Literature DB >> 34031601

Partial recovery of visual function in a blind patient after optogenetic therapy.

José-Alain Sahel1,2,3,4, Elise Boulanger-Scemama5,6, Chloé Pagot7, Angelo Arleo8, Francesco Galluppi9, Joseph N Martel10, Simona Degli Esposti11, Alexandre Delaux8, Jean-Baptiste de Saint Aubert8, Caroline de Montleau7, Emmanuel Gutman7, Isabelle Audo8,5, Jens Duebel8, Serge Picaud8, Deniz Dalkara8, Laure Blouin9, Magali Taiel9, Botond Roska12,13.   

Abstract

Optogenetics may enable mutation-independent, circuit-specific restoration of neuronal function in neurological diseases. Retinitis pigmentosa is a neurodegenerative eye disease where loss of photoreceptors can lead to complete blindness. In a blind patient, we combined intraocular injection of an adeno-associated viral vector encoding ChrimsonR with light stimulation via engineered goggles. The goggles detect local changes in light intensity and project corresponding light pulses onto the retina in real time to activate optogenetically transduced retinal ganglion cells. The patient perceived, located, counted and touched different objects using the vector-treated eye alone while wearing the goggles. During visual perception, multichannel electroencephalographic recordings revealed object-related activity above the visual cortex. The patient could not visually detect any objects before injection with or without the goggles or after injection without the goggles. This is the first reported case of partial functional recovery in a neurodegenerative disease after optogenetic therapy.
© 2021. The Author(s), under exclusive licence to Springer Nature America, Inc.

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Year:  2021        PMID: 34031601     DOI: 10.1038/s41591-021-01351-4

Source DB:  PubMed          Journal:  Nat Med        ISSN: 1078-8956            Impact factor:   87.241


  40 in total

1.  Efficacy and safety of voretigene neparvovec (AAV2-hRPE65v2) in patients with RPE65-mediated inherited retinal dystrophy: a randomised, controlled, open-label, phase 3 trial.

Authors:  Stephen Russell; Jean Bennett; Jennifer A Wellman; Daniel C Chung; Zi-Fan Yu; Amy Tillman; Janet Wittes; Julie Pappas; Okan Elci; Sarah McCague; Dominique Cross; Kathleen A Marshall; Jean Walshire; Taylor L Kehoe; Hannah Reichert; Maria Davis; Leslie Raffini; Lindsey A George; F Parker Hudson; Laura Dingfield; Xiaosong Zhu; Julia A Haller; Elliott H Sohn; Vinit B Mahajan; Wanda Pfeifer; Michelle Weckmann; Chris Johnson; Dina Gewaily; Arlene Drack; Edwin Stone; Katie Wachtel; Francesca Simonelli; Bart P Leroy; J Fraser Wright; Katherine A High; Albert M Maguire
Journal:  Lancet       Date:  2017-07-14       Impact factor: 79.321

Review 2.  Gene therapy for blindness.

Authors:  José-Alain Sahel; Botond Roska
Journal:  Annu Rev Neurosci       Date:  2013-05-31       Impact factor: 12.449

Review 3.  Depicting brighter possibilities for treating blindness.

Authors:  José-Alain Sahel; Jean Bennett; Botond Roska
Journal:  Sci Transl Med       Date:  2019-05-29       Impact factor: 17.956

Review 4.  Emerging therapies for inherited retinal degeneration.

Authors:  Hendrik P N Scholl; Rupert W Strauss; Mandeep S Singh; Deniz Dalkara; Botond Roska; Serge Picaud; José-Alain Sahel
Journal:  Sci Transl Med       Date:  2016-12-07       Impact factor: 17.956

Review 5.  Restoring vision.

Authors:  Botond Roska; José-Alain Sahel
Journal:  Nature       Date:  2018-05-16       Impact factor: 49.962

6.  Digenic retinitis pigmentosa due to mutations at the unlinked peripherin/RDS and ROM1 loci.

Authors:  K Kajiwara; E L Berson; T P Dryja
Journal:  Science       Date:  1994-06-10       Impact factor: 47.728

7.  [Electric bistoury: practical applications in periodontal surgery].

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8.  Influence of rumen fermentation on response to endophyte-infected tall fescue seed measured by a rat bioassay.

Authors:  M L Westendorf; G E Mitchell; R E Tucker
Journal:  Drug Chem Toxicol       Date:  1992       Impact factor: 3.356

9.  Light-activated channels targeted to ON bipolar cells restore visual function in retinal degeneration.

Authors:  Pamela S Lagali; David Balya; Gautam B Awatramani; Thomas A Münch; Douglas S Kim; Volker Busskamp; Constance L Cepko; Botond Roska
Journal:  Nat Neurosci       Date:  2008-04-27       Impact factor: 24.884

10.  In vivo-directed evolution of a new adeno-associated virus for therapeutic outer retinal gene delivery from the vitreous.

Authors:  Deniz Dalkara; Leah C Byrne; Ryan R Klimczak; Meike Visel; Lu Yin; William H Merigan; John G Flannery; David V Schaffer
Journal:  Sci Transl Med       Date:  2013-06-12       Impact factor: 17.956

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  64 in total

Review 1.  Optophysiology: Illuminating cell physiology with optogenetics.

Authors:  Peng Tan; Lian He; Yun Huang; Yubin Zhou
Journal:  Physiol Rev       Date:  2022-01-24       Impact factor: 37.312

Review 2.  Chemogenetics as a neuromodulatory approach to treating neuropsychiatric diseases and disorders.

Authors:  Jingwei Song; Ruchit V Patel; Massoud Sharif; Anagha Ashokan; Michael Michaelides
Journal:  Mol Ther       Date:  2021-12-01       Impact factor: 11.454

3.  Activation of retinal ganglion cells using a biomimetic artificial retina.

Authors:  Jordan A Greco; Nicole L Wagner; Ralph J Jensen; Daniel B Lawrence; Matthew J Ranaghan; Megan N Sandberg; Daniel J Sandberg; Robert R Birge
Journal:  J Neural Eng       Date:  2021-12-01       Impact factor: 5.379

4.  How the world's biggest brain maps could transform neuroscience.

Authors:  Alison Abbott
Journal:  Nature       Date:  2021-10       Impact factor: 49.962

Review 5.  Brighten the Future: Photobiomodulation and Optogenetics.

Authors:  Li-Da Huang
Journal:  Focus (Am Psychiatr Publ)       Date:  2022-01-25

6.  Restoring partial vision to a blind patient.

Authors:  Larry I Benowitz; John E Dowling; Roman J Giger; Thomas V Johnson; Donald J Zack
Journal:  Fac Rev       Date:  2022-06-27

7.  Directed evolution of adeno-associated virus for efficient gene delivery to microglia.

Authors:  Rui Lin; Youtong Zhou; Ting Yan; Ruiyu Wang; Heng Li; Zhaofa Wu; Xinshuang Zhang; Xiangyu Zhou; Fei Zhao; Li Zhang; Yulong Li; Minmin Luo
Journal:  Nat Methods       Date:  2022-07-25       Impact factor: 47.990

8.  Karl Deisseroth, Peter Hegemann, and Dieter Oesterhelt receive the 2021 Albert Lasker Basic Medical Research Award.

Authors:  Lisa R Conti
Journal:  J Clin Invest       Date:  2021-09-24       Impact factor: 19.456

Review 9.  Regenerative and restorative medicine for eye disease.

Authors:  Russell N Van Gelder; Michael F Chiang; Michael A Dyer; Thomas N Greenwell; Leonard A Levin; Rachel O Wong; Clive N Svendsen
Journal:  Nat Med       Date:  2022-06-17       Impact factor: 87.241

Review 10.  Bioengineering strategies for restoring vision.

Authors:  Jasmina Cehajic-Kapetanovic; Mandeep S Singh; Eberhart Zrenner; Robert E MacLaren
Journal:  Nat Biomed Eng       Date:  2022-01-31       Impact factor: 25.671

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