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发育停滞:白化病中黄斑形态的高分辨率成像

Arrested development: high-resolution imaging of foveal morphology in albinism.

作者信息

McAllister John T, Dubis Adam M, Tait Diane M, Ostler Shawn, Rha Jungtae, Stepien Kimberly E, Summers C Gail, Carroll Joseph

机构信息

Department of Ophthalmology, Medical College of Wisconsin, Milwaukee, WI, USA.

出版信息

Vision Res. 2010 Apr 7;50(8):810-7. doi: 10.1016/j.visres.2010.02.003. Epub 2010 Feb 10.

Abstract

Albinism, an inherited disorder of melanin biosynthesis, disrupts normal retinal development, with foveal hypoplasia as one of the more commonly associated ocular phenotypes. However the cellular integrity of the fovea in albinism is not well understood - there likely exist important anatomical differences that underlie phenotypic variability within the disease and that also may affect responsiveness to therapeutic intervention. Here, using spectral-domain optical coherence tomography (SD-OCT) and adaptive optics (AO) retinal imaging, we obtained high-resolution images of the foveal region in six individuals with albinism. We provide a quantitative analysis of cone density and outer segment elongation demonstrating that foveal cone specialization is variable in albinism. In addition, our data reveal a continuum of foveal pit morphology, roughly aligning with schematics of normal foveal development based on post-mortem analyses. Different albinism subtypes, genetic mutations, and constitutional pigment background likely play a role in determining the degree of foveal maturation.

摘要

白化病是一种遗传性黑色素生物合成障碍疾病,会破坏正常的视网膜发育,黄斑发育不全是较为常见的相关眼部表型之一。然而,白化病患者黄斑的细胞完整性尚未得到充分了解——很可能存在重要的解剖学差异,这些差异是该疾病表型变异的基础,也可能影响对治疗干预的反应。在这里,我们使用光谱域光学相干断层扫描(SD-OCT)和自适应光学(AO)视网膜成像技术,获得了6名白化病患者黄斑区域的高分辨率图像。我们对视锥细胞密度和外段伸长进行了定量分析,结果表明白化病患者黄斑区视锥细胞的特化存在差异。此外,我们的数据揭示了黄斑凹陷形态的连续性,大致与基于尸检分析的正常黄斑发育示意图相符。不同的白化病亚型、基因突变和体质色素背景可能在决定黄斑成熟程度方面发挥作用。

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