Improvements in visual acuity and macular morphology following cessation of anti-estrogen drugs in a patient with anti-estrogen maculopathy resembling macular telangiectasia type 2: a pathogenic hypothesis

Abstract Background The relationship between anti-estrogen drugs and macular telangiectasia type 2 (MacTel-2) remains unknown. Here we report a case with anti-estrogen maculopathy resembling MacTel-2 with improved visual function and macular morphology following cessation of anti-estrogen drugs. Cas...

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Main Authors: Akihiro Shinkai, Wataru Saito, Yuki Hashimoto, Susumu Ishida
Format: Article
Language:English
Published: BMC 2019-12-01
Series:BMC Ophthalmology
Subjects:
Online Access:https://doi.org/10.1186/s12886-019-1280-1
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author Akihiro Shinkai
Wataru Saito
Yuki Hashimoto
Susumu Ishida
author_facet Akihiro Shinkai
Wataru Saito
Yuki Hashimoto
Susumu Ishida
author_sort Akihiro Shinkai
collection DOAJ
description Abstract Background The relationship between anti-estrogen drugs and macular telangiectasia type 2 (MacTel-2) remains unknown. Here we report a case with anti-estrogen maculopathy resembling MacTel-2 with improved visual function and macular morphology following cessation of anti-estrogen drugs. Case presentation A 53-year-old woman presented with a 5-month history of central vision loss and anorthopia in both eyes. She had received oral tamoxifen followed by toremifene for 69 months. Funduscopy, fluorescein angiography, and optical coherence tomography (OCT) revealed MacTel-2-like findings OU. Fundus autofluorescence (FAF) showed hyper-autofluorescence at the fovea OU. Visual acuity, macular morphology on OCT, and FAF findings gradually improved after cessation of anti-estrogen drugs. Conclusions In the present case, visual acuity, macular morphology, and impairment of the retinal pigment epithelium (RPE) improved following cessation of anti-estrogen drugs, suggesting the relationship between retinal toxicity of anti-estrogen drugs and the development of MacTel-2-like findings. From these results and the previous observations, toxicity of both photoreceptor and RPE cells caused by anti-estrogen drugs may contribute to the development of anti-estrogen maculopathy similar to MacTel-2.
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spelling doaj.art-f850e8cba5dc4e1d80dd11a1a9cabd092022-12-21T22:56:40ZengBMCBMC Ophthalmology1471-24152019-12-011911410.1186/s12886-019-1280-1Improvements in visual acuity and macular morphology following cessation of anti-estrogen drugs in a patient with anti-estrogen maculopathy resembling macular telangiectasia type 2: a pathogenic hypothesisAkihiro Shinkai0Wataru Saito1Yuki Hashimoto2Susumu Ishida3Department of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, Hokkaido UniversityDepartment of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, Hokkaido UniversityDepartment of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, Hokkaido UniversityDepartment of Ophthalmology, Faculty of Medicine and Graduate School of Medicine, Hokkaido UniversityAbstract Background The relationship between anti-estrogen drugs and macular telangiectasia type 2 (MacTel-2) remains unknown. Here we report a case with anti-estrogen maculopathy resembling MacTel-2 with improved visual function and macular morphology following cessation of anti-estrogen drugs. Case presentation A 53-year-old woman presented with a 5-month history of central vision loss and anorthopia in both eyes. She had received oral tamoxifen followed by toremifene for 69 months. Funduscopy, fluorescein angiography, and optical coherence tomography (OCT) revealed MacTel-2-like findings OU. Fundus autofluorescence (FAF) showed hyper-autofluorescence at the fovea OU. Visual acuity, macular morphology on OCT, and FAF findings gradually improved after cessation of anti-estrogen drugs. Conclusions In the present case, visual acuity, macular morphology, and impairment of the retinal pigment epithelium (RPE) improved following cessation of anti-estrogen drugs, suggesting the relationship between retinal toxicity of anti-estrogen drugs and the development of MacTel-2-like findings. From these results and the previous observations, toxicity of both photoreceptor and RPE cells caused by anti-estrogen drugs may contribute to the development of anti-estrogen maculopathy similar to MacTel-2.https://doi.org/10.1186/s12886-019-1280-1Macular telangiectasia type 2PhotoreceptorTamoxifenRetinal pigment epitheliumToremifeneToxicity
spellingShingle Akihiro Shinkai
Wataru Saito
Yuki Hashimoto
Susumu Ishida
Improvements in visual acuity and macular morphology following cessation of anti-estrogen drugs in a patient with anti-estrogen maculopathy resembling macular telangiectasia type 2: a pathogenic hypothesis
BMC Ophthalmology
Macular telangiectasia type 2
Photoreceptor
Tamoxifen
Retinal pigment epithelium
Toremifene
Toxicity
title Improvements in visual acuity and macular morphology following cessation of anti-estrogen drugs in a patient with anti-estrogen maculopathy resembling macular telangiectasia type 2: a pathogenic hypothesis
title_full Improvements in visual acuity and macular morphology following cessation of anti-estrogen drugs in a patient with anti-estrogen maculopathy resembling macular telangiectasia type 2: a pathogenic hypothesis
title_fullStr Improvements in visual acuity and macular morphology following cessation of anti-estrogen drugs in a patient with anti-estrogen maculopathy resembling macular telangiectasia type 2: a pathogenic hypothesis
title_full_unstemmed Improvements in visual acuity and macular morphology following cessation of anti-estrogen drugs in a patient with anti-estrogen maculopathy resembling macular telangiectasia type 2: a pathogenic hypothesis
title_short Improvements in visual acuity and macular morphology following cessation of anti-estrogen drugs in a patient with anti-estrogen maculopathy resembling macular telangiectasia type 2: a pathogenic hypothesis
title_sort improvements in visual acuity and macular morphology following cessation of anti estrogen drugs in a patient with anti estrogen maculopathy resembling macular telangiectasia type 2 a pathogenic hypothesis
topic Macular telangiectasia type 2
Photoreceptor
Tamoxifen
Retinal pigment epithelium
Toremifene
Toxicity
url https://doi.org/10.1186/s12886-019-1280-1
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