Ciliary Beating Recovery in Deficient Human Airway Epithelial Cells after Lentivirus Ex Vivo Gene Therapy
2009

Gene Therapy for Ciliary Beating Recovery in Airway Cells

Sample size: 1 publication 10 minutes Evidence: moderate

Author Information

Author(s): Chhin Brigitte, Negre Didier, Merrot Olivier, Pham Jacqueline, Tourneur Yves, Ressnikoff Denis, Jaspers Martine, Jorissen Mark, Cosset François-Loïc, Bouvagnet Patrice

Primary Institution: Université de Lyon, Lyon, France

Hypothesis

Gene therapy could restore ciliary function in DNAI1-mutated airway epithelial cells to prevent patients from infectious complications.

Conclusion

The study demonstrated that gene therapy can restore ciliary beating in DNAI1-deficient human airway epithelial cells.

Supporting Evidence

  • Transduction of DNAI1-mutated HAECs with wild-type DNAI1 can restore ciliary beating.
  • Normal ciliary beating was recorded in treated cells, while untreated cells remained immotile.
  • Electron microscopy showed the presence of outer dynein arms in treated cells.

Takeaway

Scientists used a special virus to fix the tiny hairs in the lungs of a sick person, helping them to work properly again.

Methodology

Lentiviral vectors were used to transduce DNAI1-deficient human airway epithelial cells, and ciliary beating was assessed post-treatment.

Limitations

The study was conducted on a single patient, and the transduction efficiency was not optimal.

Participant Demographics

A male patient with Primary Ciliary Dyskinesia and compound heterozygous mutations in the DNAI1 gene.

Statistical Information

P-Value

p<0.0001

Statistical Significance

p<0.0001

Digital Object Identifier (DOI)

10.1371/journal.pgen.1000422

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