Can structural grading of foveal hypoplasia predict future vision in infantile nystagmus? A longitudinal study
Can structural grading of foveal hypoplasia predict future vision in infantile nystagmus? A longitudinal study
Purpose: To evaluate structural grading and quantitative segmentation of foveal hypoplasia using handheld OCT, versus preferential looking (PL), as predictors of future vision in preverbal children with infantile nystagmus. Design: Longitudinal cohort study. Participants: Forty-two patients with infantile nystagmus (19 with albinism, 17 with idiopathic infantile nystagmus, and 6 with achromatopsia) were examined. Methods: Spectral-domain handheld OCT was performed in preverbal children up to 36 months of age. Foveal tomograms were graded using our 6-point grading system for foveal hypoplasia and were segmented for quantitative analysis: photoreceptor length, outer segment (OS) length, and foveal developmental index (FDI; a ratio of inner layers versus total foveal thickness). Patients were followed up until they could perform chart visual acuity (VA) testing. Data were analyzed using linear mixed regression models. Visual acuity predicted by foveal grading was compared with prediction by PL, the current gold standard for visual assessment in infants and young children. Main Outcome Measures: Grade of foveal hypoplasia, quantitative parameters (photoreceptor length, OS length, FDI), and PL VA were obtained in preverbal children for comparison with future chart VA outcomes. Results: We imaged 81 eyes from 42 patients with infantile nystagmus of mean age 19.8 months (range, 0.9–33.4 months; standard deviation [SD], 9.4 months) at the first handheld OCT scan. Mean follow-up was 44.1 months (range, 18.4–63.2 months; SD, 12.0 months). Structural grading was the strongest predictor of future VA (grading: r = 0.80, F = 67.49, P < 0.0001) compared with quantitative measures (FDI: r = 0.74, F = 28.81, P < 0.001; OS length: r = 0.65; F = 7.94, P < 0.008; photoreceptor length: r = 0.65; F = 7.94, P < 0.008). Preferential looking was inferior to VA prediction by foveal grading (PL: r = 0.42, F = 3.12, P < 0.03). Conclusions: Handheld OCT can predict future VA in infantile nystagmus. Structural grading is a better predictor of future VA than quantitative segmentation and PL testing. Predicting future vision may avert parental anxiety and may optimize childhood development.
492-500
Rufai, Sohaib R.
b8a329cd-17a2-4b8d-967e-3895cdb8a7be
Thomas, Mervyn
43580622-2633-4694-960c-098d045d6144
Purohit, Ravi
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Bunce, Catey
7c13264f-265d-4b0f-8a3b-e29fefd55df2
Lee, Helena
5d36fd1e-9334-4db5-b201-034d147133fb
Proudlock, Frank
b871e8dd-b6ff-47b3-bf00-3da75c685947
Gottlob, Irene
a504e420-9d7f-4dd5-8e3e-ad48a24a8f9e
April 2020
Rufai, Sohaib R.
b8a329cd-17a2-4b8d-967e-3895cdb8a7be
Thomas, Mervyn
43580622-2633-4694-960c-098d045d6144
Purohit, Ravi
35a65d7f-5133-41ce-a2f8-0fc40b137616
Bunce, Catey
7c13264f-265d-4b0f-8a3b-e29fefd55df2
Lee, Helena
5d36fd1e-9334-4db5-b201-034d147133fb
Proudlock, Frank
b871e8dd-b6ff-47b3-bf00-3da75c685947
Gottlob, Irene
a504e420-9d7f-4dd5-8e3e-ad48a24a8f9e
Rufai, Sohaib R., Thomas, Mervyn, Purohit, Ravi, Bunce, Catey, Lee, Helena, Proudlock, Frank and Gottlob, Irene
(2020)
Can structural grading of foveal hypoplasia predict future vision in infantile nystagmus? A longitudinal study.
Ophthalmology, 127 (4), .
(doi:10.1016/j.ophtha.2019.10.037).
Abstract
Purpose: To evaluate structural grading and quantitative segmentation of foveal hypoplasia using handheld OCT, versus preferential looking (PL), as predictors of future vision in preverbal children with infantile nystagmus. Design: Longitudinal cohort study. Participants: Forty-two patients with infantile nystagmus (19 with albinism, 17 with idiopathic infantile nystagmus, and 6 with achromatopsia) were examined. Methods: Spectral-domain handheld OCT was performed in preverbal children up to 36 months of age. Foveal tomograms were graded using our 6-point grading system for foveal hypoplasia and were segmented for quantitative analysis: photoreceptor length, outer segment (OS) length, and foveal developmental index (FDI; a ratio of inner layers versus total foveal thickness). Patients were followed up until they could perform chart visual acuity (VA) testing. Data were analyzed using linear mixed regression models. Visual acuity predicted by foveal grading was compared with prediction by PL, the current gold standard for visual assessment in infants and young children. Main Outcome Measures: Grade of foveal hypoplasia, quantitative parameters (photoreceptor length, OS length, FDI), and PL VA were obtained in preverbal children for comparison with future chart VA outcomes. Results: We imaged 81 eyes from 42 patients with infantile nystagmus of mean age 19.8 months (range, 0.9–33.4 months; standard deviation [SD], 9.4 months) at the first handheld OCT scan. Mean follow-up was 44.1 months (range, 18.4–63.2 months; SD, 12.0 months). Structural grading was the strongest predictor of future VA (grading: r = 0.80, F = 67.49, P < 0.0001) compared with quantitative measures (FDI: r = 0.74, F = 28.81, P < 0.001; OS length: r = 0.65; F = 7.94, P < 0.008; photoreceptor length: r = 0.65; F = 7.94, P < 0.008). Preferential looking was inferior to VA prediction by foveal grading (PL: r = 0.42, F = 3.12, P < 0.03). Conclusions: Handheld OCT can predict future VA in infantile nystagmus. Structural grading is a better predictor of future VA than quantitative segmentation and PL testing. Predicting future vision may avert parental anxiety and may optimize childhood development.
Text
Ophthalmology Manuscript CLEAN 15-10-19
- Accepted Manuscript
More information
Accepted/In Press date: 28 October 2019
e-pub ahead of print date: 4 November 2019
Published date: April 2020
Additional Information:
Funding Information:
Supported by the Medical Research Council, United Kingdom (grant nos.: MR/N004566/1 and MR/J004189/1); Fight for Sight, United Kingdom (grant nos.: 24NN181 and 5009/5010); Ulverscroft Foundation, United Kingdom; Nystagmus Network, United Kingdom; and the National Institute for Health Research (Academic Clinical Fellowship [S.R.R.], Academic Clinical Lectureships [M.G.T., H.L.], and Biomedical Research Centre based at Guy's and St Thomas? NHS Foundation Trust and King's College London [C.B.]). The sponsor or funding organization had no role in the design or conduct of this research. The views expressed are those of the authors and not necessarily those of the National Health Service, the National Institute for Health Research, or the Department of Health.
Publisher Copyright:
© 2019 American Academy of Ophthalmology
Identifiers
Local EPrints ID: 435435
URI: http://eprints.soton.ac.uk/id/eprint/435435
ISSN: 0161-6420
PURE UUID: 1115ff3e-5df4-4df0-bf1e-d43739ccd829
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Date deposited: 06 Nov 2019 17:30
Last modified: 17 Mar 2024 03:37
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Contributors
Author:
Sohaib R. Rufai
Author:
Mervyn Thomas
Author:
Ravi Purohit
Author:
Catey Bunce
Author:
Frank Proudlock
Author:
Irene Gottlob
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