Ten-Year Follow-Up after Bilateral Submacular Neovascular Membrane Removal in a Case of Autosomal Recessive Bestrophinopathy

Herein, we report the case of an 8-year-old girl who presented in December 2000 with a submacular neovascular membrane in the right eye, with a clinical diagnosis of Best disease. At that time, she underwent pars plana vitrectomy (PPV) with removal of the subretinal choroidal neovascularization (CNV...

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Main Authors: Carlos A. Moreira Jr., Carlos A. Moreira-Neto, Mario Junqueira Nobrega, Eduardo Cunha de Souza
Format: Article
Language:English
Published: Karger Publishers 2017-04-01
Series:Case Reports in Ophthalmology
Subjects:
Online Access:http://www.karger.com/Article/FullText/473696
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author Carlos A. Moreira Jr.
Carlos A. Moreira-Neto
Mario Junqueira Nobrega
Eduardo Cunha de Souza
author_facet Carlos A. Moreira Jr.
Carlos A. Moreira-Neto
Mario Junqueira Nobrega
Eduardo Cunha de Souza
author_sort Carlos A. Moreira Jr.
collection DOAJ
description Herein, we report the case of an 8-year-old girl who presented in December 2000 with a submacular neovascular membrane in the right eye, with a clinical diagnosis of Best disease. At that time, she underwent pars plana vitrectomy (PPV) with removal of the subretinal choroidal neovascularization (CNV). Her vision improved from 20/200 to 20/25. Four years later, a new CNV developed in the other eye. Initially, she underwent unsuccessful photodynamic therapy. As her vision worsened, she underwent a second, this time successful, PPV with membrane removal in the left eye, with vision improving to 20/30. Ten years later, she returned complaining of vision loss over the last year. Her vision was 20/200 OU, and optical coherence tomography demonstrated very large intraretinal cystoid spaces resembling bilateral macular schisis. Four ranibizumab injections as well as dorzolamide eye drops were tried, both without success. Finally, she underwent PPV with internal limiting membrane peeling and gas-fluid exchange in the left eye. One month later, the macula appeared flat and vision had improved to 20/60. The same procedure was performed 1 year later for the right eye, with vision improving to 20/80. One year later, mild cystic spaces developed again in both eyes, although much smaller than previously observed. Her vision remained stable.
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spelling doaj.art-80a6f7f3d1ae4560b332ffe70af11ce72022-12-22T00:33:00ZengKarger PublishersCase Reports in Ophthalmology1663-26992017-04-018126527010.1159/000473696473696Ten-Year Follow-Up after Bilateral Submacular Neovascular Membrane Removal in a Case of Autosomal Recessive BestrophinopathyCarlos A. Moreira Jr.Carlos A. Moreira-NetoMario Junqueira NobregaEduardo Cunha de SouzaHerein, we report the case of an 8-year-old girl who presented in December 2000 with a submacular neovascular membrane in the right eye, with a clinical diagnosis of Best disease. At that time, she underwent pars plana vitrectomy (PPV) with removal of the subretinal choroidal neovascularization (CNV). Her vision improved from 20/200 to 20/25. Four years later, a new CNV developed in the other eye. Initially, she underwent unsuccessful photodynamic therapy. As her vision worsened, she underwent a second, this time successful, PPV with membrane removal in the left eye, with vision improving to 20/30. Ten years later, she returned complaining of vision loss over the last year. Her vision was 20/200 OU, and optical coherence tomography demonstrated very large intraretinal cystoid spaces resembling bilateral macular schisis. Four ranibizumab injections as well as dorzolamide eye drops were tried, both without success. Finally, she underwent PPV with internal limiting membrane peeling and gas-fluid exchange in the left eye. One month later, the macula appeared flat and vision had improved to 20/60. The same procedure was performed 1 year later for the right eye, with vision improving to 20/80. One year later, mild cystic spaces developed again in both eyes, although much smaller than previously observed. Her vision remained stable.http://www.karger.com/Article/FullText/473696BestrophinopathyBest dystrophyRetinaVitrectomy
spellingShingle Carlos A. Moreira Jr.
Carlos A. Moreira-Neto
Mario Junqueira Nobrega
Eduardo Cunha de Souza
Ten-Year Follow-Up after Bilateral Submacular Neovascular Membrane Removal in a Case of Autosomal Recessive Bestrophinopathy
Case Reports in Ophthalmology
Bestrophinopathy
Best dystrophy
Retina
Vitrectomy
title Ten-Year Follow-Up after Bilateral Submacular Neovascular Membrane Removal in a Case of Autosomal Recessive Bestrophinopathy
title_full Ten-Year Follow-Up after Bilateral Submacular Neovascular Membrane Removal in a Case of Autosomal Recessive Bestrophinopathy
title_fullStr Ten-Year Follow-Up after Bilateral Submacular Neovascular Membrane Removal in a Case of Autosomal Recessive Bestrophinopathy
title_full_unstemmed Ten-Year Follow-Up after Bilateral Submacular Neovascular Membrane Removal in a Case of Autosomal Recessive Bestrophinopathy
title_short Ten-Year Follow-Up after Bilateral Submacular Neovascular Membrane Removal in a Case of Autosomal Recessive Bestrophinopathy
title_sort ten year follow up after bilateral submacular neovascular membrane removal in a case of autosomal recessive bestrophinopathy
topic Bestrophinopathy
Best dystrophy
Retina
Vitrectomy
url http://www.karger.com/Article/FullText/473696
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