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转录因子 AP-2β 在发育中的小鼠小梁网区域的缺失导致进行性青光眼改变。

Deletion of transcription factor AP-2β from the developing murine trabecular meshwork region leads to progressive glaucomatous changes.

机构信息

Department of Pathology and Molecular Medicine, McMaster University, Hamilton, ON, Canada.

Department of Chemical Engineering, McMaster University, Hamilton, ON, Canada.

出版信息

J Neurosci Res. 2022 Feb;100(2):638-652. doi: 10.1002/jnr.24982. Epub 2021 Nov 25.

Abstract

Glaucoma is one of the leading causes of irreversible blindness and can result from abnormalities in anterior segment structures required for aqueous humor outflow, including the trabecular meshwork (TM) and Schlemm's canal (SC). Transcription factors such as AP-2β play critical roles in anterior segment development. Here, we show that the Mgp-Cre knock-in (Mgp-Cre.KI) mouse can be used to target the embryonic periocular mesenchyme giving rise to the TM and SC. Fate mapping of male and female mice indicates that AP-2β loss causes a decrease in iridocorneal angle cells derived from Mgp-Cre.KI-expressing populations compared to controls. Moreover, histological analyses revealed peripheral iridocorneal adhesions in AP-2β mutants that were accompanied by a decrease in expression of TM and SC markers, as observed using immunohistochemistry. In addition, rebound tonometry showed significantly higher intraocular pressure (IOP) that was correlated with a progressive significant loss of retinal ganglion cells, reduced retinal thickness, and reduced retinal function, as measured using an electroretinogram, in AP-2β mutants compared with controls, reflecting pathology described in late-stage glaucoma patients. Importantly, elevated IOP in AP-2β mutants was significantly reduced by treatment with latanoprost, a prostaglandin analog that increases unconventional outflow. These findings demonstrate that AP-2β is critical for TM and SC development, and that these mutant mice can serve as a model for understanding and treating progressive human primary angle-closure glaucoma.

摘要

青光眼是导致不可逆性失明的主要原因之一,可能是由于房水流出所需的前节结构异常引起的,包括小梁网 (TM) 和施莱姆氏管 (SC)。转录因子如 AP-2β 在前段发育中起着关键作用。在这里,我们展示了 Mgp-Cre 敲入 (Mgp-Cre.KI) 小鼠可用于靶向产生 TM 和 SC 的胚胎眶周间充质。雄性和雌性小鼠的命运图谱表明,与对照相比,AP-2β 缺失导致源自 Mgp-Cre.KI 表达群体的虹膜角膜角细胞减少。此外,组织学分析显示,AP-2β 突变体中存在周边虹膜角膜粘连,免疫组织化学显示 TM 和 SC 标志物的表达减少。此外,回弹眼压测量显示,AP-2β 突变体的眼内压明显升高,与视网膜神经节细胞的进行性显著丧失、视网膜厚度减少和视网膜功能降低相关,通过视网膜电图测量,与晚期青光眼患者描述的病理情况相符。重要的是,AP-2β 突变体的高眼压通过前列腺素类似物拉坦前列素的治疗显著降低,拉坦前列素可增加非传统流出。这些发现表明,AP-2β 对 TM 和 SC 的发育至关重要,这些突变体小鼠可作为理解和治疗进行性原发性闭角型青光眼的模型。

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