Literature DB >> 33278565

Clinical Perspective: Treating RPE65-Associated Retinal Dystrophy.

Albert M Maguire1, Jean Bennett1, Elena M Aleman2, Bart P Leroy3, Tomas S Aleman4.   

Abstract

Until recently, there was no approved treatment for a retinal degenerative disease. Subretinal injection of a recombinant adeno-associated virus (AAV) delivering the normal copy of the human RPE65 cDNA led to reversal of blindness first in animal models and then in humans. This led to the first US Food and Drug Administration (FDA)-approved gene therapy product for a genetic disease, voretigene neparvovec-rzyl (Luxturna). Luxturna was then approved by the European Medicines Association and is now available in the US through Spark Therapeutics and worldwide through Novartis. Not only has treatment with Luxturna changed the lives of people previously destined to live a life of blindness, but it has fueled interest in developing additional gene therapy reagents targeting numerous other genetic forms of inherited retinal disease. This review describes many of the considerations for administration of Luxturna and describes how lessons from experience with Luxturna could lead to additional gene-based treatments of blindness.
Copyright © 2020 The American Society of Gene and Cell Therapy. Published by Elsevier Inc. All rights reserved.

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Year:  2020        PMID: 33278565      PMCID: PMC7854308          DOI: 10.1016/j.ymthe.2020.11.029

Source DB:  PubMed          Journal:  Mol Ther        ISSN: 1525-0016            Impact factor:   11.454


  173 in total

Review 1.  Shedding new light on the generation of the visual chromophore.

Authors:  Krzysztof Palczewski; Philip D Kiser
Journal:  Proc Natl Acad Sci U S A       Date:  2020-08-05       Impact factor: 11.205

2.  In vivo dynamics of retinal injury and repair in the rhodopsin mutant dog model of human retinitis pigmentosa.

Authors:  Artur V Cideciyan; Samuel G Jacobson; Tomas S Aleman; Danian Gu; Susan E Pearce-Kelling; Alexander Sumaroka; Gregory M Acland; Gustavo D Aguirre
Journal:  Proc Natl Acad Sci U S A       Date:  2005-03-22       Impact factor: 11.205

3.  Improved Intravitreal AAV-Mediated Inner Retinal Gene Transduction after Surgical Internal Limiting Membrane Peeling in Cynomolgus Monkeys.

Authors:  Kazuhisa Takahashi; Tsutomu Igarashi; Koichi Miyake; Maika Kobayashi; Chiemi Yaguchi; Osamu Iijima; Yoshiyuki Yamazaki; Yuko Katakai; Noriko Miyake; Shuhei Kameya; Takashi Shimada; Hiroshi Takahashi; Takashi Okada
Journal:  Mol Ther       Date:  2017-01-04       Impact factor: 11.454

4.  Differential macular morphology in patients with RPE65-, CEP290-, GUCY2D-, and AIPL1-related Leber congenital amaurosis.

Authors:  Sirichai Pasadhika; Gerald A Fishman; Edwin M Stone; Martin Lindeman; Ruth Zelkha; Irma Lopez; Robert K Koenekoop; Mahnaz Shahidi
Journal:  Invest Ophthalmol Vis Sci       Date:  2009-12-03       Impact factor: 4.799

5.  Human retinal gene therapy for Leber congenital amaurosis shows advancing retinal degeneration despite enduring visual improvement.

Authors:  Artur V Cideciyan; Samuel G Jacobson; William A Beltran; Alexander Sumaroka; Malgorzata Swider; Simone Iwabe; Alejandro J Roman; Melani B Olivares; Sharon B Schwartz; András M Komáromy; William W Hauswirth; Gustavo D Aguirre
Journal:  Proc Natl Acad Sci U S A       Date:  2013-01-22       Impact factor: 11.205

6.  Safety and durability of effect of contralateral-eye administration of AAV2 gene therapy in patients with childhood-onset blindness caused by RPE65 mutations: a follow-on phase 1 trial.

Authors:  Jean Bennett; Jennifer Wellman; Kathleen A Marshall; Sarah McCague; Manzar Ashtari; Julie DiStefano-Pappas; Okan U Elci; Daniel C Chung; Junwei Sun; J Fraser Wright; Dominique R Cross; Puya Aravand; Laura L Cyckowski; Jeannette L Bennicelli; Federico Mingozzi; Alberto Auricchio; Eric A Pierce; Jason Ruggiero; Bart P Leroy; Francesca Simonelli; Katherine A High; Albert M Maguire
Journal:  Lancet       Date:  2016-06-30       Impact factor: 79.321

7.  Impairment of the transient pupillary light reflex in Rpe65(-/-) mice and humans with leber congenital amaurosis.

Authors:  Tomas S Aleman; Samuel G Jacobson; John D Chico; Michele L Scott; Andy Y Cheung; Elizabeth A M Windsor; Masatoshi Furushima; T Michael Redmond; Jean Bennett; Krzysztof Palczewski; Artur V Cideciyan
Journal:  Invest Ophthalmol Vis Sci       Date:  2004-04       Impact factor: 4.799

8.  Human cone photoreceptor dependence on RPE65 isomerase.

Authors:  Samuel G Jacobson; Tomas S Aleman; Artur V Cideciyan; Elise Heon; Marcin Golczak; William A Beltran; Alexander Sumaroka; Sharon B Schwartz; Alejandro J Roman; Elizabeth A M Windsor; James M Wilson; Gustavo D Aguirre; Edwin M Stone; Krzysztof Palczewski
Journal:  Proc Natl Acad Sci U S A       Date:  2007-09-11       Impact factor: 11.205

9.  Evaluation of an Optimized Injection System for Retinal Gene Therapy in Human Patients.

Authors:  M Dominik Fischer; Doron G Hickey; Mandeep S Singh; Robert E MacLaren
Journal:  Hum Gene Ther Methods       Date:  2016-08       Impact factor: 2.396

10.  Treatment Potential for LCA5-Associated Leber Congenital Amaurosis.

Authors:  Katherine E Uyhazi; Puya Aravand; Brent A Bell; Zhangyong Wei; Lanfranco Leo; Leona W Serrano; Denise J Pearson; Ivan Shpylchak; Jennifer Pham; Vidyullatha Vasireddy; Jean Bennett; Tomas S Aleman
Journal:  Invest Ophthalmol Vis Sci       Date:  2020-05-11       Impact factor: 4.799

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  11 in total

Review 1.  Prenatal Somatic Cell Gene Therapies: Charting a Path Toward Clinical Applications (Proceedings of the CERSI-FDA Meeting).

Authors:  Akos Herzeg; Graça Almeida-Porada; R Alta Charo; Anna L David; Juan Gonzalez-Velez; Nalin Gupta; Larissa Lapteva; Billie Lianoglou; William Peranteau; Christopher Porada; Stephan J Sanders; Teresa N Sparks; David H Stitelman; Evi Struble; Charlotte J Sumner; Tippi C MacKenzie
Journal:  J Clin Pharmacol       Date:  2022-09       Impact factor: 2.860

2.  Short-Term Outcomes of the First in Vivo Gene Therapy for RPE65-Mediated Retinitis Pigmentosa.

Authors:  Jay Jiyong Kwak; Hae Rang Kim; Suk Ho Byeon
Journal:  Yonsei Med J       Date:  2022-07       Impact factor: 3.052

Review 3.  Retinal pigment epithelium 65 kDa protein (RPE65): An update.

Authors:  Philip D Kiser
Journal:  Prog Retin Eye Res       Date:  2021-10-02       Impact factor: 19.704

4.  A Virtual Reality Orientation and Mobility Test for Inherited Retinal Degenerations: Testing a Proof-of-Concept After Gene Therapy.

Authors:  Tomas S Aleman; Alexander J Miller; Katherine H Maguire; Elena M Aleman; Leona W Serrano; Keli B O'Connor; Emma C Bedoukian; Bart P Leroy; Albert M Maguire; Jean Bennett
Journal:  Clin Ophthalmol       Date:  2021-03-02

5.  Intravenous infusion of small umbilical cord mesenchymal stem cells could enhance safety and delay retinal degeneration in RCS rats.

Authors:  Qingling Liang; Qiyou Li; Bangqi Ren; Zheng Qin Yin
Journal:  BMC Ophthalmol       Date:  2022-02-11       Impact factor: 2.209

6.  RPE65-Associated Retinopathies in the Italian Population: A Longitudinal Natural History Study.

Authors:  Francesco Testa; Vittoria Murro; Sabrina Signorini; Leonardo Colombo; Giancarlo Iarossi; Francesco Parmeggiani; Benedetto Falsini; Anna Paola Salvetti; Raffaella Brunetti-Pierri; Giorgia Aprile; Chiara Bertone; Agnese Suppiej; Francesco Romano; Marianthi Karali; Simone Donati; Paolo Melillo; Andrea Sodi; Luciano Quaranta; Luca Rossetti; Luca Buzzonetti; Marzio Chizzolini; Stanislao Rizzo; Giovanni Staurenghi; Sandro Banfi; Claudio Azzolini; Francesca Simonelli
Journal:  Invest Ophthalmol Vis Sci       Date:  2022-02-01       Impact factor: 4.799

Review 7.  Delivery Systems of Retinoprotective Proteins in the Retina.

Authors:  Ivan T Rebustini; Alexandra Bernardo-Colón; Alejandra Isasi Nalvarte; S Patricia Becerra
Journal:  Int J Mol Sci       Date:  2021-05-19       Impact factor: 5.923

8.  Bardet-Biedl syndrome-7 (BBS7) shows treatment potential and a cone-rod dystrophy phenotype that recapitulates the non-human primate model.

Authors:  Tomas S Aleman; Erin C O'Neil; Keli O'Connor; Yu You Jiang; Isabella A Aleman; Jean Bennett; Jessica I W Morgan; Brian W Toussaint
Journal:  Ophthalmic Genet       Date:  2021-03-17       Impact factor: 1.274

9.  Inherited retinal diseases are the most common cause of blindness in the working-age population in Australia.

Authors:  Rachael C Heath Jeffery; Syed Aqif Mukhtar; Ian L McAllister; William H Morgan; David A Mackey; Fred K Chen
Journal:  Ophthalmic Genet       Date:  2021-05-03       Impact factor: 1.803

10.  Perspectives of people with inherited retinal diseases on ocular gene therapy in Australia: protocol for a national survey.

Authors:  Heather G Mack; Fred K Chen; John Grigg; Robyn Jamieson; John De Roach; Fleur O'Hare; Alexis Ceecee Britten-Jones; Myra McGuinness; Nicole Tindill; Lauren Ayton
Journal:  BMJ Open       Date:  2021-06-22       Impact factor: 2.692

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