Identifier (DOI)

10.1186/1471-2350-8-24

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