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

Alemán, Tomás S.; Miller, Alexander J; Maguire, Katherine H; Aleman, Elena M; Serrano, Leona; O’Connor, Keli; Bedoukian, Emma; Leroy, Bart P.; Maguire, Albert M.; Bennett, Jean

doi:10.2147/opth.s292527

Cited by 18 publications

(11 citation statements)

References 45 publications

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“…139 A recent study has tested a VR MT in patients with RPE65-LCA, providing proof-of-concept of the utility of this approach and encouraging further broader application to IRD, and potentially resulting in mobility assessments being more accessible and varied. 140 Another interesting field has been the development of tools and applications (apps) that can assess aspects of vision while we use our own digital devices. 141 Information about VF, tracking, CV, CS and VA can be estimated through the use of apps, again potentially providing a more accessible (and arguably more directly functionally relevant) way of characterizing and monitoring vision 141,142 Standardization and validation of such approaches will be necessary.…”

Section: Virtual Reality and New Methodologiesmentioning

confidence: 99%

Functional evaluation in inherited retinal disease

et al. 2021

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Functional assessments are a fundamental part of the clinical evaluation of patients with inherited retinal diseases (IRDs). Their importance and impact have become increasingly notable, given the significant breadth and number of clinical trials and studies investigating multiple avenues of intervention across a wide range of IRDs, including gene, pharmacological and cellular therapies. Moreover, the fact that many clinical trials are reporting improvements in vision, rather than the previously anticipated structural stability/slowing of degeneration, makes functional evaluation of primary relevance. In this review, we will describe a range of methods employed to characterise retinal function and functional vision, beginning with tests variably included in the clinic, such as visual acuity, electrophysiological assessment and colour discrimination, and then discussing assessments often reserved for clinical trials/research studies such as photoaversion testing, full-field static perimetry and microperimetry, and vision-guided mobility testing; addressing perimetry in greatest detail, given it is commonly a primary outcome metric. We will focus on how these tests can help diagnose and monitor particular genotypes, also noting their limitations/challenges and exploring analytical methodologies for better exploiting functional measurements, as well as how they facilitate patient inclusion and stratification in clinical trials and serve as outcome measures.

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Section: Virtual Reality and New Methodologiesmentioning

confidence: 99%

Functional evaluation in inherited retinal disease

et al. 2021

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“…Since then, the MLMT has been adopted in various ongoing therapeutic interventional trials. Virtual reality (VR)-based O&M platforms provide promising alternatives pending further validation,[ 33 ] and a Phase 1 study evaluating a new VR mobility tool (clinicaltrials.gov identifier, NCT04289571) in IRD patients and healthy volunteers is underway. Established artificial systems for naturalistic test approximating daily activities, such as StreetLab ® and Homelab ® , are also available.…”

Section: Natural History Studies and Defining Clinical Trial Outcomesmentioning

confidence: 99%

Therapeutic landscape for inherited ocular diseases: current and emerging therapies

Chan

Leroy

2023

Singapore Medical Journal

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Inherited ocular diseases comprise a heterogeneous group of rare and complex diseases, including inherited retinal diseases (IRDs) and inherited optic neuropathies. Recent success in adeno-associated virus-based gene therapy, voretigene neparvovec (Luxturna ® ) for RPE65 -related IRDs, has heralded rapid evolution in gene therapy platform technologies and strategies, from gene augmentation to RNA editing, as well as gene agnostic approaches such as optogenetics. This review discusses the fundamentals underlying the mode of inheritance, natural history studies and clinical trial outcomes, as well as current and emerging therapies covering gene therapy strategies, cell-based therapies and bionic vision.

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“…Aleman and coworkers describe a VR test based loosely on the principles of the MLMT described above. 3 In this task, the subject navigates a similar VR course in which arrows (red instead of black, and adjusted in luminance to ensure that they are just visible to the subject) mark the direction to be taken. A head tracker incorporated into the goggles and limb (hand/ankle) trackers monitor the patient's movements.…”

Section: Streetlab Platforms (Institut De La Vision)mentioning

confidence: 99%

“…A pilot study involving 4 patients with LCA2 and 6 control subjects suggests that this may be a promising approach for future studies. 3 Knopf et al 72 have developed a virtual reality obstacle course incorporating real world experiences such as walking along a crowded foot path or across a virtual carpark that can offer a safe, affordable, and standardized orientation and mobility assessment tool that provides objective outcome measures not previously achieved by physical courses.…”

Section: Streetlab Platforms (Institut De La Vision)mentioning

confidence: 99%