Clinical and genetic investigation of amantadine-associated corneal edema

Michelle M Hessen, Sina Vahedi, Chloe T Khoo, Gelareh Vakili, Allen O Eghrari Division of Cornea, Cataract, & External Diseases, The Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA Purpose: Amantadine use has been temporally associated with bilat...

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Autores principales:	Hessen MM, Vahedi S, Khoo CT, Vakili G, Eghrari AO
Formato:	article
Lenguaje:	EN
Publicado:	Dove Medical Press 2018
Materias:	Amantadine cornea corneal edema corneal endothelium Fuchs dystrophy Scheimpflug imaging Ophthalmology RE1-994
Acceso en línea:	https://doaj.org/article/69495b443fe54b14af5b23579e0d5467
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spelling	oai:doaj.org-article:69495b443fe54b14af5b23579e0d54672021-12-02T01:38:22ZClinical and genetic investigation of amantadine-associated corneal edema1177-5483https://doaj.org/article/69495b443fe54b14af5b23579e0d54672018-08-01T00:00:00Zhttps://www.dovepress.com/clinical-and-genetic-investigation-of-amantadine-associated-corneal-ed-peer-reviewed-article-OPTHhttps://doaj.org/toc/1177-5483Michelle M Hessen, Sina Vahedi, Chloe T Khoo, Gelareh Vakili, Allen O Eghrari Division of Cornea, Cataract, & External Diseases, The Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA Purpose: Amantadine use has been temporally associated with bilateral corneal edema in a series of cases; however, its pathophysiological mechanisms have yet to be elucidated. We sought to rule out subclinical Fuchs dystrophy as a contributor, characterize its pattern of corneal edema, and describe the long-term outcome of concurrent topical steroids while resuming amantadine. Patient and methods: After a 44-year-old woman presented with new acute onset bilateral corneal edema, amantadine was discontinued, with clinical improvement. However, neurological decompensation required restarting amantadine, which she did concurrently with topical loteprednol. To determine whether subclinical Fuchs dystrophy might be present, triplet-primed polymerase chain reaction was conducted to measure copy number of the CTG18.1 trinucleotide repeat in TCF4. Specular microscopy and Scheimpflug imaging were conducted and followed for 32 months to assess for resolution and stability. Literature review was conducted to assess for consistency of the clinical phenotype. Results: Corneal edema resolved clinically 4 weeks after discontinuation of amantadine. Serial Scheimpflug imaging demonstrated resolution of posterior and central corneal edema and specular microscopy revealed intracellular opacities with loss of endothelial cell density. Despite resuming amantadine, Scheimpflug imaging and specular microscopy measurements remained stable at 32 months. Triplet-primed PCR of CTG18.1 in TCF4 revealed no trinucleotide repeat expansion. Conclusions: Amantadine-associated corneal edema is characteristically posterior and central and appears unlikely to represent early or subclinical decompensation of Fuchs dystrophy. We describe the unique outcome of continued corneal clearance after restarting amantadine concurrently with steroids, a pattern that has persisted over 32 months to date. Keywords: amantadine, cornea, corneal edema, corneal endothelium, Fuchs dystrophy, Scheimpflug imagingHessen MMVahedi SKhoo CTVakili GEghrari AODove Medical PressarticleAmantadinecorneacorneal edemacorneal endotheliumFuchs dystrophyScheimpflug imagingOphthalmologyRE1-994ENClinical Ophthalmology, Vol Volume 12, Pp 1367-1371 (2018)
institution	DOAJ
collection	DOAJ
language	EN
topic	Amantadine cornea corneal edema corneal endothelium Fuchs dystrophy Scheimpflug imaging Ophthalmology RE1-994
spellingShingle	Amantadine cornea corneal edema corneal endothelium Fuchs dystrophy Scheimpflug imaging Ophthalmology RE1-994 Hessen MM Vahedi S Khoo CT Vakili G Eghrari AO Clinical and genetic investigation of amantadine-associated corneal edema
description	Michelle M Hessen, Sina Vahedi, Chloe T Khoo, Gelareh Vakili, Allen O Eghrari Division of Cornea, Cataract, & External Diseases, The Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA Purpose: Amantadine use has been temporally associated with bilateral corneal edema in a series of cases; however, its pathophysiological mechanisms have yet to be elucidated. We sought to rule out subclinical Fuchs dystrophy as a contributor, characterize its pattern of corneal edema, and describe the long-term outcome of concurrent topical steroids while resuming amantadine. Patient and methods: After a 44-year-old woman presented with new acute onset bilateral corneal edema, amantadine was discontinued, with clinical improvement. However, neurological decompensation required restarting amantadine, which she did concurrently with topical loteprednol. To determine whether subclinical Fuchs dystrophy might be present, triplet-primed polymerase chain reaction was conducted to measure copy number of the CTG18.1 trinucleotide repeat in TCF4. Specular microscopy and Scheimpflug imaging were conducted and followed for 32 months to assess for resolution and stability. Literature review was conducted to assess for consistency of the clinical phenotype. Results: Corneal edema resolved clinically 4 weeks after discontinuation of amantadine. Serial Scheimpflug imaging demonstrated resolution of posterior and central corneal edema and specular microscopy revealed intracellular opacities with loss of endothelial cell density. Despite resuming amantadine, Scheimpflug imaging and specular microscopy measurements remained stable at 32 months. Triplet-primed PCR of CTG18.1 in TCF4 revealed no trinucleotide repeat expansion. Conclusions: Amantadine-associated corneal edema is characteristically posterior and central and appears unlikely to represent early or subclinical decompensation of Fuchs dystrophy. We describe the unique outcome of continued corneal clearance after restarting amantadine concurrently with steroids, a pattern that has persisted over 32 months to date. Keywords: amantadine, cornea, corneal edema, corneal endothelium, Fuchs dystrophy, Scheimpflug imaging
format	article
author	Hessen MM Vahedi S Khoo CT Vakili G Eghrari AO
author_facet	Hessen MM Vahedi S Khoo CT Vakili G Eghrari AO
author_sort	Hessen MM
title	Clinical and genetic investigation of amantadine-associated corneal edema
title_short	Clinical and genetic investigation of amantadine-associated corneal edema
title_full	Clinical and genetic investigation of amantadine-associated corneal edema
title_fullStr	Clinical and genetic investigation of amantadine-associated corneal edema
title_full_unstemmed	Clinical and genetic investigation of amantadine-associated corneal edema
title_sort	clinical and genetic investigation of amantadine-associated corneal edema
publisher	Dove Medical Press
publishDate	2018
url	https://doaj.org/article/69495b443fe54b14af5b23579e0d5467
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Clinical and genetic investigation of amantadine-associated corneal edema

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