Measurement and Reproducibility of Preserved Ellipsoid Zone Area and Preserved Retinal Pigment Epithelium Area in Eyes With Choroideremia - 27/09/17

Doi : 10.1016/j.ajo.2017.05.002

Amir H. Hariri ^a,^b, Swetha B. Velaga ^a, Aniz Girach ^c, Michael S. Ip ^a,^b, Phuc V. Le ^a,^b, Byron L. Lam ^d, M. Dominik Fischer ^e, Eeva-Marja Sankila ^f, Mark E. Pennesi ^g, Frank G. Holz ^h, Robert E. MacLaren ^i,^j, David G. Birch ^k, Carel B. Hoyng ^l, Ian M. MacDonald ^m, Graeme C. Black ⁿ, Stephen H. Tsang ^o, Neil M. Bressler ^p, Michael Larsen ^q, Michael B. Gorin ^b, Andrew R. Webster ^r,^s, SriniVas R. Sadda ^a,^b,^⁎
for the
Natural History of the Progression of Choroideremia (NIGHT) Study Group
^a Doheny Image Reading Center, Doheny Eye Institute, Los Angeles, California
^b Department of Ophthalmology, David Geffen School of Medicine of the University of California-Los Angeles, Los Angeles, California
^c NightstaRx Ltd, London, United Kingdom
^d Department of Ophthalmology, Bascom Palmer Eye Institute, University of Miami Miller School of Medicine, Miami, Florida
^e Centre for Ophthalmology, University of Tübingen, Tübingen, Germany
^f Helsinki University Eye Hospital, Finland
^g Casey Eye Institute, Oregon Health & Science University, Portland, Oregon
^h Department of Ophthalmology, University of Bonn, Bonn, Germany
ⁱ Nuffield Laboratory of Ophthalmology, Department of Clinical Neurosciences, University of Oxford and Oxford University Eye Hospital, NHS Foundation Trust, NIHR Biomedical Research Centre, Oxford, United Kingdom
^j Moorfields Eye Hospital, NHS Foundation Trust, NIHR Biomedical Research Centre, London, United Kingdom
^k Retina Foundation of the Southwest, Dallas, Texas
^l Department of Ophthalmology, Radboud University Medical Center, Nijmegen, Netherlands
^m Department of Ophthalmology and Visual Sciences, University of Alberta, Edmonton, Alberta, Canada
ⁿ Manchester Centre for Genomic Medicine, Central Manchester University Hospitals NHS Foundation Trust, Manchester Academic Health Sciences Centre, St Mary's Hospital, Manchester, United Kingdom
^o Departments of Ophthalmology and of Pathology and Cell Biology, Columbia University, New York, New York
^p Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland
^q Department of Ophthalmology, Rigshospitalet and University of Copenhagen, Copenhagen, Denmark
^r Moorfields Eye Hospital NHS Foundation Trust, London, United Kingdom
^s UCL Institute of Ophthalmology, London, United Kingdom

^∗Inquiries to SriniVas R. Sadda, President and Chief Scientific Officer, Doheny Eye Institute, David Geffen School of Medicine, University of California, Los Angeles, CaliforniaPresident and Chief Scientific OfficerDoheny Eye InstituteDavid Geffen School of MedicineUniversity of CaliforniaLos AngelesCalifornia

Abstract

Purpose

To identify valid and reproducible methods for quantifying anatomic outcome measures for eyes with choroideremia (CHM) in clinical trials.

Design

Reliability analysis study.

Methods

In this multicenter study, patients with confirmed genetic diagnosis of CHM were enrolled. All cases underwent spectral-domain optical coherence tomography (SDOCT) and fundus autofluorescence (FAF) imaging. Two graders independently delineated boundaries of preserved autofluorescence (PAF) and preserved ellipsoid zone (EZ) on FAF and OCT images, respectively. The results of the 2 independent gradings of both FAF and OCT images were compared to assess the reproducibility of the grading methods.

Results

A total of 148 eyes from 75 cases were included. In 21% of eyes PAF and in 43% of eyes preserved EZ had extended beyond the image capture area. After exclusion of these eyes and low-quality images, 114 FAF and 77 OCT images were graded. The mean PAF areas from 2 independent gradings were 3.720 ± 3.340 mm² and 3.692 ± 3.253 mm², respectively. Intraclass correlation coefficient (ICC) for these gradings was 0.996. The mean preserved EZ areas from 2 independent gradings were 2.746 ± 2.319 mm² and 2.858 ± 2.446 mm², respectively. ICC for these gradings was 0.991.

Conclusions

Quantifying preserved retinal pigment epithelium and EZ areas on FAF and OCT images, respectively, in CHM patients is highly reproducible. These variables would be potential anatomic outcome measures for CHM clinical trials and could be studied and tracked longitudinally in choroideremia.

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Vol 179

P. 110-117 - juillet 2017 Retour au numéro

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Measurement and Reproducibility of Preserved Ellipsoid Zone Area and Preserved Retinal Pigment Epithelium Area in Eyes With Choroideremia - 27/09/17

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