Literature DB >> 9701253

Ribozyme rescue of photoreceptor cells in a transgenic rat model of autosomal dominant retinitis pigmentosa.

A S Lewin1, K A Drenser, W W Hauswirth, S Nishikawa, D Yasumura, J G Flannery, M M LaVail.   

Abstract

Ribozymes, catalytic RNA molecules that cleave a complementary mRNA sequence, have potential as therapeutics for dominantly inherited disease. Twelve percent of American patients with the blinding disease autosomal dominant retinitis pigmentosa (ADRP) carry a substitution of histidine for proline at codon 23 (P23H) in their rhodopsin gene, resulting in photoreceptor cell death from the synthesis of the abnormal gene product. Ribozymes can discriminate and catalyze the in vitro destruction of P23H mutant mRNAs from a transgenic rat model of ADRP. Here, we demonstrate that in vivo expression of either a hammerhead or hairpin ribozyme in this rat model considerably slows the rate of photoreceptor degeneration for at least three months. Catalytically inactive control ribozymes had less effect on the retinal degeneration. Intracellular production of ribozymes in photoreceptors was achieved by transduction with a recombinant adeno-associated virus (rAAV) incorporating a rod opsin promoter. Ribozyme-directed cleavage of mutant mRNAs, therefore, may be an effective therapy for ADRP and also may be applicable to other inherited diseases.

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Year:  1998        PMID: 9701253     DOI: 10.1038/nm0898-967

Source DB:  PubMed          Journal:  Nat Med        ISSN: 1078-8956            Impact factor:   53.440


  131 in total

1.  Stable transgene expression in rod photoreceptors after recombinant adeno-associated virus-mediated gene transfer to monkey retina.

Authors:  J Bennett; A M Maguire; A V Cideciyan; M Schnell; E Glover; V Anand; T S Aleman; N Chirmule; A R Gupta; Y Huang; G P Gao; W C Nyberg; J Tazelaar; J Hughes; J M Wilson; S G Jacobson
Journal:  Proc Natl Acad Sci U S A       Date:  1999-08-17       Impact factor: 11.205

2.  Adenovirus-mediated expression of a ribozyme to c-myb mRNA inhibits smooth muscle cell proliferation and neointima formation in vivo.

Authors:  D G Macejak; H Lin; S Webb; J Chase; K Jensen; T C Jarvis; J M Leiden; L Couture
Journal:  J Virol       Date:  1999-09       Impact factor: 5.103

Review 3.  Molecular therapy in ocular wound healing.

Authors:  M F Cordeiro; G S Schultz; R R Ali; S S Bhattacharya; P T Khaw
Journal:  Br J Ophthalmol       Date:  1999-11       Impact factor: 4.638

Review 4.  Molecular ophthalmology: an update on animal models for retinal degenerations and dystrophies.

Authors:  F Hafezi; C Grimm; B C Simmen; A Wenzel; C E Remé
Journal:  Br J Ophthalmol       Date:  2000-08       Impact factor: 4.638

5.  Gene therapy: new "magic bullets" to prevent ocular scarring.

Authors:  P T Khaw; A D Cambrey; G A Limb; J T Daniels
Journal:  Br J Ophthalmol       Date:  2002-05       Impact factor: 4.638

6.  Ribozyme rescue of photoreceptor cells in P23H transgenic rats: long-term survival and late-stage therapy.

Authors:  M M LaVail; D Yasumura; M T Matthes; K A Drenser; J G Flannery; A S Lewin; W W Hauswirth
Journal:  Proc Natl Acad Sci U S A       Date:  2000-10-10       Impact factor: 11.205

Review 7.  The hairpin ribozyme. Discovery, mechanism, and development for gene therapy.

Authors:  R Shippy; R Lockner; M Farnsworth; A Hampel
Journal:  Mol Biotechnol       Date:  1999-08       Impact factor: 2.695

Review 8.  The Gordon Wilson Lecture. From basic virology to human gene therapy.

Authors:  K I Berns
Journal:  Trans Am Clin Climatol Assoc       Date:  1999

Review 9.  Prevention and therapy in hereditary retinal degenerations.

Authors:  Dorothea Besch; Eberhart Zrenner
Journal:  Doc Ophthalmol       Date:  2003-01       Impact factor: 2.379

10.  Hammerhead ribozymes selectively suppress mutant type I collagen mRNA in osteogenesis imperfecta fibroblasts.

Authors:  P A Dawson; J C Marini
Journal:  Nucleic Acids Res       Date:  2000-10-15       Impact factor: 16.971

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