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Lecture October 2017

Concepts and Strategies in Retinal Gene Therapy

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Gustavo D. Aguirre
TAGS: gene therapy, retinal cone, dog, domestic, genes, oncogenes, photoreceptors, retina, spatial vectors
Invest. Ophthalmol. Vis. Sci.. 2017; 58(12):5399-5411. doi: 10.1167/iovs.17-22978

Includes: Supplemental Content

ARVO Annual Meeting Abstract June 2017

Variable cone dysfunction and progressive ophthalmoscopic changes in a canine model of RPGRIP1 and MAP9 multigenic cone-rod dystrophy

Keiko Miyadera; Felipe Pompeo Marinho; Kendra McDaid; Valerie Liliane Dufour; Simone Iwabe; Gautami Das; Gustavo D Aguirre

Invest. Ophthalmol. Vis. Sci.. 2017; 58(8):269. doi:

ARVO Annual Meeting Abstract June 2017

Influence of Ambient Illumination on the Course of Retinal Degeneration in a Canine model of RHO-ADRP

Valerie Liliane Dufour; Simone Iwabe; Gustavo D Aguirre; William A Beltran

Invest. Ophthalmol. Vis. Sci.. 2017; 58(8):278. doi:

ARVO Annual Meeting Abstract June 2017

Development of an optimized light damage paradigm to assess efficacy of gene therapy in a canine model of RHO-ADRP

Raghavi Sudharsan; Kristina M Simone; Nathan P Anderson; Gustavo D Aguirre; William A Beltran

Invest. Ophthalmol. Vis. Sci.. 2017; 58(8):282. doi:

ARVO Annual Meeting Abstract June 2017

NDR kinases regulate retinal development, homeostasis and gene expression

Hélène M Léger; Daniel P. Beiting; William A Beltran; Gustavo D Aguirre; Francis Luca

Invest. Ophthalmol. Vis. Sci.. 2017; 58(8):353. doi:

ARVO Annual Meeting Abstract June 2017

Outcome measures for BEST1 gene augmentation therapy: restoration of RPE-photoreceptor interface homeostasis

Karina E Guziewicz; William A Beltran; Artur V Cideciyan; Valerie Liliane Dufour; Andras M Komaromy; Emily McTish; Alfred S Lewin; William W Hauswirth; Samuel G Jacobson; Gustavo D Aguirre

Invest. Ophthalmol. Vis. Sci.. 2017; 58(8):2273. doi:

ARVO Annual Meeting Abstract June 2017

Upregulation of pro-inflammatory genes accompanies photoreceptors demise in canine models of retinal degeneration

Tatyana Appelbaum; Evelyn Santana; Gustavo D Aguirre

Invest. Ophthalmol. Vis. Sci.. 2017; 58(8):3587. doi:

ARVO Annual Meeting Abstract June 2017

AAV-mediated Knockdown and Replacement of Rhodopsin Protects Rods in a Canine Model of RHO -ADRP

William A Beltran; Artur V Cideciyan; Raghavi Sudharsan; Michael Massengill; Brian Rossmiller; Valerie Liliane Dufour; felipe Pompeo Marinho; Tatyana Appelbaum; Malgorzata Swider; Mychajlo S. Kosyk; Simone Iwabe; William W Hauswirth; Samuel G Jacobson; Alfred S Lewin; Gustavo D Aguirre

Invest. Ophthalmol. Vis. Sci.. 2017; 58(8):4483. doi:

ARVO Annual Meeting Abstract June 2017

Evaluation of AAV2tYF-GRK1-RPGR vectors in a canine model of RPGR -XLRP

Guo-jie Ye; William A Beltran; Valerie Liliane Dufour; Felipe Pompeo Marinho; Inna Martynyuk; Chuanjian Song; David Knop; Gustavo D Aguirre; Jeffrey D Chulay; Mark S Shearman

Invest. Ophthalmol. Vis. Sci.. 2017; 58(8):4486. doi:

Retinal Cell Biology January 2017

Acute and Protracted Cell Death in Light-Induced Retinal Degeneration in the Canine Model of Rhodopsin Autosomal Dominant Retinitis Pigmentosa

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Raghavi Sudharsan; Kristina M. Simone; Nathan P. Anderson; Gustavo D. Aguirre; William A. Beltran
TAGS: cell death, dog, domestic, in situ nick-end labeling, photoreceptors, retinal degeneration, retinitis pigmentosa, rhodopsin, retina, light exposure
Invest. Ophthalmol. Vis. Sci.. 2017; 58(1):270-281. doi: 10.1167/iovs.16-20749

Includes: Supplemental Content

ARVO Annual Meeting Abstract September 2016

Photoreceptor structure and gene expression in the canine NPHP5-LCA ciliopathy model

Louise M Downs; Erin M Scott; Sem Genini; William A Beltran; Gustavo D Aguirre

Invest. Ophthalmol. Vis. Sci.. 2016; 57(12):3188. doi:

ARVO Annual Meeting Abstract September 2016

Bestrophinopathy is an RPE-photoreceptor interface disease

Karina E Guziewicz; William A Beltran; Simone Iwabe; Artur V Cideciyan; Samuel G Jacobson; Anuradha Dhingra; Kathleen Battaglia; Gustavo D Aguirre

Invest. Ophthalmol. Vis. Sci.. 2016; 57(12) doi:

ARVO Annual Meeting Abstract September 2016

A quantitative trait locus on canine chromosome 31 influences disease severity phenotype of XLPRA1

Tatyana Appelbaum; Doreen Becker; Evelyn Santana; Gustavo D Aguirre

Invest. Ophthalmol. Vis. Sci.. 2016; 57(12):6569. doi:

ARVO Annual Meeting Abstract September 2016

Study of pro-apoptotic and pro-survival pathways at late stage disease in two non-allelic forms of photoreceptor degeneration

Raghavi Sudharsan; Gustavo D Aguirre; William A Beltran

Invest. Ophthalmol. Vis. Sci.. 2016; 57(12):166. doi:

ARVO Annual Meeting Abstract September 2016

RPGRIP1 mutant dogs show variable cone ERG functions

Gautami Das; Evelyn Santana; Simone Iwabe; Gustavo D Aguirre; Keiko Miyadera

Invest. Ophthalmol. Vis. Sci.. 2016; 57(12):167. doi:

ARVO Annual Meeting Abstract September 2016

Azobenzene-based DENAQ photoswitch confers light sensitivity to degenerated but not to healthy canine retina

Sergei Nikonov; Natalia Dolgova; Ivan Tochitsky; Russell N Van Gelder; Richard Kramer; Gustavo D Aguirre; William A Beltran

Invest. Ophthalmol. Vis. Sci.. 2016; 57(12):602. doi:

ARVO Annual Meeting Abstract September 2016

Directed evolution identifies novel AAV capsid variants for targeting the canine outer retina following intravitreal delivery

Leah C Byrne; Meike Visel; Valerie Dufour; Felipe Pompeo Marinho; Gustavo D Aguirre; William A Beltran; David Schaffer; John Gerard Flannery

Invest. Ophthalmol. Vis. Sci.. 2016; 57(12):776. doi:

ARVO Annual Meeting Abstract September 2016

Two AAV2/5-mediated RPGR gene augmentation constructs driven by the GRK1 promoter rescue photoreceptor structure and function in a canine model o f RPGR X-linked retinitis pigmentosa

William A Beltran; Artur V Cideciyan; Simone Iwabe; Valerie Dufour; Felipe Pompeo Marinho; Guo-jie Ye; Mark Shearman; Jeffrey D Chulay; William Hauswirth; Samuel G Jacobson; Gustavo D Aguirre

Invest. Ophthalmol. Vis. Sci.. 2016; 57(12):785. doi:

ARVO Annual Meeting Abstract September 2016

AAV-mediated gene augmentation restores retinal function and vision in the canine model of NPHP5 Leber congenital amaurosis

Gustavo D Aguirre; Artur V Cideciyan; Sanford L. Boye; Simone Iwabe; Valerie Dufour; Felipe Pompeo Marinho; Louise M Downs; William Hauswirth; Samuel G Jacobson; William A Beltran

Invest. Ophthalmol. Vis. Sci.. 2016; 57(12) doi:

ARVO Annual Meeting Abstract June 2015

Long-term Cone ERG Functional Rescue in CNGB3 Mutant Achromatopsia Dogs by AAV-hCNGB3 Vectors Containing the PR1.7 Promoter and Packaged in AAV5, AAV9 or Mutant AAV2 Capsids

Andras M Komaromy; Guo-jie Ye; Christine Harman; Kristin L Koehl; Gustavo D Aguirre; Savitri Mandapati; Jeffrey D Chulay

Invest. Ophthalmol. Vis. Sci.. 2015; 56(7 ):2066. doi:

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Concepts and Strategies in Retinal Gene Therapy

Variable cone dysfunction and progressive ophthalmoscopic changes in a canine model of RPGRIP1 and MAP9 multigenic cone-rod dystrophy

Influence of Ambient Illumination on the Course of Retinal Degeneration in a Canine model of RHO-ADRP

Development of an optimized light damage paradigm to assess efficacy of gene therapy in a canine model of RHO-ADRP

NDR kinases regulate retinal development, homeostasis and gene expression

Outcome measures for BEST1 gene augmentation therapy: restoration of RPE-photoreceptor interface homeostasis

Upregulation of pro-inflammatory genes accompanies photoreceptors demise in canine models of retinal degeneration

AAV-mediated Knockdown and Replacement of Rhodopsin Protects Rods in a Canine Model of RHO -ADRP

Evaluation of AAV2tYF-GRK1-RPGR vectors in a canine model of RPGR -XLRP

Acute and Protracted Cell Death in Light-Induced Retinal Degeneration in the Canine Model of Rhodopsin Autosomal Dominant Retinitis Pigmentosa

Photoreceptor structure and gene expression in the canine NPHP5-LCA ciliopathy model

Bestrophinopathy is an RPE-photoreceptor interface disease

A quantitative trait locus on canine chromosome 31 influences disease severity phenotype of XLPRA1

Study of pro-apoptotic and pro-survival pathways at late stage disease in two non-allelic forms of photoreceptor degeneration

RPGRIP1 mutant dogs show variable cone ERG functions

Azobenzene-based DENAQ photoswitch confers light sensitivity to degenerated but not to healthy canine retina

Directed evolution identifies novel AAV capsid variants for targeting the canine outer retina following intravitreal delivery

Two AAV2/5-mediated RPGR gene augmentation constructs driven by the GRK1 promoter rescue photoreceptor structure and function in a canine model o f RPGR X-linked retinitis pigmentosa

AAV-mediated gene augmentation restores retinal function and vision in the canine model of NPHP5 Leber congenital amaurosis

Long-term Cone ERG Functional Rescue in CNGB3 Mutant Achromatopsia Dogs by AAV-hCNGB3 Vectors Containing the PR1.7 Promoter and Packaged in AAV5, AAV9 or Mutant AAV2 Capsids

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