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一名接受曲安奈德球后注射治疗的儿童因回旋状萎缩继发黄斑囊样水肿。

Cystoid macular edema secondary to gyrate atrophy in a child treated with sub-tenon injection of triamcinolone acetonide.

作者信息

Alparslan Şahin, Fatih Mehmet Türkcü, Muhammed Şahin, Adnan Yıldırım

机构信息

Department of Ophthalmology, Batman Private Hospital, Batman, Turkey.

Department of Ophthalmology, Batman Zilan Hospital, Batman, Turkey.

出版信息

Rom J Ophthalmol. 2018 Jul-Sep;62(3):246-249.

PMID:30505995
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6256076/
Abstract

Gyrate atrophy (GA) of the fundus is a rare, progressive metabolic disease secondary to the deficiency of the pyridoxal phosphate-dependent enzyme, ornithine aminotransferase. GA may lead to cystoid macular edema (CME) resulting from chronic inflammation. We aimed to report a child case with CME secondary to gyrate atrophy. Herein we presented a GA case treated with posterior sub-Tenon triamcinolone acetonide injection. Optical coherence tomography examination revealed the disappearance of the macular edema that is a vision-threatening complication in GA. The present case showed that the posterior sub-Tenon injection of long acting steroids might be a promising treatment in CME secondary to GA.

摘要

视网膜回旋状萎缩(GA)是一种罕见的进行性代谢性疾病,继发于磷酸吡哆醛依赖性酶鸟氨酸转氨酶缺乏。GA可能导致慢性炎症引起的黄斑囊样水肿(CME)。我们旨在报告1例继发于视网膜回旋状萎缩的CME患儿病例。在此,我们呈现1例接受后Tenon囊下曲安奈德注射治疗的GA病例。光学相干断层扫描检查显示黄斑水肿消失,黄斑水肿是GA中一种威胁视力的并发症。本病例表明,后Tenon囊下注射长效类固醇可能是继发于GA的CME的一种有前景的治疗方法。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5492/6256076/d029b1cad54d/RomJOphthalmol-62-246-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5492/6256076/085301bf2612/RomJOphthalmol-62-246-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5492/6256076/d895c32f7d65/RomJOphthalmol-62-246-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5492/6256076/d029b1cad54d/RomJOphthalmol-62-246-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5492/6256076/085301bf2612/RomJOphthalmol-62-246-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5492/6256076/d895c32f7d65/RomJOphthalmol-62-246-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5492/6256076/d029b1cad54d/RomJOphthalmol-62-246-g003.jpg

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Rom J Ophthalmol. 2018 Oct-Dec;62(4):317-318.

本文引用的文献

1
Reversal of cystoid macular edema in gyrate atrophy patients.回旋状萎缩患者黄斑囊样水肿的逆转
Ophthalmic Genet. 2017 Dec;38(6):549-554. doi: 10.1080/13816810.2017.1301966. Epub 2017 Apr 7.
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Arginine-Restricted Therapy Resistant Bilateral Macular Edema Associated with Gyrate Atrophy.与回旋状萎缩相关的精氨酸限制治疗抵抗性双侧黄斑水肿
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Macular edema associated with gyrate atrophy managed with intravitreal triamcinolone: a case report.玻璃体内注射曲安奈德治疗与视网膜色素变性相关的黄斑水肿:一例报告
Arq Bras Oftalmol. 2007 Sep-Oct;70(5):858-61. doi: 10.1590/s0004-27492007000500024.
5
Cystoid macular edema in gyrate atrophy of the choroid and retina: a fluorescein angiography and optical coherence tomography evaluation.脉络膜视网膜回旋状萎缩中的黄斑囊样水肿:荧光素血管造影和光学相干断层扫描评估
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Experimental model of gyrate atrophy in animals.动物回旋状萎缩的实验模型
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Gyrate atrophy of the choroid and retina associated with hyperornithinaemia.与高鸟氨酸血症相关的脉络膜和视网膜回旋状萎缩。
Br J Ophthalmol. 1974 Jan;58(1):3-23. doi: 10.1136/bjo.58.1.3.
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The ornithine aminotransferase (OAT) locus: analysis of RFLPs in gyrate atrophy.鸟氨酸转氨酶(OAT)基因座:视网膜色素变性患者中限制性片段长度多态性分析
Am J Hum Genet. 1988 Feb;42(2):365-72.