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Doyne honeycomb retinal dystrophy – functional improvement following subthreshold nanopulse laser treatment: a case report

BACKGROUND: Based on phenotypic similarities between age-related macular degeneration and the autosomal disorder Doyne honeycomb retinal dystrophy, we report on a single nanolaser treatment of a patient with genotype Doyne honeycomb retinal dystrophy confirmation and evidence of disease progression...

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Autores principales: Cusumano, Andrea, Falsini, Benedetto, Giardina, Emiliano, Cascella, Raffaella, Sebastiani, Jacopo, Marshall, John
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6327555/
https://www.ncbi.nlm.nih.gov/pubmed/30626431
http://dx.doi.org/10.1186/s13256-018-1935-1
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author Cusumano, Andrea
Falsini, Benedetto
Giardina, Emiliano
Cascella, Raffaella
Sebastiani, Jacopo
Marshall, John
author_facet Cusumano, Andrea
Falsini, Benedetto
Giardina, Emiliano
Cascella, Raffaella
Sebastiani, Jacopo
Marshall, John
author_sort Cusumano, Andrea
collection PubMed
description BACKGROUND: Based on phenotypic similarities between age-related macular degeneration and the autosomal disorder Doyne honeycomb retinal dystrophy, we report on a single nanolaser treatment of a patient with genotype Doyne honeycomb retinal dystrophy confirmation and evidence of disease progression over 12 months. The case study is the first report of short-term results of subthreshold nanolaser treatment in a patient with Doyne honeycomb retinal dystrophy. CASE PRESENTATION: A 43-year-old Caucasian man with moderate loss of visual acuity in his left eye (20/40) and normal visual acuity in his right eye (20/20), with clinical Doyne honeycomb retinal dystrophy diagnosis and genetic confirmation of the common heterozygous mutation (EFEMP1) by genetic testing, underwent nanopulse subthreshold laser treatment in his left eye. A safety examination, carried out 7 days after treatment, and clinical follow-up, conducted 60 days following laser treatment, showed improvement of visual acuity from baseline by two letters and a subjective improvement of blurring. While no apparent morphological changes were found on fundoscopy, increased autofluorescence in the treated eye was observed on imaging. In addition, 2 months after nanopulse subthreshold laser treatment, rod-mediated and cone-mediated full-field electroretinography b-wave amplitudes showed an increase from baseline in both the treated eye (300%) and untreated eye (50%). At 2 months after nanopulse subthreshold laser treatment, multifocal electroretinograms showed improvement. Acuity and full-field electroretinography improvement persisted at 6-month follow-up. CONCLUSIONS: Sustained improvements in retinal function on electroretinography persisted in both eyes 6 months after treatment, suggesting an enhancement of phototransduction and retinoid recycling induced by nanopulse subthreshold laser treatment. The functional improvement observed in the untreated eye is hypothesized to arise from an increased expression and release of metalloproteinases that circulate systemically.
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spelling pubmed-63275552019-01-15 Doyne honeycomb retinal dystrophy – functional improvement following subthreshold nanopulse laser treatment: a case report Cusumano, Andrea Falsini, Benedetto Giardina, Emiliano Cascella, Raffaella Sebastiani, Jacopo Marshall, John J Med Case Rep Case Report BACKGROUND: Based on phenotypic similarities between age-related macular degeneration and the autosomal disorder Doyne honeycomb retinal dystrophy, we report on a single nanolaser treatment of a patient with genotype Doyne honeycomb retinal dystrophy confirmation and evidence of disease progression over 12 months. The case study is the first report of short-term results of subthreshold nanolaser treatment in a patient with Doyne honeycomb retinal dystrophy. CASE PRESENTATION: A 43-year-old Caucasian man with moderate loss of visual acuity in his left eye (20/40) and normal visual acuity in his right eye (20/20), with clinical Doyne honeycomb retinal dystrophy diagnosis and genetic confirmation of the common heterozygous mutation (EFEMP1) by genetic testing, underwent nanopulse subthreshold laser treatment in his left eye. A safety examination, carried out 7 days after treatment, and clinical follow-up, conducted 60 days following laser treatment, showed improvement of visual acuity from baseline by two letters and a subjective improvement of blurring. While no apparent morphological changes were found on fundoscopy, increased autofluorescence in the treated eye was observed on imaging. In addition, 2 months after nanopulse subthreshold laser treatment, rod-mediated and cone-mediated full-field electroretinography b-wave amplitudes showed an increase from baseline in both the treated eye (300%) and untreated eye (50%). At 2 months after nanopulse subthreshold laser treatment, multifocal electroretinograms showed improvement. Acuity and full-field electroretinography improvement persisted at 6-month follow-up. CONCLUSIONS: Sustained improvements in retinal function on electroretinography persisted in both eyes 6 months after treatment, suggesting an enhancement of phototransduction and retinoid recycling induced by nanopulse subthreshold laser treatment. The functional improvement observed in the untreated eye is hypothesized to arise from an increased expression and release of metalloproteinases that circulate systemically. BioMed Central 2019-01-10 /pmc/articles/PMC6327555/ /pubmed/30626431 http://dx.doi.org/10.1186/s13256-018-1935-1 Text en © The Author(s). 2019 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 Case Report
Cusumano, Andrea
Falsini, Benedetto
Giardina, Emiliano
Cascella, Raffaella
Sebastiani, Jacopo
Marshall, John
Doyne honeycomb retinal dystrophy – functional improvement following subthreshold nanopulse laser treatment: a case report
title Doyne honeycomb retinal dystrophy – functional improvement following subthreshold nanopulse laser treatment: a case report
title_full Doyne honeycomb retinal dystrophy – functional improvement following subthreshold nanopulse laser treatment: a case report
title_fullStr Doyne honeycomb retinal dystrophy – functional improvement following subthreshold nanopulse laser treatment: a case report
title_full_unstemmed Doyne honeycomb retinal dystrophy – functional improvement following subthreshold nanopulse laser treatment: a case report
title_short Doyne honeycomb retinal dystrophy – functional improvement following subthreshold nanopulse laser treatment: a case report
title_sort doyne honeycomb retinal dystrophy – functional improvement following subthreshold nanopulse laser treatment: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6327555/
https://www.ncbi.nlm.nih.gov/pubmed/30626431
http://dx.doi.org/10.1186/s13256-018-1935-1
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