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Treatment of cystic cavities in X-linked juvenile retinoschisis: The first sequential cross-over treatment regimen with dorzolamide

PURPOSE: To report the first sequential cross-over treatment with the longest ophthalmic follow-up in a case of X-linked juvenile retinoschisis (XLRS) successfully treated with topical dorzolamide. OBSERVATIONS: A healthy 34 year-old man presented with one month history of decreased visual acuity in...

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Autores principales: Coussa, Razek Georges, Kapusta, Michael Alton
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5731553/
https://www.ncbi.nlm.nih.gov/pubmed/29260104
http://dx.doi.org/10.1016/j.ajoc.2017.07.008
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author Coussa, Razek Georges
Kapusta, Michael Alton
author_facet Coussa, Razek Georges
Kapusta, Michael Alton
author_sort Coussa, Razek Georges
collection PubMed
description PURPOSE: To report the first sequential cross-over treatment with the longest ophthalmic follow-up in a case of X-linked juvenile retinoschisis (XLRS) successfully treated with topical dorzolamide. OBSERVATIONS: A healthy 34 year-old man presented with one month history of decreased visual acuity in his left eye. Funduscopy was significant for a blunted and cystoid-like foveal reflex in both eyes. The macular OCT showed cystic foveal changes OU. The patient was diagnosed with XLRS and was observed. On two subsequent follow-ups, a significant decrease in the patient's visual acuity warranted the use of topical dorzolamide for treating the cystic foveal changes, which completely resolved two months post-treatment initiation. CONCLUSION AND IMPORTANCE: Previous reports showed the benefit of dorzolamide in treating foveal cystic cavities in XLRS. To our knowledge, this is the first case of XLRS demonstrating the benefits of topical dorzolamide based on a sequential cross-over treatment regimen. It may also represent a case with the longest ophthalmic follow-up providing, in consequence, long-term understanding of the natural history and complications of this rare disease After ruling out major causes of cystoid macular edema, XLRS patients presenting with worsening of their visual acuities due to larger cystic macular changes may benefit from an alternating ON/OFF regimen of topical dorzolamide, which offers a significant treatment advantage outweighing its well-known side effects. Our study consolidates the importance of “medication vacation” by showing its efficacy in providing anatomical and visual functional improvements in patients with chronic cystic macular changes.
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spelling pubmed-57315532017-12-19 Treatment of cystic cavities in X-linked juvenile retinoschisis: The first sequential cross-over treatment regimen with dorzolamide Coussa, Razek Georges Kapusta, Michael Alton Am J Ophthalmol Case Rep Case report PURPOSE: To report the first sequential cross-over treatment with the longest ophthalmic follow-up in a case of X-linked juvenile retinoschisis (XLRS) successfully treated with topical dorzolamide. OBSERVATIONS: A healthy 34 year-old man presented with one month history of decreased visual acuity in his left eye. Funduscopy was significant for a blunted and cystoid-like foveal reflex in both eyes. The macular OCT showed cystic foveal changes OU. The patient was diagnosed with XLRS and was observed. On two subsequent follow-ups, a significant decrease in the patient's visual acuity warranted the use of topical dorzolamide for treating the cystic foveal changes, which completely resolved two months post-treatment initiation. CONCLUSION AND IMPORTANCE: Previous reports showed the benefit of dorzolamide in treating foveal cystic cavities in XLRS. To our knowledge, this is the first case of XLRS demonstrating the benefits of topical dorzolamide based on a sequential cross-over treatment regimen. It may also represent a case with the longest ophthalmic follow-up providing, in consequence, long-term understanding of the natural history and complications of this rare disease After ruling out major causes of cystoid macular edema, XLRS patients presenting with worsening of their visual acuities due to larger cystic macular changes may benefit from an alternating ON/OFF regimen of topical dorzolamide, which offers a significant treatment advantage outweighing its well-known side effects. Our study consolidates the importance of “medication vacation” by showing its efficacy in providing anatomical and visual functional improvements in patients with chronic cystic macular changes. Elsevier 2017-07-21 /pmc/articles/PMC5731553/ /pubmed/29260104 http://dx.doi.org/10.1016/j.ajoc.2017.07.008 Text en © 2017 The Authors http://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case report
Coussa, Razek Georges
Kapusta, Michael Alton
Treatment of cystic cavities in X-linked juvenile retinoschisis: The first sequential cross-over treatment regimen with dorzolamide
title Treatment of cystic cavities in X-linked juvenile retinoschisis: The first sequential cross-over treatment regimen with dorzolamide
title_full Treatment of cystic cavities in X-linked juvenile retinoschisis: The first sequential cross-over treatment regimen with dorzolamide
title_fullStr Treatment of cystic cavities in X-linked juvenile retinoschisis: The first sequential cross-over treatment regimen with dorzolamide
title_full_unstemmed Treatment of cystic cavities in X-linked juvenile retinoschisis: The first sequential cross-over treatment regimen with dorzolamide
title_short Treatment of cystic cavities in X-linked juvenile retinoschisis: The first sequential cross-over treatment regimen with dorzolamide
title_sort treatment of cystic cavities in x-linked juvenile retinoschisis: the first sequential cross-over treatment regimen with dorzolamide
topic Case report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5731553/
https://www.ncbi.nlm.nih.gov/pubmed/29260104
http://dx.doi.org/10.1016/j.ajoc.2017.07.008
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