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

  title={Retinoschisin gene therapy and natural history in the Rs1h-KO mouse: long-term rescue from retinal degeneration.},
  author={S. Kjellstrom and R. Bush and Yong Zeng and Y. Takada and P. Sieving},
  journal={Investigative ophthalmology & visual science},
  volume={48 8},
  • S. Kjellstrom, R. Bush, +2 authors P. Sieving
  • Published 2007
  • Biology, Medicine
  • Investigative ophthalmology & visual science
  • 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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