Retinoschisin gene therapy and natural history in the Rs1h-KO mouse: long-term rescue from retinal degeneration.

@article{Kjellstrom2007RetinoschisinGT,
  title={Retinoschisin gene therapy and natural history in the Rs1h-KO mouse: long-term rescue from retinal degeneration.},
  author={Sten Kjellstrom and Ronald A. Bush and Yong Zeng and Yuichiro Takada and Paul A. Sieving},
  journal={Investigative ophthalmology & visual science},
  year={2007},
  volume={48 8},
  pages={3837-45}
}
PURPOSE The authors characterized the natural history of a retinoschisin gene knockout (Rs1h-KO) mouse model and evaluated the long-term effects of retinal rescue after AAV(2/2)-CMV-Rs1h gene delivery. METHODS Full-field scotopic electroretinograms (ERGs) were recorded from 44 male hemizygous Rs1h-KO and 44 male wild-type (WT) C57BL/6J mice at six ages between 1 and 16 months. Retinal morphometry included outer segment layer (OSL) width, photoreceptor cell count, and grading of schisis cavity… CONTINUE READING

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