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Efficacy of rituximab in non-paraneoplastic autoimmune retinopathy

BACKGROUND: Autoimmune retinopathy (AIR) is a rare but potentially blinding condition that is often underdiagnosed. Common features in AIR presentation include rapidly progressive vision loss with abnormal electrophysiological responses of the retina associated with positive anti-retinal antibodies....

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Autores principales: Boudreault, Katherine, Justus, Sally, Sengillo, Jesse D., Schuerch, Kaspar, Lee, Winston, Cabral, Thiago, Tsang, Stephen H.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5512938/
https://www.ncbi.nlm.nih.gov/pubmed/28709429
http://dx.doi.org/10.1186/s13023-017-0680-7
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author Boudreault, Katherine
Justus, Sally
Sengillo, Jesse D.
Schuerch, Kaspar
Lee, Winston
Cabral, Thiago
Tsang, Stephen H.
author_facet Boudreault, Katherine
Justus, Sally
Sengillo, Jesse D.
Schuerch, Kaspar
Lee, Winston
Cabral, Thiago
Tsang, Stephen H.
author_sort Boudreault, Katherine
collection PubMed
description BACKGROUND: Autoimmune retinopathy (AIR) is a rare but potentially blinding condition that is often underdiagnosed. Common features in AIR presentation include rapidly progressive vision loss with abnormal electrophysiological responses of the retina associated with positive anti-retinal antibodies. AIR is also challenging to treat, and thus, the introduction of new potential therapeutic agents is welcomed. The goal of this communication is to assess the effects of rituximab infusions on electroretinogram (ERG) responses and visual function outcomes in patients with non-paraneoplastic autoimmune retinopathy (npAIR). RESULTS: Following infusion(s), three out of five patients showed no evidence of disease progression or improved, while two patients continued to progress on ERG. One patient demonstrated improvement in visual acuity (2 lines) in both eyes. ERG responses provided objective monitoring of patients’ visual function and response to immunosuppression over time. CONCLUSIONS: These findings suggest that patients with npAIR unresponsive to other immunosuppression therapies may benefit from rituximab infusion, although stabilization rather than improvement was more frequently the outcome in our case series. Furthermore, regularly scheduled ERG follow-up examinations are recommended for monitoring patients’ progression during treatment.
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spelling pubmed-55129382017-07-19 Efficacy of rituximab in non-paraneoplastic autoimmune retinopathy Boudreault, Katherine Justus, Sally Sengillo, Jesse D. Schuerch, Kaspar Lee, Winston Cabral, Thiago Tsang, Stephen H. Orphanet J Rare Dis Research BACKGROUND: Autoimmune retinopathy (AIR) is a rare but potentially blinding condition that is often underdiagnosed. Common features in AIR presentation include rapidly progressive vision loss with abnormal electrophysiological responses of the retina associated with positive anti-retinal antibodies. AIR is also challenging to treat, and thus, the introduction of new potential therapeutic agents is welcomed. The goal of this communication is to assess the effects of rituximab infusions on electroretinogram (ERG) responses and visual function outcomes in patients with non-paraneoplastic autoimmune retinopathy (npAIR). RESULTS: Following infusion(s), three out of five patients showed no evidence of disease progression or improved, while two patients continued to progress on ERG. One patient demonstrated improvement in visual acuity (2 lines) in both eyes. ERG responses provided objective monitoring of patients’ visual function and response to immunosuppression over time. CONCLUSIONS: These findings suggest that patients with npAIR unresponsive to other immunosuppression therapies may benefit from rituximab infusion, although stabilization rather than improvement was more frequently the outcome in our case series. Furthermore, regularly scheduled ERG follow-up examinations are recommended for monitoring patients’ progression during treatment. BioMed Central 2017-07-15 /pmc/articles/PMC5512938/ /pubmed/28709429 http://dx.doi.org/10.1186/s13023-017-0680-7 Text en © The Author(s). 2017 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Research
Boudreault, Katherine
Justus, Sally
Sengillo, Jesse D.
Schuerch, Kaspar
Lee, Winston
Cabral, Thiago
Tsang, Stephen H.
Efficacy of rituximab in non-paraneoplastic autoimmune retinopathy
title Efficacy of rituximab in non-paraneoplastic autoimmune retinopathy
title_full Efficacy of rituximab in non-paraneoplastic autoimmune retinopathy
title_fullStr Efficacy of rituximab in non-paraneoplastic autoimmune retinopathy
title_full_unstemmed Efficacy of rituximab in non-paraneoplastic autoimmune retinopathy
title_short Efficacy of rituximab in non-paraneoplastic autoimmune retinopathy
title_sort efficacy of rituximab in non-paraneoplastic autoimmune retinopathy
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5512938/
https://www.ncbi.nlm.nih.gov/pubmed/28709429
http://dx.doi.org/10.1186/s13023-017-0680-7
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