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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....
Autores principales: | , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2017
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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. |
format | Online Article Text |
id | pubmed-5512938 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2017 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
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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