Identification of novel cis-mutations in the GJA8 gene in a 3-generation Iranian family with autosomal dominant congenital nuclear cataract.

BACKGROUND: Cataract is mainly due to the presence of high molecular weight protein, which disrupts the normal function of the lens. Pathogenic variants in Gap Junction protein alpha-8 (GJA8) have been associated with autosomal dominant congenital nuclear cataract. In general, mutations in those genes that have important functions in lens development lead to congenital cataract. METHODS: We conducted whole-exome sequencing (WES) in a four-year-old male patient referred to the genetic center for genetic analysis. He had developed cataract at an early age. DNAs were extracted from the blood samples of all family members and subjected to PCR-Sanger sequencing to confirm the WES results. RESULTS: WES analysis on the proband revealed two mutations in the GJA8 gene (c.G12C, c.G58A). His mother, alongside several other members of the third-generation family, had developed cataract. Sanger sequencing of the interested regions showed that these two mutations were co-segregated in all affected members. However, none of the healthy individuals carried these mutations confirming that these two mutations are located in the same allele (complex allele). Bioinformatics analysis of the mutated GJA8 RNA and protein structure confirmed the pathogenicity of the cis-mutations. CONCLUSIONS: Genetic segregation analysis in a three-generation family and also bioinformatics analysis showed that the complex-allele containing c.G12C+c.G58A mutations in the GJA8 gene is a pathogenic variant that causes autosomal-dominant congenital nuclear cataract.

Role of CFH Y402H and ARMS2 A69S polymorphisms in susceptibility to post rhegmatogenous retinal detachment macular complications.

INTRODUCTION: Rhegmatogenous retinal detachment (RRD) is the most common type of retinal detachment. Purpose of this study is to evaluate the possible association of ARMS2 (age-related macular susceptibility 2) A69S and CFH (complement factor H) Y402H polymorphisms with post-surgical macular complications. MATERIALS AND METHODS: One hundred and two RRD patients with macular involvement and proliferative vitreoretinopathy grade A prospectively were enrolled in the study. All patients were genotyped for two polymorphisms of CFH Y402H and ARMS2 A69S by applying Polymerase Chain Reaction (PCR)-Restriction Fragment Length Polymorphism (RFLP). Scleral buckling or deep vitrectomy performed based on surgeon decision. Optical coherence tomography (OCT) for all patients was performed on three, six, and twelve months after operation. RESULTS: The ARMS2 A69S GT genotype showed significant association with postoperative cystoid macular edema (OR = 3.11, P = 0.039). Logistic regression analysis showed that the effect of ARMS2 GT vs GG genotype remained significant on CME after confounding factors correction. (ARMS2 GT vs GG OR = 4.79, p value = 0.035). No association was observed between studied genotypes and postoperative persistent subfoveal fluid, macular atrophy, and macular epiretinal membrane. CONCLUSIONS: The ARMS2 A69S GT genotype was significantly associated with postoperative cystoid macular edema in RRD cases with macular involvement.

Lack of Association between Monocyte Chemoattractant Protein-1 (MCP-1) Gene Promoter Polymorphism and Behcet's Disease with and without Ocular Involvement in Iranian Population: A Case-Control Study.

PURPOSE: This case-control study aimed to evaluate the possible association of MCP-1 - 2518A/G genetic polymorphism with Behcet's disease (BD) in the Iranian patients. MATERIALS AND METHODS: This study was performed in 135 Behcet's patients (51 ocular and 84 non-ocular) and 79 healthy individuals. Peripheral blood samples were genotyped for MCP-1 - 2518A/G using the PCR-RFLP technique. RESULTS: The statistical analysis of MCP-1 - 2518A/G showed no significant differences in genotype/allele frequencies between Behcet's patients and controls. There was no significant association in genotype/allele frequencies between either ocular or non-ocular BD patients and controls. Also, different genotype/allele frequencies between ocular and non-ocular BD were not statistically significant. CONCLUSIONS: In this study, with a threshold P-value of 0.05 and an estimated power of 0.81 to detect a significant association (odds ratio ≥1.2), we did not observe any association of this variant with Behcet's disease.

Association of Saitohin gene rs62063857 polymorphism with dry type age-related macular degeneration.

PURPOSE: Age-related macular degeneration (AMD) as the leading cause of central visual loss in the developed countries has extensive pathologic similarities with Alzheimer's disease (AD). Saitohin rs62063857 Q7 R polymorphism is associated with increased risk of AD though we decided to evaluate the possible association of this polymorphism with advanced AMD. MATERIALS AND METHODS: 152 advanced AMD patients (134 wet AMD and 18 geographic atrophy) and 75 healthy controls included in this study. Cases and controls went through a standard ophthalmologic examination by a retinal specialist. Saitohin gene rs62063857 polymorphism determined by using PCR technique and restriction enzyme HinFI. To evaluate the differences between groups we used t-test, Chi-Squared and one-tailed Fisher exact test. RESULTS: Distribution of genotypes was not significantly different between total AMD or wet AMD patients compared to that of controls (total AMD RR+QR: OR = 1.51, CI = 0.82-2.79, P = .12; wet AMD RR+QR: OR = 1.39, CI = 0.74-2.59, P = .19). The RR+QR genotypes were significantly higher in dry AMD group compared to that of controls (RR+QR: OR = 2.75, CI = 0.96-7.9, P = .05). CONCLUSION: Our results showed that although STH Q7 R polymorphism was not associated with wet AMD susceptibility it was significantly associated with geographic atrophy.

Association of combined complement factor H Y402H and ARMS2/LOC387715 A69S polymorphisms with age-related macular degeneration: an updated meta-analysis.

BACKGROUND: Complement factor H (CFH) Y402 H (rs1061170) and age-related maculopathy susceptibility2 (ARMS2)/LOC387715 A69 S (rs10490924) polymorphisms shown to have significant association with AMD. In this meta-analysis, we updated and pooled the results of available association studies between combined ARMS2/LOC387715A69 S-CFHY402 H genotypes and AMD to estimate the synergistic effects. METHODS: Heterogeneity of studies was evaluated using Cochran Q-test and I-square index. To modify the heterogeneity in the variables we used random effects model. Meta-analysis was performed using STATA. To estimate the additive or supra-additive effects we calculated RERI (relative excess risk due to interaction), AP (attributable proportion due to interaction), S (synergy index) and V (multiplicative index). RESULTS: We included 12 studies with 4668 AMD patients and 4936 control subjects. Considering the GGTT genotypes as reference line, the pooled AMD odds ratios for stratified combined genotypes was 2.13 (95% CI 1.64-2.78) for GGnonTT, 2.17 (95% CI 1.63-2.89) for nonGGTT and 7.23 (95% CI 4.95-10.55) for nonGGnonTT. Pooled synergy analysis revealed RERI = 3.90 (95% CI 0.58-10.03), AP = .53 (95% CI 0.09-0.69), S = 2.57 (95% CI 1.27-5.22) and V = 1.47 (95% CI 1.21-1.80). CONCLUSION: This updated analysis showed a strong synergistic and positive multiplicative effect of these two genes indicating that there is common pathway of ARMS2/LOC387715 A69 S and CFH Y402 H in AMD pathogenesis which may be complement system pathway.

Tumor necrosis factor (TNF)-308, -1031, and angiotensin-converting enzyme (ACE) DD/II polymorphisms' role in Behcet's disease with and without uveitis: a meta-analysis.

PURPOSE: There are conflicting results of studies investigating the association between the tumor necrosis factor (TNF) and angiotensin-converting enzyme (ACE) gene polymorphisms and Behcet's disease (BD). The aim of this meta-analysis is to assess the association between these gene polymorphisms and ocular involvement in BD. METHODS: We identified relevant studies and reviewed the full-text manuscripts of the studies in order to select those for inclusion. Heterogeneity of studies was evaluated using Cochran Q-test and I-square index. To modify the heterogeneity in the variables we used random effects model. Meta-analysis was performed using STATA. RESULTS: We analyzed TNF-1031, -308 and ACE DD/II genotype difference between BD patients with and without uveitis. Among these polymorphic genetic loci TNF-308 AA genotype has a statistically significant protective effect against BD uveitis (OR = 0.45 vs 1.23, p = .017). Such a statistically significant effect was not seen for other studied genotypes. CONCLUSION: This meta-analysis revealed a significant protective effect of TNF-308 AA genotype against ocular involvement in Behcet's disease.

Correlation of Macular Thickness and Visual Acuity in DME Treated by Two Doses of Intravitreal Ziv-Aflibercept Versus Bevacizumab: Analysis of a Randomized, Three-Armed Clinical Trial.

BACKGROUND AND OBJECTIVE: To report the correlation of central macular thickness (CMT) and best-corrected visual acuity (BCVA) after 1-year treatment by two doses (2.5 mg or 1.25 mg) of intravitreal ziv-aflibercept (IVZ) versus bevacizumab (IVB) in eyes with diabetic macular edema (DME). PATIENTS AND METHODS: In this study, the correlation of CMT and BCVA changes of the eyes enrolled in a previous clinical trial of 123 eyes were re-evaluated. The correlation of BCVA and CMT changes at each visit was evaluated in the three study arms individually. Then, the eyes in each of the arms were classified at each follow-up visit into three subgroups based on their CMT changes related to the baseline CMT: CMT decrease of 30% or more of baseline CMT, between 10% to 29% of baseline CMT, and less than 9% of baseline CMT or CMT increase. RESULTS: BCVA and CMT changes were correlated significantly (P < .05) in all and in half of the follow-up visits, respectively, in the eyes treated by IVZ 1.25 mg and IVB (r = 0.554 and r = 0.617 at 1 year, respectively). Nevertheless, such a significant correlation was not detected in the eyes treated by IVZ 2.5 mg in any of the follow-up visits (r = 0.202 at 1 year; P = .259). In the IVZ 2.5 mg group, BCVA improvement was observed in all subgroups with each level of CMT reductions. CONCLUSION: Ziv-aflibercept 2.5 mg might have a beneficial effect on DME beyond thickness reduction. [Ophthalmic Surg Lasers Imaging Retina. 2019;50:684-690.].

Repeated Injection of Methotrexate into Silicone Oil-Filled Eyes for Grade C Proliferative Vitreoretinopathy: A Pilot Study.

PURPOSE: To evaluate the effects of repeated intra-silicone oil (SO) injections of methotrexate (MTX) on the outcomes of surgery for rhegmatogenous retinal detachment (RRD) with grade C proliferative vitreoretinopathy (PVR-C). METHODS: In this prospective pilot case series, eyes with RRD and PVR-C underwent pars plana vitrectomy and intraocular injection of SO. At the conclusion of the procedure, 250 µg of MTX was injected into the SO-filled vitreous cavity. Intra-SO injection was repeated at weeks 3 and 6; the minimum follow-up period was 6 months. The main outcome measure was retinal reattachment rate. RESULTS: Eleven eyes of 11 patients (mean age, 52.73 ± 18.01 years) were included. The mean follow-up period was 9 ± 3 months (range, 6-15 months). Total retinal detachment with anterior and/or posterior PVR-C was present in all eyes before surgery. Mean preoperative best-corrected visual acuity (BCVA) was 2.62 ± 0.04 logMAR. All operated eyes exhibited retinal reattachment posterior to the equator during the follow-up period. Mean postoperative BCVA was significantly improved to 1.02 ± 0.51 logMAR (p = 0.003). No ocular or systemic side effects were observed. CONCLUSION: Repeated intra-SO injection of MTX as an adjunctive therapy for RRD complicated by PVR showed promising results and was not associated with adverse effects. Further studies are needed to confirm its possible beneficial effects on the final anatomic and functional outcomes in these cases.

Association of polymorphisms of complement factor I rs141853578 (G119R) with age-related macular degeneration in Iranian population.

BACKGROUND: Age-related macular degeneration (AMD) is a complex disease, and recent studies have shown role of complement system genes in its development. Complement factor I regulates the complement pathways, and relationship between CFI polymorphisms and AMD is controversial. We evaluated the possible association of complement factor I rs141853578 (G119R) variation with advanced AMD in Iranian patients. MATERIALS AND METHODS: We included 371 case-control samples consisting of 220 advanced AMD patients and 151 genetically unrelated healthy controls. Extracted DNA samples amplified to obtain fragment including the polymorphic complement factor I rs141853578 (G119R) region. RESULTS: The distribution of the genotypes was significantly different in the AMD patients compared to that of controls (p = 0.035). The TT genotype frequencies for CFI were significantly higher in AMD group (7.7 vs. 2%, OR 4.67, CI 1.33-16.45, p = 0.016). This significant difference was maintained after adjustment for the effects of age and gender (OR 5.09, CI 1.42-18.20, p = 0.012). The minor allele frequency (T allele) was also significantly higher in AMD patients compared to that of controls (29.3 vs. 21.5% OR 1.51, CI 1.07-2.13, p = 0.018). CONCLUSION: Current study showed that CFI rs141853578 (G119R) is a risk factor for developing advanced type AMD. This study also suggests that the frequency of G119R polymorphism in our population is not as rare as reported from other populations.

Giraffe or leopard spot chorioretinopathy as an outstanding finding: case report and literature review.

PURPOSE: Presentation of two typical cases with characteristic leopard retinopathy secondary to bilateral diffuse uveal melanocytic proliferation (BDUMP) and idiopathic uveal effusion syndrome (IUES) and brief review of the literature about leopard spot retinopathy. CASE REPORT: A 43-year-old women, who was a known case of ovarian carcinoma, referred with gradual bilateral visual loss. In ophthalmic examination, subretinal fluid, multiple patchy subretinal hyperpigmented lesions and leopard spot chorioretinopathy were evident in her both eyes. Fluorescein angiography showed multiple nummular hyperfluorescent lesions surrounded by zones of hypofluorescence. Spectral domain optical coherence tomography revealed increased retinal thickness, subretinal fluid and RPE irregularities in both eyes. Enhanced depth imaging OCT (EDI-OCT) showed bilateral subfoveal choroidal thickening. During next 2-year follow-up, she underwent cataract surgery and later on developed neovascular glaucoma in her both eyes. The second case was a 45-year-old man who had developed decreased visual acuity in his left eye for 3 years. Anterior segment examination was unremarkable, and both eyes had normal intraocular pressure. No vitreous inflammation was observed. Fundoscopy revealed diffuse exudative retinal detachment in his left eye. Fluorescein angiography showed leopard spot retinopathy of posterior pole, and EDI-OCT disclosed subfoveal choroidal thickening. After exclusion of other causes of exudative retinal detachment and with diagnosis of IUES, he underwent intravitreal triamcinolone injection (2 mg) which improved his final vision to 20/40. CONCLUSION: Leopard spot retinopathy is an uncommon but clinically distinct manifestation of various disorders. BDUMP may present with leopard spot retinopathy, anterior uveal tract involvement and neovascular glaucoma. As EDI-OCT showed involvement and increased thickening of choroid in both cases of BDUMP and IUES, it may be better to consider such cases as leopard chorioretinopathy and categorize these entities as a member of pachychoroid pigment retinopathy disorders.

Safety of 5914 intravitreal ziv-aflibercept injections.

PURPOSE: To analyse the pooled safety data of intravitreal ziv-aflibercept (IVZ) therapy for various retinal conditions. METHODS: This was a retrospective, observational study which included patients from 14 participating centres who received IVZ. The medical records of patients who received IVZ from March 2015 through October 2017 were evaluated. Patient demographics and ocular details were compiled. Ocular and systemic adverse events that occurred within 1 month of IVZ injections were recorded and defined as either procedure-related or drug-related. RESULTS: A total of 1704 eyes of 1562 patients received 5914 IVZ injections (mean±SD: 3.73±3.94) during a period of 2.5 years. The age of patients was 60.6±12.8 years (mean±SD) and included diverse chorioretinal pathologies. Both ocular (one case of endophthalmitis, three cases of intraocular inflammation, and one case each of conjunctival thinning/necrosis and scleral nodule) and systemic adverse events (two cases of myocardial infarction, one case of stroke and two deaths) were infrequent. CONCLUSION: This constitutes the largest pooled safety report on IVZ use and includes patients from 14 centres distributed across the globe. It shows that IVZ has an acceptable ocular and systemic safety profile with incidences of adverse events similar to those of other vascular endothelial growth factor inhibitory drugs. The analysis supports the continued use of IVZ in various retinal disorders.

Comparison of ARMS2/LOC387715 A69S and CFH Y402H risk effect in wet-type age-related macular degeneration: a meta-analysis.

PURPOSE: We designed this meta-analysis to pool studies which have analyzed both CFH (Y402H or I62V) and ARMS2 A69S in the same samples to compare the effect of CFH and ARMS2 in neovascular AMD. METHODS: Relevant studies identified and reviewed separately in order to select those for inclusion. Included studies had genotype data of studied groups for both ARMS2 A69S and CFH. To modify the heterogeneity in the variables, we used random effects model. Meta-analysis was performed using STATA. Funnel plot and Egger's regression test used for evaluation of the possible publication bias. RESULTS: Overall, we included 6676 neovascular AMD cases and 7668 controls. Pooled overall odds ratios (ORs) (95% CI) for neovascular AMD/control were ARMS2 A69S: OR = 2.35 (2.01-2.75) for GT versus GG; OR = 8.57 (6.91-10.64) for TT versus GG; CFH Y402H: OR = 1.94 (1.73-2.18) for CT versus TT; OR = 4.89 (3.96-6.05) for CC versus TT. ARMS2 A69S genotype OR/CFH Y402H genotype OR (homogeneous genotypes): Asia = 2.14, Europe: 1.87, America: 1.82, Middle East: 3.56, pooled: 1.75. ARMS2 A69S genotype OR/CFH Y402H genotype OR (heterogeneous genotypes): Asia = 0.93, Europe: 1.39, America: 2.06, Middle East: 1.20, pooled: 1.21. ARMS2 A69S risk genotypes have stronger predisposing effect on neovascular AMD compared to CFH Y402H risk genotypes. CONCLUSION: Our inclusion criteria to select those studies which have analyzed the effect of these two loci in the same case-control samples showed much stronger effect of ARMS2 A69S in neovascular AMD compared to the CFH Y402H.

Ubiquitin Associated and SH3 Domain Containing B (UBASH3B) Gene Association with Behcet's Disease in Iranian Population.

PURPOSE: To evaluate the possible association of UBASH3B gene rs4936742 (T > C) polymorphism with Behcet's disease (BD) and posterior uveitis in BD. MATERIALS AND METHODS: One hundred and thirty-one patients with BD (51 Behcet's posterior uveitis and 80 non-ocular Behcet's patients) and 61 unrelated age-matched healthy individuals as a control group without any inflammatory disease were selected. All BD cases were under follow-up and treatment in uveitis or rheumatology clinics for at least 5 years. All research subjects, including control individuals, received a comprehensive rheumatologic evaluation. All patients and controls were genotyped for UBASH3B rs4936742 (T > C) polymorphism by PCR-RFLP technique. RESULTS: The observed frequencies of genotypes were significantly different among patients and controls (19.7% versus 30.5% for TT, OR = 2.9, P = 0.011 and 36.1% versus 45.8% for CT, OR = 2.38, P = 0.017). Frequencies of T allele showed significantly higher values in Behcet's patients (OR = 1.9, P = 0.004). Subgroup genotypic and allelic analyses disclosed no significant difference between Behcet's posterior uveitis and control groups, neither between Behcet's posterior uveitis and non-ocular BD groups. However, genotypic and allelic analyses between non-ocular BD and control groups revealed statistically significant difference (36.3% versus 19.7% for TT, OR = 4.08, P = 0.003 and 43.8% versus 36.1% for CT, OR = 2.68, P = 0.018, 58.1% versus 37.7% for T allele, OR = 2.29, P = 0.001). Individuals carrying the TT genotype for UBASH3B were four times more likely to develop non-ocular BD than unaffected, control individuals. CONCLUSION: Our results showed that the UBASH3B gene rs4936742 (T > C) polymorphism is associated with an increased risk of Behcet's disease, especially non-ocular BD, in Iranian population. We could not find any susceptibility role of this genetic locus for posterior uveitis in Behcet's disease.

One-Year Results of a Trial Comparing 2 Doses of Intravitreal Ziv-aflibercept versus Bevacizumab for Treatment of Diabetic Macular Edema.

PURPOSE: To report 1-year findings of a trial comparing 2 doses of intravitreal ziv-aflibercept (IVZ) with intravitreal bevacizumab (IVB) for treatment of center-involving diabetic macular edema (DME). DESIGN: Three-armed double-blind randomized clinical trial. PARTICIPANTS: A total of 123 eyes with center-involving DME. METHODS: In this clinical trial, eyes with DME were randomly assigned to 2.5 mg intravitreal ziv-aflibercept (42 eyes), 1.25 mg intravitreal ziv-aflibercept (42 eyes), and 1.25 mg IVB injections (39 eyes), every 4 weeks for 3 loading injections and then every 4 weeks for IVB and every 8 weeks for ziv-aflibercept injections. The patients were followed up to 1 year with complete ophthalmologic examination and central macular thickness (CMT) measurement by optical coherence tomography. MAIN OUTCOME MEASURES: Change in best-corrected visual acuity (BCVA) at 1 year. RESULTS: Although no significant difference was evident between the 2 ziv-aflibercept groups at 1 year, BCVA change was significantly better in both ziv-aflibercept groups (-0.33±0.26 and -0.38±0.34 logarithm of the minimum angle of resolution (logMAR) equal to 16 and 18 Early Treatment Diabetic Retinopathy Study (ETDRS) letters, for IVZ 2.5 and 1.25 mg, respectively) than in the IVB group (-0.26±0.35 logMAR, equal to 14 ETDRS letters) at final follow-up (P = 0.007 for IVZ 2.5 mg and P = 0.029 for IVZ 1.25 mg). Regarding CMT changes, there was no significant difference between the 2 ziv-aflibercept groups; however, a significantly greater reduction in CMT was observed in the ziv-aflibercept 2.5 mg group in comparison to the IVB group at 1 year (P = 0.029). Subgroup analysis disclosed no difference in BCVA outcomes at 1 year among the eyes with baseline BCVA >20/50. In the eyes with baseline BCVA ≤20/50, however, the improvement was significantly better at 1 year in both ziv-aflibercept groups compared with the IVB group (P = 0.002 for IVZ 2.5 mg and P = 0.001 for IVZ 1.25 mg). CONCLUSIONS: The 1-year results of this trial disclosed more vision improvement with IVZ compared with IVB in the treatment of center-involving DME. This stronger effect of IVZ, however, was detected in the eyes with initial worse level of vision (≤20/50).

Association of risk genotypes of ARMS2/LOC387715 A69S and CFH Y402H with age-related macular degeneration with and without reticular pseudodrusen: a meta-analysis.

To pool the results of published data regarding association of ARMS2/LOC387715 A69S, CFH Y402H and CFH I62V genotypes with age-related macular degeneration (AMD) with and without reticular pseudodrusen (RPD). The results of this pooled data used to estimate the contribution of each of these genes in the pathogenesis of RPD. Heterogeneity of studies was evaluated using Cochran Q-test and I2 index. To modify the heterogeneity in the variables, we used the random effects model. Meta-analysis was performed using STATA. Odds ratio (OR) of genotypes in each study was calculated. Six studies of AMD with RPD and AMD without RPD cases included in this analysis. Analysis of pooled data showed that risk genotypes frequency of ARMS2 A69S was significantly different between AMD with RPD and AMD without RPD [OR = 1.82, 95% confidence interval (CI): 1.26-2.63 for GT versus GG ARMS2 A69S; OR = 2.40, 95% CI: 1.50-3.84 for TT versus GG ARMS2 A69S]. Further analysis also showed that the risk genotype frequency of CFH Y402H was not significantly different between these two groups (OR = 1.02, 95% CI: 0.69-1.50 for CT versus TT CFH Y402H; OR = 1.09, 95% CI: 0.74-1.60 for CC versus TT CFH Y402H). Comparison of above-mentioned ORs revealed statistically higher values for GT and TT genotypes of ARMS2 A69S compared with CFH Y402H genotypes (p = 0.011, p = 0.014, respectively).Our analysis showed stronger contribution of ARMS2 in AMD with RPD group versus AMD without RPD group, in comparison with CFH genotypes.

Recurrent focal choroidal excavation following multiple evanescent white dot syndrome (MEWDS) associated with acute idiopathic blind spot enlargement.

PURPOSE: To present a recurrent case of conforming focal choroidal excavation (FCE) following multiple evanescent white dot syndrome (MEWDS) in a 25-year-old woman. METHODS: Following spontaneous MEWDS sings resolution our patient noted a recurrent decrease in vision. Repeated OCT revealed elevation and mild disruption of RPE layer at fovea without previous angiographic MEWDS signs. At this time, short-term systemic steroid therapy was started and visual acuity became normal. RESULTS: Following quiescence of the new-onset phase, the conforming type of FCE located in inferior macula appeared in OCT. In the following next 2 years recurrence of presumptive focal subfoveal choriocapillaritis occurred for three times presenting with blurred vision. During every acute attack, above-mentioned FCE disappeared and returned back again after resolution of presumptive focal choriocapillaritis. CONCLUSIONS: This is the first and unique case of recurrent type of FCE following MEWDS. It seems to disappear during active phase of presumptive focal choriocapillaritis and then returns after the eye has become quiescent.