Literature DB >> 9335046

Genetically engineered large animal model for studying cone photoreceptor survival and degeneration in retinitis pigmentosa.

R M Petters1, C A Alexander, K D Wells, E B Collins, J R Sommer, M R Blanton, G Rojas, Y Hao, W L Flowers, E Banin, A V Cideciyan, S G Jacobson, F Wong.   

Abstract

Patients with retinitis pigmentosa (RP) typically develop night blindness early in life due to loss of rod photoreceptors. The remaining cone photoreceptors are the mainstay of their vision; however, over years or decades, these cones slowly degenerate, leading to blindness. We created transgenic pigs that express a mutated rhodopsin gene (Pro347Leu). Like RP patients with the same mutation, these pigs have early and severe rod loss; initially their cones are relatively spared, but these surviving cones slowly degenerate. By age 20 months, there is only a single layer of morphologically abnormal cones and the cone electroretinogram is markedly reduced. Given the strong similarities in phenotype to that of RP patients, these transgenic pigs will provide a large animal model for study of the protracted phase of cone degeneration found in RP and for preclinical treatment trials.

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Year:  1997        PMID: 9335046     DOI: 10.1038/nbt1097-965

Source DB:  PubMed          Journal:  Nat Biotechnol        ISSN: 1087-0156            Impact factor:   54.908


  92 in total

Review 1.  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

2.  A mutation of early photoreceptor development, mikre oko, reveals cell-cell interactions involved in the survival and differentiation of zebrafish photoreceptors.

Authors:  G Doerre; J Malicki
Journal:  J Neurosci       Date:  2001-09-01       Impact factor: 6.167

3.  Self-complementary AAV-mediated gene therapy restores cone function and prevents cone degeneration in two models of Rpe65 deficiency.

Authors:  J Pang; S E Boye; B Lei; S L Boye; D Everhart; R Ryals; Y Umino; B Rohrer; J Alexander; J Li; X Dai; Q Li; B Chang; R Barlow; W W Hauswirth
Journal:  Gene Ther       Date:  2010-03-18       Impact factor: 5.250

4.  Cone degeneration following rod ablation in a reversible model of retinal degeneration.

Authors:  Rene Y Choi; Gustav A Engbretson; Eduardo C Solessio; Georgette A Jones; Adam Coughlin; Ilija Aleksic; Michael E Zuber
Journal:  Invest Ophthalmol Vis Sci       Date:  2011-01-21       Impact factor: 4.799

Review 5.  Genetically engineered livestock for biomedical models.

Authors:  Christopher S Rogers
Journal:  Transgenic Res       Date:  2016-01-28       Impact factor: 2.788

Review 6.  Genome editing revolutionize the creation of genetically modified pigs for modeling human diseases.

Authors:  Jing Yao; Jiaojiao Huang; Jianguo Zhao
Journal:  Hum Genet       Date:  2016-07-18       Impact factor: 4.132

7.  Synchronization and superovulation of mature cycling gilts for the collection of pronuclear stage embryos.

Authors:  Jeffrey R Sommer; E Bruce Collins; Jose L Estrada; Robert M Petters
Journal:  Anim Reprod Sci       Date:  2006-10-14       Impact factor: 2.145

8.  P23H opsin knock-in mice reveal a novel step in retinal rod disc morphogenesis.

Authors:  Sanae Sakami; Alexander V Kolesnikov; Vladimir J Kefalov; Krzysztof Palczewski
Journal:  Hum Mol Genet       Date:  2013-11-07       Impact factor: 6.150

9.  Inner and outer retinal changes in retinal degenerations associated with ABCA4 mutations.

Authors:  Wei Chieh Huang; Artur V Cideciyan; Alejandro J Roman; Alexander Sumaroka; Rebecca Sheplock; Sharon B Schwartz; Edwin M Stone; Samuel G Jacobson
Journal:  Invest Ophthalmol Vis Sci       Date:  2014-03-20       Impact factor: 4.799

10.  A battery of cell- and structure-specific markers for the adult porcine retina.

Authors:  Ulrica Englund Johansson; Sajedeh Eftekhari; Karin Warfvinge
Journal:  J Histochem Cytochem       Date:  2010-01-19       Impact factor: 2.479

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