Measurement and Reproducibility of Preserved Ellipsoid Zone Area and Preserved Retinal Pigment Epithelium Area in Eyes With Choroideremia

Amir H Hariri; Swetha B Velaga; Aniz Girach; Michael S Ip; Phuc V Le; Byron L Lam; M Dominik Fischer; Eeva-Marja Sankila; Mark E Pennesi; Frank G Holz; Robert E MacLaren; David G Birch; Carel B Hoyng; Ian M MacDonald; Graeme C Black; Stephen H Tsang; Neil M Bressler; Michael Larsen; Michael B Gorin; Andrew R Webster; SriniVas R Sadda

doi:10.1016/j.ajo.2017.05.002

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Measurement and Reproducibility of Preserved Ellipsoid Zone Area and Preserved Retinal Pigment Epithelium Area in Eyes With Choroideremia

Journal article

Open access

Peer reviewed

Measurement and Reproducibility of Preserved Ellipsoid Zone Area and Preserved Retinal Pigment Epithelium Area in Eyes With Choroideremia

Amir H Hariri, Swetha B Velaga, Aniz Girach, Michael S Ip, Phuc V Le, Byron L Lam, M Dominik Fischer, Eeva-Marja Sankila, Mark E Pennesi, Frank G Holz, …

American journal of ophthalmology, Vol.179, pp.110-117

2017-07

DOI: https://doi.org/10.1016/j.ajo.2017.05.002

PMID: 28499705

Abstract

Choroideremia - physiopathology

Reproducibility of Results

Choroideremia - diagnosis

Fluorescein Angiography - methods

Humans

Middle Aged

Male

Tomography, Optical Coherence - methods

Visual Acuity

Retinal Pigment Epithelium - pathology

Female

Aged

Ophthalmoscopy - methods

Fundus Oculi

To identify valid and reproducible methods for quantifying anatomic outcome measures for eyes with choroideremia (CHM) in clinical trials. Reliability analysis study. 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. 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 mm and 3.692 ± 3.253 mm , 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 mm and 2.858 ± 2.446 mm , respectively. ICC for these gradings was 0.991. 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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Collaboration types: Domestic collaboration; International collaboration
Citation topics: 1 Clinical & Life Sciences; 1.36 Ophthalmology; 1.36.212 Retina
Web Of Science research areas: Ophthalmology
ESI research areas: Clinical Medicine

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Title: Measurement and Reproducibility of Preserved Ellipsoid Zone Area and Preserved Retinal Pigment Epithelium Area in Eyes With Choroideremia
Creators: Amir H Hariri - Doheny Image Reading Center, Doheny Eye Institute, Los Angeles, California; Department of Ophthalmology, David Geffen School of Medicine of the University of California-Los Angeles, Los Angeles, California
Swetha B Velaga - Doheny Image Reading Center, Doheny Eye Institute, Los Angeles, California
Aniz Girach - NightstaRx Ltd, London, United Kingdom
Michael S Ip - Doheny Image Reading Center, Doheny Eye Institute, Los Angeles, California; Department of Ophthalmology, David Geffen School of Medicine of the University of California-Los Angeles, Los Angeles, California
Phuc V Le - Doheny Image Reading Center, Doheny Eye Institute, Los Angeles, California; Department of Ophthalmology, David Geffen School of Medicine of the University of California-Los Angeles, Los Angeles, California
Byron L Lam - Department of Ophthalmology, Bascom Palmer Eye Institute, University of Miami Miller School of Medicine, Miami, Florida
M Dominik Fischer - Centre for Ophthalmology, University of Tübingen, Tübingen, Germany
Eeva-Marja Sankila - Helsinki University Eye Hospital, Finland
Mark E Pennesi - Casey Eye Institute, Oregon Health & Science University, Portland, Oregon
Frank G Holz - Department of Ophthalmology, University of Bonn, Bonn, Germany
Robert E MacLaren - 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; Moorfields Eye Hospital, NHS Foundation Trust, NIHR Biomedical Research Centre, London, United Kingdom
David G Birch - Retina Foundation of the Southwest, Dallas, Texas
Carel B Hoyng - Department of Ophthalmology, Radboud University Medical Center, Nijmegen, Netherlands
Ian M MacDonald - Department of Ophthalmology and Visual Sciences, University of Alberta, Edmonton, Alberta, Canada
Graeme C Black - Manchester Centre for Genomic Medicine, Central Manchester University Hospitals NHS Foundation Trust, Manchester Academic Health Sciences Centre, St Mary's Hospital, Manchester, United Kingdom
Stephen H Tsang - Departments of Ophthalmology and of Pathology and Cell Biology, Columbia University, New York, New York
Neil M Bressler - Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland
Michael Larsen - Department of Ophthalmology, Rigshospitalet and University of Copenhagen, Copenhagen, Denmark
Michael B Gorin - Department of Ophthalmology, David Geffen School of Medicine of the University of California-Los Angeles, Los Angeles, California
Andrew R Webster - Moorfields Eye Hospital NHS Foundation Trust, London, United Kingdom; UCL Institute of Ophthalmology, London, United Kingdom
SriniVas R Sadda - Doheny Image Reading Center, Doheny Eye Institute, Los Angeles, California; Department of Ophthalmology, David Geffen School of Medicine of the University of California-Los Angeles, Los Angeles, California. Electronic address: ssadda@doheny.org
Publication Details: American journal of ophthalmology, Vol.179, pp.110-117
Publisher: United States
Grant note: R01 EY009076 / NEI NIH HHS EME/12/66/35 / Department of Health
Academic Unit: Miller School of Medicine; UMMG Department of Ophthalmology
Language: English
Resource Type: Journal article
PMID: 28499705
Record Identifier: 991031575944602976