Ganglion Cell Layer–Inner Plexiform Layer Thickness and Vision Loss in Young Children With Optic Pathway Gliomas

Gu, Sherry; Glaug, Natalie C.; Cnaan, Avital; Packer, Roger J.; Avery, R. A.

doi:10.1167/iovs.13-13119

Cited by 69 publications

(86 citation statements)

References 43 publications

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“…Also, there is an absence of a direct comparison to an established visual field method at first visit of our children, which would confirm or disprove our SVOP results, since there is no commonly available and recognized methodology for children under the age of 5 years. Fortunately, the segmentation of GCL-IPL was shown to be a suitable method to discriminate between children with and without vision loss [9]. SD-OCT remains the most appropriate method for objective analysis of hemianopic defects due to anterior pathway pathologies and thus permits reliable predictions of visual function in children.…”

Section: Discussionmentioning

confidence: 99%

“…An appropriate method to describe morphological changes in the retina of children with hemianopia due to anterior visual pathway pathologies is optical coherence tomography (OCT) [9]. It allows measurements of retinal thickness and boundaries in healthy and affected eyes with high accuracy and reproducibility [9][10][11].…”

mentioning

confidence: 99%

“…It allows measurements of retinal thickness and boundaries in healthy and affected eyes with high accuracy and reproducibility [9][10][11]. Recent studies focused on measuring the peripapillary retinal nerve fiber layer [9,12] and the ganglion cell layer + inner plexiform layer (GCL + IPL = GCL+) of the macular region.…”

mentioning

confidence: 99%

“…Recent studies focused on measuring the peripapillary retinal nerve fiber layer [9,12] and the ganglion cell layer + inner plexiform layer (GCL + IPL = GCL+) of the macular region. GCL+ thickness in the macular area has been shown to be a more accurate parameter for visual function [9,13,14] and demonstrates a more precise relationship with visual acuity and visual field [15][16][17][18].…”

mentioning

confidence: 99%

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Structure-Function Correlation in Hemianopic Vision Loss in Children Aged 3-6 Years Using OCT and SVOP, and Comparison with Adult Eyes

Bowl

Knobloch²,

Schweinfurth³

et al. 2018

Ophthalmic Res

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Purpose: To correlate visual field assessment with saccadic vector optokinetic perimetry (SVOP) in children with ganglion cell loss due to anterior pathway pathologies resulting in hemianopic visual field defects measured with optical coherence tomography (OCT). Methods: 5 young (aged 3-6 years) and 5 adult patients with hemianopia, 10 healthy preschoolers (mean age 4.4 years), and 10 healthy adults (mean age 25.3 years) were tested with SVOP and OCT (focusing on the ganglion cell layer, GCL+). In adults, visual field testing was also performed with static and fundus-controlled perimetry. Results: OCT allowed precise structure analysis and showed a vertical border with GCL+ loss on the hemianopic side in children and adults compared to controls. SVOP showed visual field defects on the hemianopic side in peripheral regions and inadequate results at the parafoveal positions in both groups. In contrast, static and fundus-controlled perimetry showed a clear border in foveal and parafoveal regions. Conclusions: All children underwent SVOP with minimal restrictions, allowing functional evaluation of peripheral visual field positions. Parafoveal positions showed multiple false-positive results. The function-structure relationship is measurable even in young children by using the GCL+ analysis. This combination of novel child-friendly techniques allows collecting objectively measured values and simplifies diagnosis and follow-up in treatment.

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Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 99%

mentioning

confidence: 99%

mentioning

confidence: 99%

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Structure-Function Correlation in Hemianopic Vision Loss in Children Aged 3-6 Years Using OCT and SVOP, and Comparison with Adult Eyes

Bowl

Knobloch²,

Schweinfurth³

et al. 2018

Ophthalmic Res

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“…97 It has been demonstrated that measurements of the RNFL, GCL, and IPL thickness can discriminate between children with and without vision loss from their optic nerve pathway gliomas, thus suggesting that HH-SDOCT can be used as a surrogate marker of visual function in infants and young children. 96,98 Similarly, OCT can be used to diagnose and monitor pediatric pseudotumor cerebri and is potentially useful as a visual prognostic tool in this condition. 99,100 An increased central macular, temporal, and superior RNFL thickness has been described in children affected by pseudotumor cerebri.…”

Section: Optic Nerve Pathologymentioning

confidence: 99%

Pediatric Optical Coherence Tomography in Clinical Practice—Recent Progress

Lee

Proudlock

Gottlob

2016

Invest. Ophthalmol. Vis. Sci.

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Citation: Lee H, Proudlock FA, Gottlob I. Pediatric optical coherence tomography in clinical practice-recent progress. Invest Ophthalmol Vis Sci. 2016;57:OCT69-OCT79. DOI:10.1167/iovs.15-18825 PURPOSE. Optical coherence tomography (OCT) has revolutionized the diagnosis and management of adult retinal and optic nerve disease. Children were deprived of this technology until the recent development of handheld spectral-domain OCT (HH-SDOCT). In this article, we review the applications of OCT imaging in pediatric ophthalmology.METHODS. This study was a review of the literature.RESULTS. The acquisition and interpretation of pediatric tomograms differ significantly from those for adults, with adjustments needed to account for the shorter axial lengths, higher refractive errors, and ongoing retinal and optic nerve development in the pediatric eye. Handheld SDOCT is increasingly being used as an adjunctive diagnostic tool in retinopathy of prematurity (ROP) and nonaccidental injury (NAI) by providing additional morphologic information that is not normally clinically discernible. The role of HH-SDOCT in streamlining diagnosis in infantile nystagmus syndrome, retinal dystrophies, and degenerations has been established. Optical coherence tomography can also help differentiate between pediatric intraocular tumors, for example, hamartomas and retinoblastoma; monitor tumor progression; and monitor treatment response. In addition, HH-SDOCT is establishing its role as a noninvasive monitoring tool in children affected by optic nerve pathology such as glaucoma, optic nerve atrophy and hypoplasia, optic pathway glioma, and pseudotumor cerebri. CONCLUSIONS.Handheld SDOCT can provide novel insights into the natural history of retinal and optic nerve diseases in young children. For example, in achromatopsia and albinism, in vivo OCT studies have provided evidence of altered but ongoing retinal development in early childhood, which suggests that potentially targeting treatment at an earlier age may optimize visual function by normalizing retinal development.

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Insights into optic pathway glioma vision loss from mouse models of neurofibromatosis type 1

Freret

Gutmann

2018

J of Neuroscience Research

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Neurofibromatosis type 1 (NF1) is a common cancer predisposition syndrome caused by mutations in the NF1 gene. The NF1-encoded protein (neurofibromin) is an inhibitor of the oncoprotein RAS and controls cell growth and survival. Individuals with NF1 are prone to developing low-grade tumors of the optic nerves, chiasm, tracts, and radiations, termed optic pathway gliomas (OPGs), which can cause vision loss. A paucity of surgical tumor specimens and of patient-derived xenografts for investigative studies has limited our understanding of human NF1-associated OPG (NF1-OPG). However, mice genetically engineered to harbor Nf1 gene mutations develop optic gliomas that share many features of their human counterparts. These genetically engineered mouse (GEM) strains have provided important insights into the cellular and molecular determinants that underlie mouse Nf1 optic glioma development, maintenance, and associated vision loss, with relevance by extension to human NF1-OPG disease. Herein, we review our current understanding of NF1-OPG pathobiology and describe the mechanisms responsible for tumor initiation, growth, and associated vision loss in Nf1 GEM models. We also discuss how Nf1 GEM and other preclinical models can be deployed to identify and evaluate molecularly targeted therapies for OPG, particularly as they pertain to future strategies aimed at preventing or improving tumor-associated vision loss in children with NF1.

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Ganglion Cell Layer–Inner Plexiform Layer Thickness and Vision Loss in Young Children With Optic Pathway Gliomas

Abstract: Decreased GCL-IPL thickness (

Cited by 69 publications

References 43 publications

Structure-Function Correlation in Hemianopic Vision Loss in Children Aged 3-6 Years Using OCT and SVOP, and Comparison with Adult Eyes

Structure-Function Correlation in Hemianopic Vision Loss in Children Aged 3-6 Years Using OCT and SVOP, and Comparison with Adult Eyes

Pediatric Optical Coherence Tomography in Clinical Practice—Recent Progress

Insights into optic pathway glioma vision loss from mouse models of neurofibromatosis type 1

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