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

Natural History of the Progression of Choroideremia (NIGHT) Study Group

Research output: Contribution to journalArticle

25 Citations (Scopus)

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.

Original languageEnglish (US)
Pages (from-to)110-117
Number of pages8
JournalAmerican Journal of Ophthalmology
Volume179
DOIs
StatePublished - Jul 1 2017

Fingerprint

Choroideremia
Retinal Pigment Epithelium
Outcome Assessment (Health Care)
Clinical Trials
Optical Imaging
Optical Coherence Tomography
Multicenter Studies

ASJC Scopus subject areas

  • Ophthalmology

Cite this

Measurement and Reproducibility of Preserved Ellipsoid Zone Area and Preserved Retinal Pigment Epithelium Area in Eyes With Choroideremia. / Natural History of the Progression of Choroideremia (NIGHT) Study Group.

In: American Journal of Ophthalmology, Vol. 179, 01.07.2017, p. 110-117.

Research output: Contribution to journalArticle

@article{d7e850c994e34c849aba313fc8f5c1f6,
title = "Measurement and Reproducibility of Preserved Ellipsoid Zone Area and Preserved Retinal Pigment Epithelium Area in Eyes With Choroideremia",
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.",
author = "{Natural History of the Progression of Choroideremia (NIGHT) Study Group} and Hariri, {Amir H.} and Velaga, {Swetha B.} and Aniz Girach and Ip, {Michael S.} and Le, {Phuc V.} and Lam, {Byron L.} and Fischer, {M. Dominik} and Sankila, {Eeva Marja} and Mark Pennesi and Holz, {Frank G.} and MacLaren, {Robert E.} and Birch, {David G.} and Hoyng, {Carel B.} and MacDonald, {Ian M.} and Black, {Graeme C.} and Tsang, {Stephen H.} and Bressler, {Neil M.} and Michael Larsen and Gorin, {Michael B.} and Webster, {Andrew R.} and Sadda, {Srini Vas R.}",
year = "2017",
month = "7",
day = "1",
doi = "10.1016/j.ajo.2017.05.002",
language = "English (US)",
volume = "179",
pages = "110--117",
journal = "American Journal of Ophthalmology",
issn = "0002-9394",
publisher = "Elsevier USA",

}

TY - JOUR

T1 - Measurement and Reproducibility of Preserved Ellipsoid Zone Area and Preserved Retinal Pigment Epithelium Area in Eyes With Choroideremia

AU - Natural History of the Progression of Choroideremia (NIGHT) Study Group

AU - Hariri, Amir H.

AU - Velaga, Swetha B.

AU - Girach, Aniz

AU - Ip, Michael S.

AU - Le, Phuc V.

AU - Lam, Byron L.

AU - Fischer, M. Dominik

AU - Sankila, Eeva Marja

AU - Pennesi, Mark

AU - Holz, Frank G.

AU - MacLaren, Robert E.

AU - Birch, David G.

AU - Hoyng, Carel B.

AU - MacDonald, Ian M.

AU - Black, Graeme C.

AU - Tsang, Stephen H.

AU - Bressler, Neil M.

AU - Larsen, Michael

AU - Gorin, Michael B.

AU - Webster, Andrew R.

AU - Sadda, Srini Vas R.

PY - 2017/7/1

Y1 - 2017/7/1

N2 - 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.

AB - 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.

UR - http://www.scopus.com/inward/record.url?scp=85019601876&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=85019601876&partnerID=8YFLogxK

U2 - 10.1016/j.ajo.2017.05.002

DO - 10.1016/j.ajo.2017.05.002

M3 - Article

C2 - 28499705

AN - SCOPUS:85019601876

VL - 179

SP - 110

EP - 117

JO - American Journal of Ophthalmology

JF - American Journal of Ophthalmology

SN - 0002-9394

ER -