Chaperone-mediated autophagy protects against retinal photoreceptor degeneration by modulating proteostasis of glucose metabolism enzymes

R Raquel Gómez-Sintes (Department of Cellular and Molecular Biology, Centro de Investigaciones Biológicas Margarita Salas, Consejo superior de Investigaciones Científicas (CSIC)) I Inmaculada Tasset (Department of Developmental and Molecular Biology, Albert Einstein College of Medicine) I Ignacio Ramírez-Pardo (Department of Cellular and Molecular Biology, Centro de Investigaciones Biológicas Margarita Salas, Consejo superior de Investigaciones Científicas (CSIC)) A Adrián Martín-Segura (Department of Developmental and Molecular Biology, Albert Einstein College of Medicine) S Sandra Alonso-Gil (Department of Cellular and Molecular Biology, Centro de Investigaciones Biológicas Margarita Salas, Consejo superior de Investigaciones Científicas (CSIC)) A Antonio Díaz (Department of Developmental and Molecular Biology, Albert Einstein College of Medicine) K Kristen Lindenau (Department of Developmental and Molecular Biology, Albert Einstein College of Medicine) C Concepción Lillo (Departamento Biología Celular y Patología, Instituto de Neurociencias de Castilla y León (INCYL), Instituto de Investigación Biomédica de Salamanca (IBSAL), Universidad de Salamanca) P Pedro de la Villa (University of Alcalá de Henares (UAH), Pl. de San Diego, s/n, Alcalá de Henares, Madrid 28801, Spain) S Simone Sidoli E Evripidis Gavathiotis A Ana María Cuervo (Department of Developmental and Molecular Biology, Albert Einstein College of Medicine) P Patricia Boya (Department of Cellular and Molecular Biology, Centro de Investigaciones Biológicas Margarita Salas, Consejo superior de Investigaciones Científicas (CSIC))

Abstract

Defective proteostasis is a hallmark of aging cells and tissues. Among the different components of the proteostasis network, in this study, we focus on a selective form of autophagy known as chaperone-mediated autophagy (CMA), and we set out to understand its physiological role in the retina. Using mice deficient for CMA [knockout for lysosome-associated membrane protein type 2A ( Lamp2A )], we have found that CMA blockade leads to impaired visual function, altered retinal proteostasis, and photoreceptor cell death. Conversely, mice that overexpress human LAMP2A show higher resistance to chemically induced photoreceptor degeneration and slower visual function decline. We found a similar protective effect against retinal degeneration upon pharmacological activation of CMA. To start elucidating the mechanisms behind CMA’s protective role in the retina, we used comparative proteomics and found elevated levels of enzymes related with glucose metabolism in CMA-deficient retinas that phenocopy those observed in old mice retinas. Overall, our results highlight a cytoprotective role for CMA in retina, in part through proteostatic regulation of enzymes involved in glucose metabolism, and support the feasibility of pharmacologically upregulating CMA against retinal degeneration.

Article Details

Volume / Issue Vol. 123, Issue 20
Published May 19, 2026
ISSN 0027-8424
Publisher National Academy of Sciences

Authors (13)

R

Raquel Gómez-Sintes

Department of Cellular and Molecular Biology, Centro de Investigaciones Biológicas Margarita Salas, Consejo superior de Investigaciones Científicas (CSIC)

I

Inmaculada Tasset

Department of Developmental and Molecular Biology, Albert Einstein College of Medicine

I

Ignacio Ramírez-Pardo

Department of Cellular and Molecular Biology, Centro de Investigaciones Biológicas Margarita Salas, Consejo superior de Investigaciones Científicas (CSIC)

A

Adrián Martín-Segura

Department of Developmental and Molecular Biology, Albert Einstein College of Medicine

S

Sandra Alonso-Gil

Department of Cellular and Molecular Biology, Centro de Investigaciones Biológicas Margarita Salas, Consejo superior de Investigaciones Científicas (CSIC)

A

Antonio Díaz

Department of Developmental and Molecular Biology, Albert Einstein College of Medicine

K

Kristen Lindenau

Department of Developmental and Molecular Biology, Albert Einstein College of Medicine

C

Concepción Lillo

Departamento Biología Celular y Patología, Instituto de Neurociencias de Castilla y León (INCYL), Instituto de Investigación Biomédica de Salamanca (IBSAL), Universidad de Salamanca

P

Pedro de la Villa

University of Alcalá de Henares (UAH), Pl. de San Diego, s/n, Alcalá de Henares, Madrid 28801, Spain

S

Simone Sidoli

E

Evripidis Gavathiotis

A

Ana María Cuervo

Department of Developmental and Molecular Biology, Albert Einstein College of Medicine

P

Patricia Boya

Department of Cellular and Molecular Biology, Centro de Investigaciones Biológicas Margarita Salas, Consejo superior de Investigaciones Científicas (CSIC)