Cargando…
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...
Autores principales: | , , , , , , , , |
---|---|
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 |
_version_ | 1783527215895412736 |
---|---|
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. |
format | Online Article Text |
id | pubmed-7187718 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2020 |
publisher | John Wiley and Sons Inc. |
record_format | MEDLINE/PubMed |
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 |
work_keys_str_mv | AT romagnolimartina idebenoneincreaseschanceofstabilizationrecoveryofvisualacuityinopa1dominantopticatrophy AT lamorgiachiara idebenoneincreaseschanceofstabilizationrecoveryofvisualacuityinopa1dominantopticatrophy AT carbonellimichele idebenoneincreaseschanceofstabilizationrecoveryofvisualacuityinopa1dominantopticatrophy AT divitolidia idebenoneincreaseschanceofstabilizationrecoveryofvisualacuityinopa1dominantopticatrophy AT amoregiulia idebenoneincreaseschanceofstabilizationrecoveryofvisualacuityinopa1dominantopticatrophy AT zenesinicorrado idebenoneincreaseschanceofstabilizationrecoveryofvisualacuityinopa1dominantopticatrophy AT cascavillamarialucia idebenoneincreaseschanceofstabilizationrecoveryofvisualacuityinopa1dominantopticatrophy AT barbonipiero idebenoneincreaseschanceofstabilizationrecoveryofvisualacuityinopa1dominantopticatrophy AT carellivalerio idebenoneincreaseschanceofstabilizationrecoveryofvisualacuityinopa1dominantopticatrophy |