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Long-term resolution of chronic macular edema after a single dose of intravitreal dexamethasone in familial retinal arterial macroaneurysm

AlZaid, Abdulrahman ; Magliyah, Moustafa ; Schatz, Patrik LU orcid and Al-Dhibi, Hassan (2020) In Ophthalmic Genetics 41(4). p.394-396
Abstract

Purpose: To report a favorable effect of intravitreal dexamethasone implantation in Familial Retinal Arterial Macroaneurysms (FRAM). Methods: Retrospective Case Report. Results: A 32-year-old male who presented with bilateral retinal macroaneurysms. Whole Exome Sequencing (WES) revealed a homozygous c.830–1 G > A mutation in Insulin Growth Factor Binding Protein 7 (IGFBP7) gene, confirming the diagnosis FRAM. The left eye was lost in the course of the disease, whereas the right eye developed a persistent macular edema due to multiple leaking retinal arterial macroaneurysms and responded poorly to intravitreal ranibizumab and only partially to intravitreal aflibercept. Intravitreal dexamethasone implantation in the right eye, on the... (More)

Purpose: To report a favorable effect of intravitreal dexamethasone implantation in Familial Retinal Arterial Macroaneurysms (FRAM). Methods: Retrospective Case Report. Results: A 32-year-old male who presented with bilateral retinal macroaneurysms. Whole Exome Sequencing (WES) revealed a homozygous c.830–1 G > A mutation in Insulin Growth Factor Binding Protein 7 (IGFBP7) gene, confirming the diagnosis FRAM. The left eye was lost in the course of the disease, whereas the right eye developed a persistent macular edema due to multiple leaking retinal arterial macroaneurysms and responded poorly to intravitreal ranibizumab and only partially to intravitreal aflibercept. Intravitreal dexamethasone implantation in the right eye, on the other hand, resulted in marked visual and structural improvement. Conclusion: Intravitreal dexamethasone injections have beneficial anatomical and visual outcomes in FRAM patients with persistent macular edema poorly responsive to intravitreal injections.

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author
; ; and
organization
publishing date
type
Contribution to journal
publication status
published
subject
keywords
CME, FRAM, IGFBP7, persistent macular edema, RAM
in
Ophthalmic Genetics
volume
41
issue
4
pages
3 pages
publisher
Taylor & Francis
external identifiers
  • scopus:85085359570
  • pmid:32429784
ISSN
1381-6810
DOI
10.1080/13816810.2020.1766088
language
English
LU publication?
yes
id
a477e0fe-5666-4f49-aac7-535f9d3f0c88
date added to LUP
2020-06-26 15:24:56
date last changed
2024-04-03 10:06:28
@article{a477e0fe-5666-4f49-aac7-535f9d3f0c88,
  abstract     = {{<p>Purpose: To report a favorable effect of intravitreal dexamethasone implantation in Familial Retinal Arterial Macroaneurysms (FRAM). Methods: Retrospective Case Report. Results: A 32-year-old male who presented with bilateral retinal macroaneurysms. Whole Exome Sequencing (WES) revealed a homozygous c.830–1 G &gt; A mutation in Insulin Growth Factor Binding Protein 7 (IGFBP7) gene, confirming the diagnosis FRAM. The left eye was lost in the course of the disease, whereas the right eye developed a persistent macular edema due to multiple leaking retinal arterial macroaneurysms and responded poorly to intravitreal ranibizumab and only partially to intravitreal aflibercept. Intravitreal dexamethasone implantation in the right eye, on the other hand, resulted in marked visual and structural improvement. Conclusion: Intravitreal dexamethasone injections have beneficial anatomical and visual outcomes in FRAM patients with persistent macular edema poorly responsive to intravitreal injections.</p>}},
  author       = {{AlZaid, Abdulrahman and Magliyah, Moustafa and Schatz, Patrik and Al-Dhibi, Hassan}},
  issn         = {{1381-6810}},
  keywords     = {{CME; FRAM; IGFBP7; persistent macular edema; RAM}},
  language     = {{eng}},
  month        = {{05}},
  number       = {{4}},
  pages        = {{394--396}},
  publisher    = {{Taylor & Francis}},
  series       = {{Ophthalmic Genetics}},
  title        = {{Long-term resolution of chronic macular edema after a single dose of intravitreal dexamethasone in familial retinal arterial macroaneurysm}},
  url          = {{http://dx.doi.org/10.1080/13816810.2020.1766088}},
  doi          = {{10.1080/13816810.2020.1766088}},
  volume       = {{41}},
  year         = {{2020}},
}