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Idebenone increases chance of stabilization/recovery of visual acuity in OPA1‐dominant optic atrophy

We previously documented that idebenone treatment in OPA1‐Dominant Optic Atrophy (OPA1‐DOA) led to some degrees of visual improvement in seven patients. We here present the results of a cohort study, which investigated the effect of off‐label idebenone administration in a larger OPA1‐DOA group compa...

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Autores principales: Romagnoli, Martina, La Morgia, Chiara, Carbonelli, Michele, Di Vito, Lidia, Amore, Giulia, Zenesini, Corrado, Cascavilla, Maria Lucia, Barboni, Piero, Carelli, Valerio
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley and Sons Inc. 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7187718/
https://www.ncbi.nlm.nih.gov/pubmed/32243103
http://dx.doi.org/10.1002/acn3.51026
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author Romagnoli, Martina
La Morgia, Chiara
Carbonelli, Michele
Di Vito, Lidia
Amore, Giulia
Zenesini, Corrado
Cascavilla, Maria Lucia
Barboni, Piero
Carelli, Valerio
author_facet Romagnoli, Martina
La Morgia, Chiara
Carbonelli, Michele
Di Vito, Lidia
Amore, Giulia
Zenesini, Corrado
Cascavilla, Maria Lucia
Barboni, Piero
Carelli, Valerio
author_sort Romagnoli, Martina
collection PubMed
description We previously documented that idebenone treatment in OPA1‐Dominant Optic Atrophy (OPA1‐DOA) led to some degrees of visual improvement in seven patients. We here present the results of a cohort study, which investigated the effect of off‐label idebenone administration in a larger OPA1‐DOA group compared with untreated patients. Inclusion criteria were: OPA1‐DOA clinical and molecular diagnosis, baseline visual acuity (VA) greater than/equal to counting fingers and treatment duration greater than 7 months. We found a significant difference between the last visit and baseline VA in favor of stabilization/recovery in idebenone‐treated as compared to untreated patients. This effect was retained after controlling for confounders.
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spelling pubmed-71877182020-04-29 Idebenone increases chance of stabilization/recovery of visual acuity in OPA1‐dominant optic atrophy Romagnoli, Martina La Morgia, Chiara Carbonelli, Michele Di Vito, Lidia Amore, Giulia Zenesini, Corrado Cascavilla, Maria Lucia Barboni, Piero Carelli, Valerio Ann Clin Transl Neurol Brief Communications We previously documented that idebenone treatment in OPA1‐Dominant Optic Atrophy (OPA1‐DOA) led to some degrees of visual improvement in seven patients. We here present the results of a cohort study, which investigated the effect of off‐label idebenone administration in a larger OPA1‐DOA group compared with untreated patients. Inclusion criteria were: OPA1‐DOA clinical and molecular diagnosis, baseline visual acuity (VA) greater than/equal to counting fingers and treatment duration greater than 7 months. We found a significant difference between the last visit and baseline VA in favor of stabilization/recovery in idebenone‐treated as compared to untreated patients. This effect was retained after controlling for confounders. John Wiley and Sons Inc. 2020-04-03 /pmc/articles/PMC7187718/ /pubmed/32243103 http://dx.doi.org/10.1002/acn3.51026 Text en © 2020 The Authors. Annals of Clinical and Translational Neurology published by Wiley Periodicals, Inc on behalf of American Neurological Association. This is an open access article under the terms of the http://creativecommons.org/licenses/by/4.0/ License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.
spellingShingle Brief Communications
Romagnoli, Martina
La Morgia, Chiara
Carbonelli, Michele
Di Vito, Lidia
Amore, Giulia
Zenesini, Corrado
Cascavilla, Maria Lucia
Barboni, Piero
Carelli, Valerio
Idebenone increases chance of stabilization/recovery of visual acuity in OPA1‐dominant optic atrophy
title Idebenone increases chance of stabilization/recovery of visual acuity in OPA1‐dominant optic atrophy
title_full Idebenone increases chance of stabilization/recovery of visual acuity in OPA1‐dominant optic atrophy
title_fullStr Idebenone increases chance of stabilization/recovery of visual acuity in OPA1‐dominant optic atrophy
title_full_unstemmed Idebenone increases chance of stabilization/recovery of visual acuity in OPA1‐dominant optic atrophy
title_short Idebenone increases chance of stabilization/recovery of visual acuity in OPA1‐dominant optic atrophy
title_sort idebenone increases chance of stabilization/recovery of visual acuity in opa1‐dominant optic atrophy
topic Brief Communications
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7187718/
https://www.ncbi.nlm.nih.gov/pubmed/32243103
http://dx.doi.org/10.1002/acn3.51026
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