Treatment of Retinitis Pigmentosa-Associated Cystoid Macular Oedema Using Intravitreal Aflibercept (Eylea) despite Minimal Response to Ranibizumab (Lucentis): A Case Report

Strong, Stacey A; Gurbaxani, Avinash; Michaelides, Michel

doi:10.1159/000448427

Cited by 9 publications

(10 citation statements)

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“…We have observed similar responses85 and have commenced a prospective study to determine safety and efficacy of intravitreal aflibercept in RP-CMO (ClinicalTrials.gov identifier NCT02661711). Aflibercept may be superior to other anti-VEGF medications due to its intermediate size (115 kDa) and higher binding affinity.…”

Section: Avenues Of Interventionmentioning

confidence: 80%

Retinitis pigmentosa-associated cystoid macular oedema: pathogenesis and avenues of intervention

2016

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Hereditary retinal diseases are now the leading cause of blindness certification in the working age population (age 16–64 years) in England and Wales, of which retinitis pigmentosa (RP) is the most common disorder. RP may be complicated by cystoid macular oedema (CMO), causing a reduction of central vision. The underlying pathogenesis of RP-associated CMO (RP-CMO) remains uncertain, however, several mechanisms have been proposed, including: (1) breakdown of the blood-retinal barrier, (2) failure (or dysfunction) of the pumping mechanism in the retinal pigment epithelial, (3) Müller cell oedema and dysfunction, (4) antiretinal antibodies and (5) vitreous traction. There are limited data on efficacy of treatments for RP-CMO. Treatments attempted to date include oral and topical carbonic anhydrase inhibitors, oral, topical, intravitreal and periocular steroids, topical non-steroidal anti-inflammatory medications, photocoagulation, vitrectomy with internal limiting membrane peel, oral lutein and intravitreal antivascular endothelial growth factor injections. This review summarises the evidence supporting these treatment modalities. Successful management of RP-CMO should aim to improve both quality and quantity of vision in the short term and may also slow central vision loss over time.

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Section: Avenues Of Interventionmentioning

confidence: 80%

Retinitis pigmentosa-associated cystoid macular oedema: pathogenesis and avenues of intervention

2016

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“…Vision loss occurs through progressive loss of photoreceptors and development of complications such as cystoid macular oedema (CME), epiretinal membrane (ERM) and cataract. 2 Treatment of these complications may improve vision [3][4][5] even if the underlying disorder itself continues to progress. The prevalence of complications is important for planning clinical trials, power calculations and service delivery.…”

Section: Introductionmentioning

confidence: 99%

Prevalence of cystoid macular oedema, epiretinal membrane and cataract in retinitis pigmentosa

et al. 2018

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Background/AimsTo report the prevalence of treatable complications (cystoid macular oedema, CME; epiretinal membrane, ERM and cataract) in patients with retinitis pigmentosa (RP).MethodsConsecutive patients with RP attending a tertiary eye clinic in 2012. Spectral domain-optical coherence tomography was used to determine presence of CME and ERM. Clinic records were reviewed to identify cataract and pseudophakia. Multivariable analyses adjusted for age, gender and other confounders.ResultsData are presented for 338 eyes from 169 patients. CME was present in 58.6% of patients and 50.9% of eyes and was bilateral in 73.7%. ERM, cataract and pseudophakia were present in 22.8%, 23.4% and 11.2% eyes, respectively. In multivariable analyses, CME was associated with younger age (OR 0.81, 95% CI 0.67 to 0.98) but not with gender. Patients with ERM and cataract/pseudophakia were less likely to also have CME (OR 0.19, 95% CI 0.09 to 0.40 and OR 0.37, 95% CI 0.16 to 0.84, respectively). CME was most prevalent in patients with autosomal-dominant inheritance (71.4%), followed by autosomal recessive/sporadic inheritance (58.9%) and least likely in persons with X linked inheritance (12.5%, p<0.001).ConclusionsThe prevalence of treatable RP complications is high and suggests it may be clinically beneficial to screen patients with RP to identify those who may benefit from current or future interventions.

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“…They found that aqueous VEGF-A levels were markedly lower in eyes with RP than in control patients. Strong et al 43 hypothesised that a localised VEGF production under pathologic conditions, for example by Muller cells, may contribute to CME formation in RP patients and this fact may also be the reason for the rarity of peripheral neovascularisation in eyes with RP. The previous reports on intravitreal anti-VEGF agent administered in eyes with RP-related CME [43][44][45][46][47][48][49] are summarised in Table 2.…”

Section: Intravitreal Anti-vascular Endothelial Growth Factor Agentsmentioning

confidence: 99%

Intravitreal Pharmacotherapy in the Treatment of Retinitis Pigmentosa-related Cystoid Macular Oedema

Dönmez¹,

Saatçi²

2017

European Ophthalmic Review

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P eripheral visual field constriction and night blindness are the main features of retinitis pigmentosa (RP). However, central vision is often impaired dramatically even in the early stages of the disease when macular complications such as cystoid macular oedema (CME) occur. The pathogenesis of RP-related CME is still not well explained. Therefore, several treatment alternatives such as topical, oral and systemic pharmacotherapy; laser photocoagulation and even vitreoretinal surgery are employed by the clinicians. In this review, we summarise the clinical data on intravitreal pharmacotherapeutic agents and focus mainly on the steroids and anti-vascular endothelial growth factor agents. KeywordsAflibercept, intravitreal injection, macula, macular oedema, Ozurdex ® , ranibizumab, retinitis pigmentosa, steroid, vascular endothelial growth factor Disclosure: Oya Donmez and Ali Osman Saatci have nothing to disclose in relation to this article. No funding was received for the publication of this article. This study involves a review of the literature and did not involve any studies with human or animal subjects performed by any of the authors Authorship: All named authors meet the International Committee of Medical Journal Editors (ICMJE) criteria for authorship for this manuscript, take responsibility for the integrity of the work as a whole and have given final approval for the version to be published.Open Access: This article is published under the Creative Commons Attribution Noncommercial License, which permits any non-commercial use, distribution, adaptation, and reproduction provided the original author(s) and source are given appropriate credit. 1,2 Night blindness together with the peripheral visual field loss is the most prominent clinical feature.1,2 Central vision is relatively spared up to the later stages of the disease process.3 However, when macular complications such as macular oedema arise, central vision is often dramatically impaired even in the early stages of the disease. The prevalence of cystoid macular oedema (CME) is reported to be 10-20% in the eyes of patients with RP. 4,5The pathogenesis of RP-related CME is still unclear. Blood-retinal barrier (BRB) impairment, retinal pigment epithelium (RPE) pump disturbance, inflammation, autoimmunity and vitreoretinal interface changes may be among the potential causes of CME. [6][7][8][9][10][11][12][13][14][15][16][17][18][19] Fishman et al. 6 examined 15 RP patients with vitreous fluorophotometry. All patients showed abnormally high concentrations of fluorescein within the vitreous which was a sign of BRB abnormality. Larsen et al. 7 demonstrated that BRB leakage was markedly increased in six RP patients with the help of ocular spectrofluorophotometry. Vinores et al. 9 investigated the immunohistochemical staining for albumin on paraffin sections of 22 normal and 29 RP eyes.Electron microscopic immunocytochemical staining for albumin was performed on additional six normal and nine RP-affected eyes. Two-thirds of the eyes with RP demonstrated extr...

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Treatment of Retinitis Pigmentosa-Associated Cystoid Macular Oedema Using Intravitreal Aflibercept (Eylea) despite Minimal Response to Ranibizumab (Lucentis): A Case Report

Cited by 9 publications

References 15 publications

Retinitis pigmentosa-associated cystoid macular oedema: pathogenesis and avenues of intervention

Retinitis pigmentosa-associated cystoid macular oedema: pathogenesis and avenues of intervention

Prevalence of cystoid macular oedema, epiretinal membrane and cataract in retinitis pigmentosa

Intravitreal Pharmacotherapy in the Treatment of Retinitis Pigmentosa-related Cystoid Macular Oedema

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