Intravitreal Ranibizumab for Choroidal Neovascularization Secondary to Gyrate Atrophy in a Young Patient: A Multimodal Imaging Analysis

Chatziralli, Irini; Theodossiadis, George; Emfietzoglou, Ioannis; Theodossiadis, Panagiotis

doi:10.5301/ejo.5000660

Cited by 11 publications

(6 citation statements)

References 8 publications

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“…1,49 Fluorescein angiography may also demonstrate leakage in cases of GA that are complicated by subretinal choroidal neovascularisation. 50,51 The evaluation of GA can be also performed and followed up using ultra-widefield imaging which has been found to allow superior imaging of the peripheral retina simultaneously with the macula. 22,52 Electroretinography (ERG) may reveal diminished a and b wave responses from both cones and rods which may become undetectable in advanced disease, 53 however, a nearly normal ERG recording in association with GA has also been previously reported following long-term follow-up.…”

Section: Investigationsmentioning

confidence: 99%

“…5,77 Pars plana vitrectomy can be performed for the treatment of macular holes, 44 rhegmatogenous retinal detachments, 78 and vitreous hemorrhage, 79 while intravitreal injection of anti-vascular endothelial growth factor agents can be used for the treatment of choroidal neovascularisation. 45,50 Finally, the use of low-vision aids may be of benefit to some patients with advanced disease, however, the diffuse chorioretinal atrophy that may be present in late disease and involves the macula as well as the retinal periphery may limit the options available for these patients. Although there is scarce information on the use of lowvision aids for patients with GA, its symptoms and unaltered natural course are similar to retinitis pigmentosa on which more studies have been performed.…”

Section: Treatmentmentioning

confidence: 99%

Section: Treatmentmentioning

confidence: 99%

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Gyrate Atrophy of the Choroid and Retina: A Review

Elnahry

2021

European Journal of Ophthalmology

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Gyrate atrophy (GA) of the choroid and retina is a rare autosomal recessive genetic condition characterized by elevation of the plasma level of the amino acid ornithine due to deficiency of the enzyme ornithine ketoacid aminotransferase. Accumulation of ornithine occurs in various body tissues but leads primarily to characteristic ophthalmic manifestations including myopia, cataract, progressive chorioretinal atrophy, and macular changes. Patients usually present with night blindness that starts in the first decade of life followed by visual field constriction and eventually diminution of the central visual acuity and blindness. The condition has been reported worldwide and its differential diagnosis is broad and includes choroideremia and retinitis pigmentosa. Treatment currently depends on life-long dietary modifications including restriction of the amino acid arginine in diet. This article describes in detail the pathogenesis, clinical features, multimodal imaging findings, and treatment options for GA of the choroid and retina and its complications.

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Section: Investigationsmentioning

confidence: 99%

Section: Treatmentmentioning

confidence: 99%

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Gyrate Atrophy of the Choroid and Retina: A Review

Elnahry

2021

European Journal of Ophthalmology

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“… 35 It has been reported that the combination of arginine restricted diet can improve the visual acuity of patients with choroidal neovascularization (CNV) after the intravitreal injection of ranibizumab. 36 L-arginine contributed additional effects on exercise-induced angiogenesis possibly by enhancing VEGF expression in the hind-leg muscles and heart. 37 On the other hand, the enhancement of NO synthesis pathway is characteristic of pro-inflammatory M1-like macrophages, whereas the expression of arginase characterizes M2 macrophages, and arginase hydrolyzes arginine to ornithine and urea while limiting the synthesis of NO derived from arginine.…”

Section: Discussionmentioning

confidence: 96%

Metabolomics Analyses of Mouse Retinas in Oxygen-Induced Retinopathy

Zhou

Tan

Zou

et al. 2021

Invest. Ophthalmol. Vis. Sci.

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Purpose Retinal neovascularization is a severe pathological process leading to irreversible blindness. This study aims to identify the altered metabolites and their related pathways that are involved in retinal neovascularization. Methods To reveal the global metabolomic profile change in the retinal neovascularization process, an untargeted metabolomics analysis of oxygen-induced retinopathy (OIR) mice retinas was carried out first, followed by the validation of amino acids and their derivatives through a targeted metabolomics analysis. The involved pathways were predicted by bioinformatic analysis. Results By untargeted metabolomics, a total of 58 and 49 metabolites altered significantly in OIR retinas under cationic and anionic modes, respectively. By bioinformatics analysis, “ABC transporters,” “central carbon metabolism in cancer.” and “alanine, aspartate, and glutamate metabolism” were the most enriched Kyoto Encyclopedia of Genes and Genomes (KEGG) pathways associated with the changed metabolites. By targeted metabolomics, no significant change was found in the assessed amino acids and their derivatives at postnatal day (P) 12, whereas significantly altered amino acids and their derivatives were recognized at P13, P17, and P42 in OIR retinas. Conclusions The metabolomic profile was significantly altered in the neovascularized retinas. In particular, numerous amino acids and their derivatives were significantly changed in OIR retinas. These altered metabolites, together with their associated pathways, might be involved in the pathogenesis of retinal neovascular diseases.

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“…CNV rarely develops. [132][133][134] OCTA shows perifoveal microvascular alterations and petaloid non-reflective areas in the deep capillary plexus. 135 Full field ERG shows impaired cone and rod esponses in early stages.…”

Section: Gyrate Atrophymentioning

confidence: 99%

Imaging in inherited retinal disorders

Müftüoğlu¹,

Al-Sheikh

Sushma

et al. 2021

European Journal of Ophthalmology

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Inherited retinal diseases, which results from mutations in over 260 identified genes, affect more than 2 million people globally. The diseases mostly cause severe vision loss in young working population and have severe impact on social economic status of the population. Advances in retinal imaging techniques along with developments in gene identification and cell biology techniques have yielded to a better understanding of the genetic and biochemical mechanisms causing these diseases. Retinal imaging along with through ophthalmological examination is essential to make an accurate diagnosis, to decrease the burden of unneccessary anciliary tests and to select the potential patients that can get benefit from the gene treatment. The purpose of the review is to yield an update on inherited retinal diseases by highlighting microstructural changes in retina and to summarize the retinal changes detected by currently available multimodal imaging techniques.

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Intravitreal Ranibizumab for Choroidal Neovascularization Secondary to Gyrate Atrophy in a Young Patient: A Multimodal Imaging Analysis

Abstract: Intravitreal ranibizumab can offer promising anatomical and functional results, maintaining visual acuity in patients with CNV secondary to GA, especially if used in combination with arginine-restricted diet and vitamin B6 supplementation.

Cited by 11 publications

References 8 publications

Gyrate Atrophy of the Choroid and Retina: A Review

Gyrate Atrophy of the Choroid and Retina: A Review

Metabolomics Analyses of Mouse Retinas in Oxygen-Induced Retinopathy

Imaging in inherited retinal disorders

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