Macular microvascular findings in familial exudative vitreoretinopathy on optical coherence tomography angiography_Chinese Journal of Ocular Fundus Diseases

Authors：

Wang Wenting , Li Shuchan , Jiang Keke , Ran Hongyun , Ai Zhaohui ,  Zhang Jie , Liu Lifeng

Weifang Eye Hospital, Weifang 261000, China;

Corresponding author：

Zhang Jie, Email: zhangjie9jiezuo@126.com

Keywords：

Retinal diseases; Eye diseases, hereditary; Tomography, optical coherence; Regional blood flow

DOI：

10.3760/cma.j.cn511434-20210207-00065

Video：

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Objective To evaluate macular microvessel changes in familial exudative vitreoretinopathy (FEVR) by optical coherence tomography angiography. Methods Cross-sectional clinical case-control study. From November 2019 to November 2020, 21 FEVR patients (41 eyes) from Weifang Eye Hospital were selected; 17 healthy volunteers (28 eyes) with the same age and gender as FEVR group were selected as normal control group. According to the best corrected visual acuity (BCVA) 1.0 and ＜1.0, FEVR group was divided into normal visual acuity group (27 eyes) and visual acuity decreased group (14 eyes). All enrollees received BCVA and OCTA. BCVA was performed with an international standard visual acuity chart, which was converted to logarithm of the minimum angle of resolution (logMAR) vision. The OCTA instrument was used to scan the macular area of all the examined eyes in the range of 3 mm×3 mm, 6 mm×6 mm, and the blood vessel density (VD) and blood perfusion density (PD) within the range of 3 mm×3 mm, 6 mm×6 mm were measured and the area, circumference, and morphological index of the foveal avascular zone (FAZ) within the range of 6 mm×6 mm. Quantitative data were compared between groups by independent sample t test. Statistical data were compared by χ² test. The area under curve (AUC) of each index was determined according to receiver operating characteristic curve (ROC curve), and the predictive value of each index was evaluated. Results In the macular area of 6 mm×6 mm, VD, PD, FAZ area and FAZ perimeter of FEVR group were all lower than those of normal control group, and the differences were statistically significant (t=−3.350, −2.387, −3.519, −3.029; P＜0.05). In macular area of 3 mm×3 mm and 6 mm×6 mm, compared with normal vision group and vision loss group, both VD and PD decreased. The differences were statistically significant (t=2.088, 2.114, 2.160, 2.545; P＜0.05). In the macular area of 6 mm×6 mm , the FAZ morphological index of the two groups was significantly different (t=2.409, P＜0.05). ROC curve analysis showed that all the indicators had low diagnostic value for FEVR (AUC＜0.5). Conclusion There are microvascular abnormalities in macular area in FEVR patients, and the decrease of blood vessels and the change of FAZ shape may be related to the loss of visual acuity.

Citation： Wang Wenting, Li Shuchan, Jiang Keke, Ran Hongyun, Ai Zhaohui, Zhang Jie, Liu Lifeng. Macular microvascular findings in familial exudative vitreoretinopathy on optical coherence tomography angiography. Chinese Journal of Ocular Fundus Diseases, 2021, 37(12): 932-936. doi: 10.3760/cma.j.cn511434-20210207-00065 Copy

1.	Al-Taie R, Simkin SK, Douçet E, et al. Persistent avascular retina in infants with a history of type 2 retinopathy of prematurity: to treat or not to treat?[J]. J Pediatr Ophthalmol Strabismus, 2019, 56(4): 222-228. DOI: 10.3928/01913913-20190501-01.
2.	Wang Z, Liu CH, Huang S, et al. Wnt signaling in vascular eye diseases[J]. Prog Retin Eye Res, 2019, 70: 110-133. DOI: 10.1016/j.preteyeres.2018.11.008.
3.	Yonekawa Y, Thomas BJ, Drenser KA, et al. Familial exudative vitreoretinopathy: spectral-domain optical coherence tomography of the vitreoretinal interface, retina, and choroid[J]. Ophthalmology, 2015, 122(11): 2270-2277. DOI: 10.1016/j.ophtha.2015.07.024.
4.	Chen J, Stahl A, Krah NM, et al. Retinal expression of Wnt-pathway mediated genes in low-density lipoprotein receptor-related protein 5 (Lrp5) knockout mice[J/OL]. PLoS One, 2012, 7(1): e30203[2012-01-17]. https://pubmed.ncbi.nlm.nih.gov/22272305/. DOI: 10.1371/journal.pone.0030203.
5.	Tauqeer Z, Yonekawa Y. Familial exudative vitreoretinopathy: pathophysiology, diagnosis, and management[J]. Asia Pac J Ophthalmol (Phila), 2018, 7(3): 176-182. DOI: 10.22608/APO.201855.
6.	Kashani AH, Brown KT, Chang E, et al. Diversity of retinal vascular anomalies in patients with familial exudative vitreoretinopathy[J]. Ophthalmology, 2014, 121(11): 2220-2207. DOI: 10.1016/j.ophtha.2014.05.029.
7.	Walker SP. The ROC curve redefined-optimizing sensitivity (and specificity) to the lived reality of cancer[J]. N Engl J Med, 2019, 380(17): 1594-1595. DOI: 10.1056/NEJMp1814951.
8.	Kashani AH, Learned D, Nudleman E, et al. High prevalence of peripheral retinal vascular anomalies in family members of patients with familial exudative vitreoretinopathy[J]. Ophthalmology, 2014, 121(1): 262-268. DOI: 10.1016/j.ophtha.2013.08.010.
9.	Sun Z, Yang D, Tang Z, et al. Optical coherence tomography angiography in diabetic retinopathy: an updated review[J]. Eye (Lond), 2021, 35(1): 149-161. DOI: 10.1038/s41433-020-01233-y.
10.	Tripathy S, Le HG, Cicinelli MV, et al. Longitudinal changes on optical coherence tomography angiography in retinal vein occlusion[J/OL]. J Clin Med, 2021, 10(7): 1423[2021-04-01]. https://pubmed.ncbi.nlm.nih.gov/33916005/. DOI: 10.3390/jcm10071423.
11.	Biçer Ö, Demirel S, Yavuz Z, et al. Comparison of morphological features of type 1 CNV in AMD and pachychoroid neovasculopathy: an OCTA study[J]. Ophthalmic Surg Lasers Imaging Retina, 2020, 51(11): 640-647. DOI: 10.3928/23258160-20201104-06.
12.	Yuan MZ, Chen LL, Yang JY, et al. Comparison of OCT and OCTA manifestations among untreated PCV, neovascular AMD, and CSC in Chinese population[J]. Int J Ophthalmol, 2020, 3(1): 93-103. DOI: 10.18240/ijo.2020.01.14.
13.	Hsu ST, Finn AP, Chen X, et al. Macular microvascular findings in familial exudative vitreoretinopathy on optical coherence tomography angiography[J]. Ophthalmic Surg Lasers Imaging Retina, 2019, 50(5): 322-329. DOI: 10.3928/23258160-20190503-11.
14.	Zhang J, Jiang C, Ruan L, et al. Macular capillary dropout in familial exudative vitreoretinopathy and its relationship with visual acuity and disease progression[J]. Retina, 2020, 40(6): 1140-1147. DOI: 10.1097/IAE.0000000000002490.
15.	Chen C, Liu C, Wang Z, et al. Optical coherence tomography angiography in familial exudative vitreoretinopathy: clinical features and phenotype-genotype correlation[J]. Invest Ophthalmol Vis Sci, 2018, 59(15): 5726-5734. DOI: 10.1167/iovs.18-25377.
16.	Koulisis N, Moysidis SN, Yonekawa Y, et al. Correlating changes in the macular microvasculature and capillary network to peripheral vascular pathologic features in familial exudative vitreoretinopathy[J]. Ophthalmology Retina, 2019, 3(7): 597-606. DOI: 10.1016/j.oret.2019.02.013.
17.	曾运考, 杨大卫, 曹丹, 等. 正常人眼黄斑血流密度及结构与年龄的相关性研究[J]. 中华眼底病杂志, 2019, 35(1): 3-7. DOI: 10.3760/cma.j.issn.1005-1015.2019.01.002.Zeng YK, Yang DW, Cao D, et al. Correlation between macular blood flow density, structure and age in normal eyes[J]. Chin J Ocul Fundus Dis, 2019, 35(1): 3-7. DOI: 10.3760/cma.j.issn.1005-1015.2019.01.002.
18.	Lu Y, Simonett JM, Wang J, et al. Evaluation of automatically quantified foveal avascular zone metrics for diagnosis of diabetic retinopathy using optical coherence tomography angiography[J]. Invest Ophthalmol Vis Sci, 2018, 59(6): 2212-2221. DOI: 10.1167/iovs.17-23498.
19.	Casselholmde Salles M, Kvanta A, Amrén U, et al. Optical coherence tomography angiography in central retinal vein occlusion: correlation between the foveal avascular zone and visual acuity[J]. Invest Ophthalmol Vis Sci, 2016, 57(9): OCT242-246. DOI: 10.1167/iovs.15-18819.
20.	Ciloglu E, Dogan NC. Optical coherence tomography angiography findings in patients with branch retinal vein occlusion treated with anti-VEGF[J]. Arq Bras Oftalmol, 2020, 83(2): 120-126. DOI: 10.5935/0004-2749.20200017.
21.	Furino C, Montrone G, Cicinelli MV, et al. Optical coherence tomography angiography in diabetic patients without diabetic retinopathy[J]. Eur J Ophthalmol, 2020, 30(6): 1418-1423. DOI: 10.1177/1120672119895701.

1. Al-Taie R, Simkin SK, Douçet E, et al. Persistent avascular retina in infants with a history of type 2 retinopathy of prematurity: to treat or not to treat?[J]. J Pediatr Ophthalmol Strabismus, 2019, 56(4): 222-228. DOI: 10.3928/01913913-20190501-01.
2. Wang Z, Liu CH, Huang S, et al. Wnt signaling in vascular eye diseases[J]. Prog Retin Eye Res, 2019, 70: 110-133. DOI: 10.1016/j.preteyeres.2018.11.008.
3. Yonekawa Y, Thomas BJ, Drenser KA, et al. Familial exudative vitreoretinopathy: spectral-domain optical coherence tomography of the vitreoretinal interface, retina, and choroid[J]. Ophthalmology, 2015, 122(11): 2270-2277. DOI: 10.1016/j.ophtha.2015.07.024.
4. Chen J, Stahl A, Krah NM, et al. Retinal expression of Wnt-pathway mediated genes in low-density lipoprotein receptor-related protein 5 (Lrp5) knockout mice[J/OL]. PLoS One, 2012, 7(1): e30203[2012-01-17]. https://pubmed.ncbi.nlm.nih.gov/22272305/. DOI: 10.1371/journal.pone.0030203.
5. Tauqeer Z, Yonekawa Y. Familial exudative vitreoretinopathy: pathophysiology, diagnosis, and management[J]. Asia Pac J Ophthalmol (Phila), 2018, 7(3): 176-182. DOI: 10.22608/APO.201855.
6. Kashani AH, Brown KT, Chang E, et al. Diversity of retinal vascular anomalies in patients with familial exudative vitreoretinopathy[J]. Ophthalmology, 2014, 121(11): 2220-2207. DOI: 10.1016/j.ophtha.2014.05.029.
7. Walker SP. The ROC curve redefined-optimizing sensitivity (and specificity) to the lived reality of cancer[J]. N Engl J Med, 2019, 380(17): 1594-1595. DOI: 10.1056/NEJMp1814951.
8. Kashani AH, Learned D, Nudleman E, et al. High prevalence of peripheral retinal vascular anomalies in family members of patients with familial exudative vitreoretinopathy[J]. Ophthalmology, 2014, 121(1): 262-268. DOI: 10.1016/j.ophtha.2013.08.010.
9. Sun Z, Yang D, Tang Z, et al. Optical coherence tomography angiography in diabetic retinopathy: an updated review[J]. Eye (Lond), 2021, 35(1): 149-161. DOI: 10.1038/s41433-020-01233-y.
10. Tripathy S, Le HG, Cicinelli MV, et al. Longitudinal changes on optical coherence tomography angiography in retinal vein occlusion[J/OL]. J Clin Med, 2021, 10(7): 1423[2021-04-01]. https://pubmed.ncbi.nlm.nih.gov/33916005/. DOI: 10.3390/jcm10071423.
11. Biçer Ö, Demirel S, Yavuz Z, et al. Comparison of morphological features of type 1 CNV in AMD and pachychoroid neovasculopathy: an OCTA study[J]. Ophthalmic Surg Lasers Imaging Retina, 2020, 51(11): 640-647. DOI: 10.3928/23258160-20201104-06.
12. Yuan MZ, Chen LL, Yang JY, et al. Comparison of OCT and OCTA manifestations among untreated PCV, neovascular AMD, and CSC in Chinese population[J]. Int J Ophthalmol, 2020, 3(1): 93-103. DOI: 10.18240/ijo.2020.01.14.
13. Hsu ST, Finn AP, Chen X, et al. Macular microvascular findings in familial exudative vitreoretinopathy on optical coherence tomography angiography[J]. Ophthalmic Surg Lasers Imaging Retina, 2019, 50(5): 322-329. DOI: 10.3928/23258160-20190503-11.
14. Zhang J, Jiang C, Ruan L, et al. Macular capillary dropout in familial exudative vitreoretinopathy and its relationship with visual acuity and disease progression[J]. Retina, 2020, 40(6): 1140-1147. DOI: 10.1097/IAE.0000000000002490.
15. Chen C, Liu C, Wang Z, et al. Optical coherence tomography angiography in familial exudative vitreoretinopathy: clinical features and phenotype-genotype correlation[J]. Invest Ophthalmol Vis Sci, 2018, 59(15): 5726-5734. DOI: 10.1167/iovs.18-25377.
16. Koulisis N, Moysidis SN, Yonekawa Y, et al. Correlating changes in the macular microvasculature and capillary network to peripheral vascular pathologic features in familial exudative vitreoretinopathy[J]. Ophthalmology Retina, 2019, 3(7): 597-606. DOI: 10.1016/j.oret.2019.02.013.
17. 曾运考, 杨大卫, 曹丹, 等. 正常人眼黄斑血流密度及结构与年龄的相关性研究[J]. 中华眼底病杂志, 2019, 35(1): 3-7. DOI: 10.3760/cma.j.issn.1005-1015.2019.01.002.Zeng YK, Yang DW, Cao D, et al. Correlation between macular blood flow density, structure and age in normal eyes[J]. Chin J Ocul Fundus Dis, 2019, 35(1): 3-7. DOI: 10.3760/cma.j.issn.1005-1015.2019.01.002.
18. Lu Y, Simonett JM, Wang J, et al. Evaluation of automatically quantified foveal avascular zone metrics for diagnosis of diabetic retinopathy using optical coherence tomography angiography[J]. Invest Ophthalmol Vis Sci, 2018, 59(6): 2212-2221. DOI: 10.1167/iovs.17-23498.
19. Casselholmde Salles M, Kvanta A, Amrén U, et al. Optical coherence tomography angiography in central retinal vein occlusion: correlation between the foveal avascular zone and visual acuity[J]. Invest Ophthalmol Vis Sci, 2016, 57(9): OCT242-246. DOI: 10.1167/iovs.15-18819.
20. Ciloglu E, Dogan NC. Optical coherence tomography angiography findings in patients with branch retinal vein occlusion treated with anti-VEGF[J]. Arq Bras Oftalmol, 2020, 83(2): 120-126. DOI: 10.5935/0004-2749.20200017.
21. Furino C, Montrone G, Cicinelli MV, et al. Optical coherence tomography angiography in diabetic patients without diabetic retinopathy[J]. Eur J Ophthalmol, 2020, 30(6): 1418-1423. DOI: 10.1177/1120672119895701.

Chinese Journal of Ocular Fundus Diseases

Macular microvascular findings in familial exudative vitreoretinopathy on optical coherence tomography angiography

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