Investigative Ophthalmology & Visual Science Cover Image for Volume 65, Issue 7
June 2024
Volume 65, Issue 7
Open Access
ARVO Annual Meeting Abstract  |   June 2024
VLDLR deficiency promotes TGF-β signaling-induced RPE energy metabolism disturbance and subretinal fibrosis
Author Affiliations & Notes
  • Yusuke Takahashi
    Biochemistry, Wake Forest University School of Medicine, Winston-Salem, North Carolina, United States
  • Xiang Ma
    Cardiovascular Biology Research Program, Oklahoma Medical Research Foundation, Oklahoma City, Oklahoma, United States
  • Wenjing Lily wu
    Dean A. McGee Eye Institute, The University of Oklahoma Health Sciences Center, Oklahoma City, Oklahoma, United States
  • Miwa Hara
    Biochemistry, Wake Forest University School of Medicine, Winston-Salem, North Carolina, United States
  • Carolina Panzarin
    Biochemistry, Wake Forest University School of Medicine, Winston-Salem, North Carolina, United States
  • Jiyang Cai
    Physiology, The University of Oklahoma Health Sciences Center, Oklahoma City, Oklahoma, United States
  • Jian-Xing (Jay) Ma
    Biochemistry, Wake Forest University School of Medicine, Winston-Salem, North Carolina, United States
  • Footnotes
    Commercial Relationships   Yusuke Takahashi None; Xiang Ma None; Wenjing Lily wu None; Miwa Hara None; Carolina Panzarin None; Jiyang Cai None; Jian-Xing (Jay) Ma None
  • Footnotes
    Support  EY032930; EY033330; EY034510; EY033477; EY028949; EY035519; EY034742; EY032931; EY034461
Investigative Ophthalmology & Visual Science June 2024, Vol.65, 731. doi:
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    • Get Citation

      Yusuke Takahashi, Xiang Ma, Wenjing Lily wu, Miwa Hara, Carolina Panzarin, Jiyang Cai, Jian-Xing (Jay) Ma; VLDLR deficiency promotes TGF-β signaling-induced RPE energy metabolism disturbance and subretinal fibrosis. Invest. Ophthalmol. Vis. Sci. 2024;65(7):731.

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      © ARVO (1962-2015); The Authors (2016-present)

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Abstract

Purpose : Subretinal fibrosis is a prominent pathological feature of neovascular age-related macular degeneration (nAMD), responsible for permanent vision loss, and remains untreatable. Although anti-VEGF therapy effectively inhibits choroidal neovascularization (CNV) in most patients with nAMD, repetitive anti-VEGF treatments may increase the risk of subretinal fibrosis. Accumulating evidence suggests that RPE metabolism disturbance may be responsible, at least in part, for promoting CNV and fibrogenesis. The purpose of this study was to elucidate the underlying mechanism between the dysfunction of RPE metabolism and subretinal fibrosis in the nAMD.

Methods : Very low-density lipoprotein receptor knock-out (Vldlr KO) mice were utilized as the experimental model since Vldlr KO mice manifest multiple nAMD-like phenotypes. A single-cell RNA sequencing (scRNA-seq) was performed to compare the transcriptomic profiles of RPE cells from age-matched wild-type (WT) and Vldlr KO mice. The identified gene changes in the scRNA-seq analysis, levels of fibrosis markers, and other proteins in the associated signaling pathway were measured by Western blot analysis, immunohistochemistry, and qRT-PCR. Vldlr-deficiency-induced metabolic reprogramming was examined using a Seahorse analyzer in primary RPE cells from Vldlr KO, WT control mice, and humans. The adeno-associated virus-mediated intervention was performed to explore the role of carnitine palmitoyltransferase 1A (CPT1A) in subretinal fibrosis in Vldlr KO mice.

Results : scRNA sequencing and qRT-PCR analyses revealed the down-regulation of CPT1A and other essential genes in fatty acid oxidation in RPE cells of Vldlr KO mice compared to age-matched WT controls. Mechanistically, we found that TGFβ2-induced fibrosis was triggered by metabolic reprogramming from mitochondrial metabolism to glycolysis through downregulation of CPT1A in an ERK-dependent manner. In addition, VLDLR selectively interacted with unglycosylated TGFβ receptor II and blocked the formation of the TGFβ receptor I/II complex. We further demonstrated that over-expression of CPT1A suppressed the expression of fibrosis markers in the RPE of Vldlr KO mice.

Conclusions : This finding identified a new regulation mechanism for RPE metabolism and the role of VLDLR in suppressing fibrosis. CPT1A may be a new therapeutic target for treating subretinal fibrosis.

This abstract was presented at the 2024 ARVO Annual Meeting, held in Seattle, WA, May 5-9, 2024.

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