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原发性开角型青光眼的孟德尔基因。

Mendelian genes in primary open angle glaucoma.

机构信息

Department of Ophthalmology and Visual Sciences, Carver College of Medicine, University of Iowa, Iowa City, IA, USA.

Department of Ophthalmology and Visual Sciences, Carver College of Medicine, University of Iowa, Iowa City, IA, USA.

出版信息

Exp Eye Res. 2019 Sep;186:107702. doi: 10.1016/j.exer.2019.107702. Epub 2019 Jun 22.

Abstract

Mutations in each of three genes, myocilin (MYOC), optineurin (OPTN), and TANK binding kinase 1 (TBK1), may cause primary open-angle glaucoma (POAG) that is inherited as a Mendelian trait. MYOC mutations cause 3-4% of POAG cases with IOP >21 mmHg, while mutations in OPTN, TBK1, and MYOC each cause ∼1% of POAG with IOP ≤21 mmHg, i.e. normal tension glaucoma. Identification of these disease-causing genes has provided insights into glaucoma pathogenesis. Mutations in MYOC cause a cascade of abnormalities in the trabecular meshwork including intracellular retention of MYOC protein, decreased aqueous outflow, higher intraocular pressure, and glaucoma. Investigation of MYOC mutations demonstrated that abnormal retention of intracellular MYOC and stimulation of endoplasmic reticular (ER) stress may be important steps in the development of MYOC-associated glaucoma. Mutations in OPTN and TBK1 cause a dysregulation of autophagy which may directly cause retinal ganglion cell damage and normal tension glaucoma. Discovery of these Mendelian causes of glaucoma has also provided a new set of potential therapeutic targets that may ultimately lead to novel, gene-directed glaucoma treatments.

摘要

在三个基因(myocilin [MYOC]、optineurin [OPTN]和 TANK 结合激酶 1 [TBK1])中的每一个基因发生突变都可能导致作为孟德尔特征遗传的原发性开角型青光眼(POAG)。MYOC 突变导致 3-4%的IOP > 21mmHg 的 POAG 病例,而 OPTN、TBK1 和 MYOC 中的突变分别导致约 1%的IOP ≤ 21mmHg 的 POAG,即正常眼压性青光眼。这些致病基因的鉴定为青光眼发病机制提供了新的见解。MYOC 突变导致小梁网一系列异常,包括 MYOC 蛋白的细胞内滞留、房水流出减少、眼内压升高和青光眼。对 MYOC 突变的研究表明,细胞内 MYOC 的异常滞留和内质网(ER)应激的刺激可能是 MYOC 相关青光眼发展的重要步骤。OPTN 和 TBK1 的突变导致自噬失调,这可能直接导致视网膜神经节细胞损伤和正常眼压性青光眼。这些孟德尔青光眼病因的发现也为一组新的潜在治疗靶点提供了依据,这些靶点最终可能导致新型的、针对基因的青光眼治疗方法。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9fb1/10207284/c11aa7039435/nihms-1893602-f0001.jpg

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Mendelian genes in primary open angle glaucoma.原发性开角型青光眼的孟德尔基因。
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