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Int J Mol Sci ; 24(4)2023 Feb 08.
Artículo en Inglés | MEDLINE | ID: mdl-36834828

RESUMEN

Age-related macular degeneration (AMD) is a blinding disease characterised by dysfunction of the retinal pigmented epithelium (RPE) which culminates in disruption or loss of the neurosensory retina. Genome-wide association studies have identified >60 genetic risk factors for AMD; however, the expression profile and functional role of many of these genes remain elusive in human RPE. To facilitate functional studies of AMD-associated genes, we developed a human RPE model with integrated CRISPR interference (CRISPRi) for gene repression by generating a stable ARPE19 cell line expressing dCas9-KRAB. We performed transcriptomic analysis of the human retina to prioritise AMD-associated genes and selected TMEM97 as a candidate gene for knockdown study. Using specific sgRNAs, we showed that knockdown of TMEM97 in ARPE19 reduced reactive oxygen species (ROS) levels and exerted a protective effect against oxidative stress-induced cell death. This work provides the first functional study of TMEM97 in RPE and supports a potential role of TMEM97 in AMD pathobiology. Our study highlights the potential for using CRISPRi to study AMD genetics, and the CRISPRi RPE platform generated here provided a useful in vitro tool for functional studies of AMD-associated genes.


Asunto(s)
Estudio de Asociación del Genoma Completo , Degeneración Macular , Humanos , Repeticiones Palindrómicas Cortas Agrupadas y Regularmente Espaciadas , Epitelio Pigmentado de la Retina/metabolismo , Degeneración Macular/metabolismo , Estrés Oxidativo , Epitelio/metabolismo
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