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Clinical Ophthalmology Volume 15, 2021 - Issue
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Original Research

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

Tomas S Aleman1 Scheie Eye Institute at the Perelman Center for Advanced Medicine, University of Pennsylvania, Philadelphia, PA, USA;2 Center for Advanced Retinal and Ocular Therapeutics, University of Pennsylvania, Philadelphia, PA, USA;3 Division of Ophthalmology at the Children’s Hospital of Philadelphia of the Department of Ophthalmology, University of Pennsylvania, Philadelphia, PA, USACorrespondence[email protected]
View further author information
,
Alexander J Miller4 Neurology Virtual Reality Laboratory of the Department of Neurology, University of Pennsylvania, Philadelphia, PA, USAORCID Iconhttps://orcid.org/0000-0002-7255-5158View further author information
ORCID Icon,
Katherine H Maguire1 Scheie Eye Institute at the Perelman Center for Advanced Medicine, University of Pennsylvania, Philadelphia, PA, USA;2 Center for Advanced Retinal and Ocular Therapeutics, University of Pennsylvania, Philadelphia, PA, USAView further author information
,
Elena M Aleman2 Center for Advanced Retinal and Ocular Therapeutics, University of Pennsylvania, Philadelphia, PA, USAView further author information
,
Leona W Serrano1 Scheie Eye Institute at the Perelman Center for Advanced Medicine, University of Pennsylvania, Philadelphia, PA, USA;2 Center for Advanced Retinal and Ocular Therapeutics, University of Pennsylvania, Philadelphia, PA, USAView further author information
,
Keli B O’Connor1 Scheie Eye Institute at the Perelman Center for Advanced Medicine, University of Pennsylvania, Philadelphia, PA, USA;2 Center for Advanced Retinal and Ocular Therapeutics, University of Pennsylvania, Philadelphia, PA, USAView further author information
,
Emma C Bedoukian5 Division of Human Genetics, The Children’s Hospital of Philadelphia, Philadelphia, PA, USAView further author information
,
Bart P Leroy3 Division of Ophthalmology at the Children’s Hospital of Philadelphia of the Department of Ophthalmology, University of Pennsylvania, Philadelphia, PA, USA;6 Department of Pediatrics, Ghent University, Ghent, Belgium;7 Department of Ophthalmology, Ghent University, Ghent, Belgium;8 Center for Medical Genetics, Ghent University Hospital, Ghent, BelgiumORCID Iconhttps://orcid.org/0000-0002-9899-2081View further author information
ORCID Icon,
Albert M Maguire1 Scheie Eye Institute at the Perelman Center for Advanced Medicine, University of Pennsylvania, Philadelphia, PA, USA;2 Center for Advanced Retinal and Ocular Therapeutics, University of Pennsylvania, Philadelphia, PA, USA;3 Division of Ophthalmology at the Children’s Hospital of Philadelphia of the Department of Ophthalmology, University of Pennsylvania, Philadelphia, PA, USAView further author information
&
Jean Bennett1 Scheie Eye Institute at the Perelman Center for Advanced Medicine, University of Pennsylvania, Philadelphia, PA, USA;2 Center for Advanced Retinal and Ocular Therapeutics, University of Pennsylvania, Philadelphia, PA, USAView further author information
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Pages 939-952 | Published online: 02 Mar 2021

References

  • Bennett CR, Bex PJ, Bauer CM, Merabet LB. The assessment of visual function and functional vision. Semin Pediatr Neurol. 2019;31:30–40. doi:10.1016/j.spen.2019.05.006
  • Shingledecker CA, Foulke E. A human factors approach to the assessment of the mobility of blind pedestrians. Hum Factors. 1978;20(3):273–286. doi:10.1177/001872087802000303
  • Marron JA, Bailey IL. Visual factors and orientation-mobility performance. Am J Optom Physiol Opt. 1982;59(5):413–426. doi:10.1097/00006324-198205000-00009
  • Chang KJ, Dillon LL, Deverell L, Boon MY, Keay L. Orientation and mobility outcome measures. Clin Exp Optom. 2020;103(4):434–448. doi:10.1111/cxo.13004
  • Maguire AM, Bennett, J, Aleman E, et al. Clinical Perspective: Treating RPE65-Associated Retinal Dystrophy. Mol Ther. 2021;29(2):442–463. doi:10.1016/j.ymthe.2020.11.029.
  • Russell S, Bennett J, Wellman JA, et al. Efficacy and safety of voretigene neparvovec (AAV2-hRPE65v2) in patients with RPE65-mediated inherited retinal dystrophy: a randomised, controlled, open-label, phase 3 trial. Lancet. 2017;390(10097):849–860. doi:10.1016/S0140-6736(17)31868-8
  • Bainbridge JW, Smith AJ, Barker SS, et al. Effect of gene therapy on visual function in Leber’s congenital amaurosis. N Engl J Med. 2008;358(21):2231–2239. doi:10.1056/NEJMoa0802268
  • Jacobson SG, Cideciyan AV, Ratnakaram R, et al. Gene therapy for leber congenital amaurosis caused by RPE65 mutations: safety and efficacy in 15 children and adults followed up to 3 years. Arch Ophthalmol. 2012;130(1):9–24. doi:10.1001/archophthalmol.2011.298
  • Bainbridge JW, Mehat MS, Sundaram V, et al. Long-term effect of gene therapy on Leber’s congenital amaurosis. N Engl J Med. 2015;372(20):1887–1897. doi:10.1056/NEJMoa1414221
  • Shapiro A, Corcoran P, Sundstrom C, et al. Development and validation of a portable visual navigation challenge for assessment of retinal disease in multi-centered clinical trials. Invest Ophthalmol Vis Sci. 2017;58(8).
  • Jacobson SG, Cideciyan AV, Sumaroka A, et al. Outcome measures for clinical trials of leber congenital amaurosis caused by the intronic mutation in the CEP290 gene. Invest Ophthalmol Vis Sci. 2017;58(5):2609. doi:10.1167/iovs.17-21560
  • Chung DC, McCague S, Yu Z-F, et al. Novel mobility test to assess functional vision in patients with inherited retinal dystrophies: multi-luminance mobility test. Clin Exp Ophthalmol. 2018;46(3):247–259.
  • Cideciyan AV, Jacobson SG, Drack AV, et al. Effect of an intravitreal antisense oligonucleotide on vision in Leber congenital amaurosis due to a photoreceptor cilium defect. Nat Med. 2019;25(2):225–228. doi:10.1038/s41591-018-0295-0
  • Black A, Lovie‐Kitchin JE, Woods RL, Arnold N, Byrnes J, Murrish J. Mobility performance with retinitis pigmentosa. Clin Exp Optom. 1997;80(1):1–12. doi:10.1111/j.1444-0938.1997.tb04841.x
  • Jacobson SG, Cideciyan AV, Peshenko IV, et al. Determining consequences of retinal membrane guanylyl cyclase (RetGC1) deficiency in human leber congenital amaurosis en route to therapy: residual cone-photoreceptor vision correlates with biochemical properties of the mutants. Hum Mol Genet. 2013;22(1):168–183. doi:10.1093/hmg/dds421
  • Chung DC, McCague S, Yu ZF, et al. Novel mobility test to assess functional vision in patients with inherited retinal dystrophies. Clin Exp Ophthalmol. 2018;46(3):247–259. doi:10.1111/ceo.13022
  • Warren WH, Kay BA, Zosh WD, Duchon AP, Sahuc S. Optic flow is used to control human walking. Nat Neurosci. 2001;4(2):213–216. doi:10.1038/84054
  • Bowers AR, Ananyev E, Mandel AJ, Goldstein RB, Peli E. Driving with hemianopia: IV. Head scanning and detection at intersections in a simulator. Invest Ophthalmol Vis Sci. 2014;55(3):1540–1548. doi:10.1167/iovs.13-12748
  • Apfelbaum H, Pelah A, Peli E. Heading assessment by “tunnel vision” patients and control subjects standing or walking in a virtual reality environment. ACM Trans Appl Percept. 2007;4(1):8. doi:10.1145/1227134.1227142
  • Peli E, Apfelbaum H, Berson EL, Goldstein RB. The risk of pedestrian collisions with peripheral visual field loss. J Vis. 2016;16(15):5. doi:10.1167/16.15.5
  • Jones PR, Somoskeoy T, Chow-Wing-Bom H, Crabb DP. Seeing other perspectives: evaluating the use of virtual and augmented reality to simulate visual impairments (OpenVisSim). NPJ Digit Med. 2020;3(1):32. doi:10.1038/s41746-020-0242-6
  • Lam AKN, To E, Weinreb RN, et al. Use of virtual reality simulation to identify vision-related disability in patients with glaucoma. JAMA Ophthalmol. 2020;138(5):490–498. doi:10.1001/jamaophthalmol.2020.0392
  • Jacobson S, Voigt W, Parel J-M, et al. Automated light- and dark-adapted perimetry for evaluating retinitis pigmentosa. Ophthalmology. 1986;93(12):1604–1611. doi:10.1016/S0161-6420(86)33522-X
  • Aleman TS, Han G, Serrano LW, et al. Natural history of the central structural abnormalities in choroideremia: a prospective cross-sectional study. Ophthalmology. 2017;124(3):359–373. doi:10.1016/j.ophtha.2016.10.022
  • Roman AJ, Schwartz SB, Aleman TS, et al. Quantifying rod photoreceptor-mediated vision in retinal degenerations: dark-adapted thresholds as outcome measures. Exp Eye Res. 2005;80(2):259–272. doi:10.1016/j.exer.2004.09.008
  • Roman AJ, Cideciyan AV, Aleman TS, Jacobson SG. Full-field stimulus testing (FST) to quantify visual perception in severely blind candidates for treatment trials. Physiol Meas. 2007;28(8):N51–56. doi:10.1088/0967-3334/28/8/N02
  • Aleman TS, Uyhazi KE, Serrano LW, et al. RDH12 mutations cause a severe retinal degeneration with relatively spared rod function. Invest Ophthalmol Vis Sci. 2018;59(12):5225–5236. doi:10.1167/iovs.18-24708
  • Uyhazi K, Aravand P, Bell B, et al. Treatment potential for LCA5-associated leber congenital amaurosis. Invest Ophthalmol Vis Sci. 2020;61(5):30. doi:10.1167/iovs.61.5.30
  • Kolarik AJ, Cirstea S, Pardhan S, Moore BC. A summary of research investigating echolocation abilities of blind and sighted humans. Hear Res. 2014;310:60–68. doi:10.1016/j.heares.2014.01.010
  • Mees L, Upadhyaya S, Kumar P, et al. Validation of a head-mounted virtual reality visual field screening device. J Glaucoma. 2020;29(2):86–91. doi:10.1097/IJG.0000000000001415
  • Walkey HC, Barbur JL. Guest editorial: shedding new light on the twilight zone. Ophthalmic Physiol Opt. 2006;26(3):223–224. doi:10.1111/j.1475-1313.2006.00420.x
  • Walkey HC, Barbur JL, Harlow JA, Hurden A, Moorhead IR, Taylor JA. Effective contrast of colored stimuli in the mesopic range: a metric for perceived contrast based on achromatic luminance contrast. J Opt Soc Am A Opt Image Sci Vis. 2005;22(1):17–28. doi:10.1364/JOSAA.22.000017
  • Walkey HC, Harlow JA, Barbur JL. Characterising mesopic spectral sensitivity from reaction times. Vision Res. 2006;46(25):4232–4243. doi:10.1016/j.visres.2006.08.002
  • Walkey HC, Harlow JA, Barbur JL. Changes in reaction time and search time with background luminance in the mesopic range. Ophthalmic Physiol Opt. 2006;26(3):288–299. doi:10.1111/j.1475-1313.2006.00412.x
  • Zele AJ, Cao D. Vision under mesopic and scotopic illumination. Front Psychol. 2014;5:1594. doi:10.3389/fpsyg.2014.01594
  • Zele AJ, Maynard ML, Feigl B. Rod and cone pathway signaling and interaction under mesopic illumination. J Vis. 2013;13(1):1. doi:10.1167/13.1.21
  • Zele AJ, Maynard ML, Joyce DS, Cao D. Effect of rod-cone interactions on mesopic visual performance mediated by chromatic and luminance pathways. J Opt Soc Am A Opt Image Sci Vis. 2014;31(4):A7–A14. doi:10.1364/JOSAA.31.0000A7
  • Mangione CM, Lee PP, Gutierrez PR, Spritzer K, Berry S, Hays RD. Development of the 25-item national eye institute Visual Function Questionnaire (VFQ-25). Arch Ophthalmol. 2001;119(7):1050–1058. doi:10.1001/archopht.119.7.1050
  • Dougherty BE, Bullimore MA. Comparison of scoring approaches for the NEI VFQ-25 in low vision. Optom Vis Sci. 2010;87(8):543–548. doi:10.1097/OPX.0b013e3181e61bd8
  • Massof RW. An interval-scaled scoring algorithm for visual function questionnaires. Optom Vis Sci. 2007;84(8):E690–705. doi:10.1097/OPX.0b013e31812f5f35
  • Massof RW. The measurement of vision disability. Optom Vis Sci. 2002;79(8):516–552. doi:10.1097/00006324-200208000-00015
  • Massof RW, Ahmadian L. What do different visual function questionnaires measure? Ophthalmic Epidemiol. 2007;14(4):198–204. doi:10.1080/09286580701487883
  • Massof RW, Hsu CT, Baker FH, et al. Visual disability variables. II: the difficulty of tasks for a sample of low-vision patients. Arch Phys Med Rehabil. 2005;86(5):954–967. doi:10.1016/j.apmr.2004.09.017
  • Massof RW, Hsu CT, Baker FH, et al. Visual disability variables. I: the importance and difficulty of activity goals for a sample of low-vision patients. Arch Phys Med Rehabil. 2005;86(5):946–953. doi:10.1016/j.apmr.2004.09.016
  • Massof RW, Rubin GS. Visual function assessment questionnaires. Surv Ophthalmol. 2001;45(6):531–548. doi:10.1016/S0039-6257(01)00194-1
  • Jeter PE, Rozanski C, Massof R, Adeyemo O, Dagnelie G. The PSG. Development of the Ultra-Low Vision Visual Functioning Questionnaire (ULV-VFQ). Transl Vis Sci Technol. 2017;6(3):11. doi:10.1167/tvst.6.3.11
  • Adeyemo O, Jeter PE, Rozanski C, et al. Living with ultra-low vision: an inventory of self-reported visually guided activities by individuals with profound visual impairment. Transl Vis Sci Technol. 2017;6(3):10. doi:10.1167/tvst.6.3.10

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