Visual Psychophysics / Physiological Optics - ARVO

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ARVO 2013 Annual Meeting Abstracts by Scientific Section/Group – Visual Psychophysics / Physiological Optics600μm and 1200μm. In two very late stage patients with sunset-glowfundus at more than 2 years after the onset, analysis of AO showed anirregularity in cone density at both 600μm and 1200μm eccentricities,even though patients’ visual acuity were better than 20/20.Conclusions: We have found that the photoreceptor damage remainlonger even after serous retinal detachment was resolved with OCT inthe convalescent and chronic stage VKH patients. Combinedexaminations using AO and SD-OCT may be useful in betterunderstanding and managing the ophthalmic consequences of VHKdisease.Commercial Relationships: Kiyoko Gocho, None; SachikoKikuchi, None; Takenori Kabuto, None; Hisatomo Takahashi,None; Shuhei Kameya, None; Kunihiko Yamaki, None; HiroshiTakahashi, NoneProgram Number: 3443 Poster Board Number: C0164Presentation Time: 11:00 AM - 12:45 PMSpectral-Domain Optical Coherence Tomographic Findings inAcute Zonal Occult Outer Retinopathy (AZOOR)-complex atInitial Visit to the HospitalYoshitsugu Matsui 1 , Mai Miyata 1 , Hisashi Matsubara 1 , Shinji Ueno 2 ,Shu Kachi 2 , Yasuki Ito 2 , Hiroko Terasaki 2 , Mineo Kondo 1 .1 Opthalmology, Mie University Graduate School of Medicine, Tsu,Japan; 2 Opthalmology, Nagoya University Graduate School ofMedicine, Nagoya, Japan.Purpose: To study outer retinal structural changes in acute zonaloccult outer retinopathy (AZOOR)-complex by using spectraldomainoptical coherence tomography (SD-OCT) at the initial visit tothe hospital.Methods: Twenty-two eyes of 22 patients (four male and 18 female;age, 19-69) with AZOOR-complex, whose periods from symptomonset to initial visit to the hospital were within six months, wereincluded in this study. The continuity of the external limitingmembrane (ELM), photoreceptor inner/outer segment (IS/OS)junction, and cone outer segment tip (COST) were evaluated usingSD-OCT at their initial visit to the hospital. These three lines weredivided into three categories of “continuous”, “discontinuous” and“absent”.Results: At the retinal area of visual field defects, the ELM wascontinuous in eight eyes, discontinuous in five eyes, and absent innine eyes. The IS/OS junction was continuous in one eye,discontinuous in nine eyes, and absent in 12 eyes. The COST linewas absent in all 22 eyes. In thee of 22 eyes, well-demarcated hyperrefractivematerials were present between the retinal pigmentepithelium (RPE) and ERM, and these materials graduallydisappeared within three months after the initial visit.Conclusions: The SD-OCT findings in AZOOR-complex at theinitial visit were variable. The structure of ELM was most likely to bepreserved, and COST line most severely affected. Our findingssupported the idea that outer retinal structure was more vulnerable atthe early stages of AZOOR-complex.Commercial Relationships: Yoshitsugu Matsui, None; MaiMiyata, None; Hisashi Matsubara, None; Shinji Ueno, None; ShuKachi, None; Yasuki Ito, None; Hiroko Terasaki, None; MineoKondo, NoneSongEun Lee 1 , Sung Pyo Park 1, 2 , Eun Jin Bae 2 , Stephen H. Tsang 1 ,Stanley Chang 1 . 1 Ophthalmology, Harkness Eye Institute, ColumbiaUniversity, New York, NY; 2 Ophthalmology, Kangdong SacredHeart Hospital, Hallym University Medical Center, Seoul, Republicof Korea.Purpose: To evaluate structural damage of photoreceptors in apatient with chloroquine/hydroxychloroquine maculopathy and therole of adaptive optics scanning laser ophthalmoscopy in screening ofchloroquine/hydroxychloroquine maculopathyMethods: A 53-year-old Asian woman who had been treated withhydroxychloroquine and chloroquine for lupus erythematosus,developed bull's eye retinopathy. Humphrey visual field, fundusautofluorescence, and adaptive optics scanning laser ophthalmoscopywere performed. The area with disrupted cone in AO-SLO imageswere matched with visual field test results.Results: Adaptive optics scanning laser ophthalmoscopy (AO-SLO)showed patch cone mosaic lesions, where cones were missing or lost.Additionally, cones that were present were asymmetrical in size andshape and varied in brightness. Disrupted cone AO-SLO images werematched with visual field test results, and functional deficits weregiven a precise location on the montage. This allowed functionalchanges (on HVF) to be correlated with histological findings.Conclusions: In the present study, bull’s eye maculopathy developedas a result of chronic use of antimalarial agents. The patient wasstudied with various imaging techniques, including AO-SLO. Wedemonstrated not only the disrupted microarchitecture of thephotoreceptors but also a correlation between functional andstructural defects associated with chloroquine retinopathy. Theadaptive optics scanning laser ophthalmoscopy will provide betterunderstanding of retinal damages in antimalarial agent-associatedretinopathy.Program Number: 3444 Poster Board Number: C0165Presentation Time: 11:00 AM - 12:45 PMEvidence of Retinal Damage in Chloroquine/HydroxychloroquineMaculopathy as Revealed by High Resolution Imaging: A CaseReport Utilizing Adaptive Optics Scanning LaserOphthalmoscopy©2013, Copyright by the Association for Research in Vision and Ophthalmology, Inc., all rights reserved. Go to iovs.org to access the version of record. For permissionto reproduce any abstract, contact the ARVO Office at arvo@arvo.org.
ARVO 2013 Annual Meeting Abstracts by Scientific Section/Group – Visual Psychophysics / Physiological OpticsCommercial Relationships: SongEun Lee, None; Sung Pyo Park,None; Eun Jin Bae, None; Stephen H. Tsang, None; StanleyChang, Alcon Laboratories (C), Alimera Sciences (C)Program Number: 3445 Poster Board Number: C0166Presentation Time: 11:00 AM - 12:45 PMAdaptive Optics Scanning Laser Ophthalmoscopy and HighResolution Imaging in Autosomal Dominant Retinitis PigmentosaCaused by a Novel PRPF31 Nonsense MutationJessica I. Morgan 1, 2 , Meera Sivalingam 1 , Grace K. Han 1 , Albert M.Maguire 1, 2 , Brian J. Forbes 1, 2 , Xiaowu Gai 3 , Eric A. Pierce 4 , JeanBennett 1, 2 , Daniel C. Chung 1, 2 . 1 Ophthalmology, University ofPennsylvania, Philadelphia, PA; 2 Ophthalmology, Children's Hospitalof Philadelphia, Philadelphia, PA; 3 Molecular Pharmacology andTherapeutics, Loyola University Chicago, Chicago, IL; 4 OcularGenomics Institute, Department of Ophthalmology, MassachusettsEye and Ear Infirmary, Harvard Medical School, Boston, MA.Purpose: To study cone photoreceptor density and retinalultrastructure in autosomal dominant retinitis pigmentosa (adRP)using adaptive optics scanning laser ophthalmoscopy (AOSLO) andspectral domain optical coherence tomography (OCT) and tocompare the results with clinical findings and geneticcharacterization.Methods: Exome sequencing was carried out in individuals negativefor previously known mutations causing adRP. The DNA variantswere confirmed in CLIA labs. Patients underwent ophthalmicexamination, visual acuity (VA), Humphrey and Goldman visualfield testing. Retinal images at several locations within 1.5mm offixation were obtained using an AOSLO developed by Canon, Inc.Infrared and color fundus photos, OCT and autofluorescence (AF)images were also obtained.Results: For all patients, peripheral pigmentary clumping and bonespicule retinopathy were observed on ophthalmic examination. Oneindividual was followed in the clinic for over 35 years. His VA wentfrom 20/200 at age 36 to 20/500 OD and hand motion OS at age 73.VA was reduced: 20/60, 20/40, 20/25 for patients aged 44, 16 and 11respectively in the second family. Goldman visual fields showedrestricted peripheral fields with remaining central vision. Humphreyvisual field showed no reduction of visual threshold in the centralretina for the 16 and 11 y.o., but reduced sensitivity in the 44 y.o.OCT for the 11 y.o. revealed central intact photoreceptors withperipheral thinning. The 16 y.o. had macular edema OU. AF revealeda hyper-AF ring within the macula region. As well, the 44 y.o.exhibited clumps of hypo-AF in the periphery corresponding toregions of degenerated retinal pigment epithelium. AOSLO imagingof the central macula revealed intact cone photoreceptor mosaics, andcone density fell within normal limits. Results of genetic testing (inboth families) revealed a PRPF31 c.1060 C>T mutation(p.Arg354Stop) which segregated with the phenotype and wasconsistent with the diagnosis of adRP.Conclusions: Here we report a novel PRPF31 mutation causingadRP in two apparently unrelated families. AOSLO imaging allowsvisualization of the cone mosaic within the remaining central retina inpatients with adRP. AOSLO in combination with other imagingmodalities will be useful for studying the mechanisms of diseaseprogression for degenerations including adRP.Commercial Relationships: Jessica I. Morgan, Canon (F), Optos(F); Meera Sivalingam, Canon Inc (F); Grace K. Han, Canon Inc.(F), Optos PLC (F); Albert M. Maguire, None; Brian J. Forbes,None; Xiaowu Gai, None; Eric A. Pierce, None; Jean Bennett,Gensight Biologics (S); Daniel C. Chung, NoneSupport: Earl and Brenda Shapiro Leber's Congenital AmaurosisResearch Initiative, Foundation Fighting Blindness, Penn GenomeFrontiers Institute, National Institute of Health EY019861,NIHEY012910, F.M. Kirby Foundation, Mackall Trust, Reserach toPrevent Blindness, Canon, Inc. Kathleen Marshall and SarahMcCague.Program Number: 3446 Poster Board Number: C0167Presentation Time: 11:00 AM - 12:45 PMCone Abnormalities in Fundus Albipunctatus Associated withRDH5 Mutation Using Adaptive Optics Scanning LaserOphthalmoscopySotaro Ooto, Yukiko Makiyama, Masanori Hangai, Ken Ogino,Norimoto Gotoh, Akio Oishi, Nagahisa Yoshimura. Ophthalmology& Visual Sciences, Kyoto University Graduate School of Medicine,Kyoto, Japan.Purpose: To asses macular photoreceptor abnormalities in eyes withfundus albipunctatus associated with RDH5 mutation using adaptiveoptics scanning laser ophthalmoscopy (AO-SLO).Methods: Ten eyes of 5 patients with fundus ablipunctatusassociated with RDH5 mutation (4 men and 1 woman from 4families; mean age, 29.6 y; range, 15-64y) and 11 eyes of 11volunteers underwent a full ophthalmologic examination, colorfundus photography, microperimetry, spectral-domain opticalcoherence tomography (SD-OCT), and imaging with a prototype AO-SLO system. Cone density and spatial organization of the conemosaic were assessed using AO-SLO images.Results: Four patients from 3 families had the same mutation inRDH5, (c.928delC/ insGAAG). One patient had another mutation inRDH5 (c.718delG), which was the novel mutation of RDH5 gene.AO-SLO revealed the presence of small patchy dark areasrepresenting cone loss in the macula of all eyes with fundusalbipunctatus, which was true even in eyes for which SD-OCT didnot reveal any visible defects in the photoreceptor layer. AO-SLOand SD-OCT showed microhole-like photoreceptor defect in thefovea in 1 eye. In eyes with fundus albipunctatus, cone density wassignificantly lower in each area at 0.5 mm and 1.0 mm from thecenter of the fovea compared to normal eyes (P = 0.014 for both). At0.5 mm and 1.0 mm from the center of the fovea, a decreased numberof cones had 6 neighbors in eyes with fundus albipunctatus (P =0.022 and 0.002). The ratio of observed mean nearest neighbordistance (NND) for each subject divided by expected NND(computed assuming a perfectly hexagonal lattice of cones with adensity equal to that observed for a given subject) was significantlylower for eyes with fundus albipunctatus than for normal eyes at 0.5©2013, Copyright by the Association for Research in Vision and Ophthalmology, Inc., all rights reserved. Go to iovs.org to access the version of record. For permissionto reproduce any abstract, contact the ARVO Office at arvo@arvo.org.
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