bioRxivCell BiologyPreprint
Ambra1 haploinsufficiency results in metabolic alterations and exacerbates age-associated retinal degeneration
I. Ramirez-Pardo, B. Villarejo-Zori, J. I. Jimenez-Loygorri, E. Sierra-Filardi, S. Alonso-Gil, G. Marino, P. de la Villa, P. S. Fitze, J. M. Fuentes, R. Garcia-Escudero, R. Gomez-Sintes, P. Boya
Abstract
Autophagy is a key process in the maintenance of cellular homeostasis. The age-dependent decline in retinal autophagy has been associated with photoreceptor degeneration. Retinal dysfunction can also result from damage to the retinal pigment epithelium (RPE), as the RPE-retina constitutes an important metabolic ecosystem that must be finely tuned to preserve visual function. While studies of mice lacking essential autophagy genes have revealed a predisposition to retinal degeneration, the consequences of a moderate reduction in autophagy, similar to that which occurs during physiological aging, remain unclear. Here, we describe a retinal phenotype consistent with accelerated aging in mice carrying a haploinsufficiency for Ambra1, a pro-autophagic gene. These mice show protein aggregation in the retina and RPE, metabolic underperformance, and premature vision loss. Moreover, Ambra1+/gt mice are more prone to retinal degeneration after RPE stress. These findings indicate that autophagy provides crucial support to RPE-retinal metabolism and protects the retina against stress and physiological aging.
- organism: mouse
- assay: proteomics
- research focus: metabolism & bioenergetics
- approach: experimental assay
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