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一例显性遗传性视神经萎缩病例中的新突变?

A novel mutation in a case of dominant optic atrophy?

作者信息

Ramkumar Hema L, Savino Peter J

机构信息

Department of Ophthalmology, Shiley Eye Center, University of California-San Diego, La Jolla, CA, USA.

出版信息

Indian J Ophthalmol. 2014 Oct;62(10):1034-6. doi: 10.4103/0301-4738.146043.

Abstract

A 39-year-old healthy woman presented for decreased vision at distance and near for 4 years. She also noted a decrease in her color vision. Her best-corrected visual acuities were 20/70 in each eye. Her visual fields were abnormal, and she had bilateral sluggish pupils, impaired color vision, and optic disc pallor. The magnetic resonance imaging of the brain, heavy metal screen, autoimmune work-up, B12, B6, folate, erythrocyte sedimentation rate, rapid plasma reagin, and Lyme titer were all normal. Optical coherence tomography of the macula and electroretinogram were normal; the visual evoked potential was unrecordable in both eyes. She denied a family history of similar ocular issues, and genotyping of the OPA1 gene revealed a novel previously unreported mutation at IVS12+10T >C.

摘要

一名39岁健康女性因远近视力下降4年前来就诊。她还注意到自己的色觉减退。她双眼的最佳矫正视力均为20/70。她的视野异常,双侧瞳孔反应迟钝,色觉受损,视盘苍白。脑部磁共振成像、重金属筛查、自身免疫检查、维生素B12、维生素B6、叶酸、红细胞沉降率、快速血浆反应素和莱姆病滴度均正常。黄斑光学相干断层扫描和视网膜电图正常;双眼视觉诱发电位均无法记录。她否认有类似眼部问题的家族史,OPA1基因分型显示在IVS12+10T>C处有一个新的、以前未报告的突变。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5205/4278120/559d509b9fc8/IJO-62-1034-g001.jpg

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本文引用的文献

1
OPA1-associated disorders: phenotypes and pathophysiology.
Int J Biochem Cell Biol. 2009 Oct;41(10):1855-65. doi: 10.1016/j.biocel.2009.04.012. Epub 2009 Apr 21.
2
Molecular genetic basis of primary inherited optic neuropathies.
Eye (Lond). 2004 Nov;18(11):1126-32. doi: 10.1038/sj.eye.6701570.
3
OPA1 (Kjer type) dominant optic atrophy: a novel mitochondrial disease.
Mol Genet Metab. 2002 Feb;75(2):97-107. doi: 10.1006/mgme.2001.3278.
4
Clinical and genetic analysis of a family affected with dominant optic atrophy (OPA1).
Arch Ophthalmol. 1997 Jan;115(1):95-9. doi: 10.1001/archopht.1997.01100150097016.
5
Visual prognosis in autosomal dominant optic atrophy (Kjer type).
Am J Ophthalmol. 1993 Mar 15;115(3):360-7. doi: 10.1016/s0002-9394(14)73589-5.
6
Dominant optic atrophy. The clinical profile.
Arch Ophthalmol. 1979 Sep;97(9):1680-6. doi: 10.1001/archopht.1979.01020020248013.

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