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
The Loss-of-function Mechanisms of OPTN in Optic Neuropathy
Author Affiliations & Notes
  • Feng Tian
    Beth Israel Deaconess Medical Center, Boston, Massachusetts, United States
  • Yiqi Wang
    Beth Israel Deaconess Medical Center, Boston, Massachusetts, United States
  • Qinglong Wang
    Beth Israel Deaconess Medical Center, Boston, Massachusetts, United States
  • Keyuan Ren
    Beth Israel Deaconess Medical Center, Boston, Massachusetts, United States
    Peking University, Beijing, Beijing, China
  • Douglas Jiang
    Beth Israel Deaconess Medical Center, Boston, Massachusetts, United States
    University of California San Diego, La Jolla, California, United States
  • Ryan Donahue
    Boston Children's Hospital, Boston, Massachusetts, United States
  • Isha Nagireddy
    Beth Israel Deaconess Medical Center, Boston, Massachusetts, United States
  • Footnotes
    Commercial Relationships   Feng Tian None; Yiqi Wang None; Qinglong Wang None; Keyuan Ren None; Douglas Jiang None; Ryan Donahue None; Isha Nagireddy None
  • Footnotes
    Support  NIH/NEI K99EY032181
Investigative Ophthalmology & Visual Science June 2024, Vol.65, 3549. doi:
  • Views
  • Share
  • Tools
    • Alerts
      ×
      This feature is available to authenticated users only.
      Sign In or Create an Account ×
    • Get Citation

      Feng Tian, Yiqi Wang, Qinglong Wang, Keyuan Ren, Douglas Jiang, Ryan Donahue, Isha Nagireddy; The Loss-of-function Mechanisms of OPTN in Optic Neuropathy. Invest. Ophthalmol. Vis. Sci. 2024;65(7):3549.

      Download citation file:


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

      ×
  • Supplements
Abstract

Purpose : Optic nerve degeneration and RGC loss are cardinal features of glaucoma, an irreversible ocular disease that accounts for the second leading cause (~10%) of blindness in the United States and world-wide. Our preliminary data has implicated novel functions of OPTN in maintaining retinal homeostasis. In the proposed study, by leveraging novel genetic, computational, and electrophysiological tools, we will explore the effects of loss-of-function mutations of OPTN in physiological and pathological conditions of the retina.

Methods : We hypothesized that OPTN loss-of-function mutations may lead to optic neuropathy. We incorporated the hypothesize by designing two models to either knock out OPTN gene in most of the cells in retina or knocking out OPTN gene in specific cell of retina.
OPTN conventional knockout: Intravitreal injection of AAV2-Cre and AAV2-PLAP vectors
By administering AAV2-Cre to OPTNfl/fl mice, we induced the knockout of OPTN genes. Using intravitreal injections, we can effectively knock out the OPTN in most of the cells in retina. This allows us to examine the retinal changes that occur in the absence of the OPTN genes. The outcomes from experimental group are then compared with those from the control group, which received AAV2-PLAP injections.
OPTN conditional knockout: Breeding pairs to knock out OPTN in specific cells.
As we have the outcomes that the complete knock out of OPTN in the whole retina has pathological changes, then we are curious if the OPTN gene is cell autonomous or non-cell autonomous. In order to find out that question, we comply with a model which only knocks out OPTN in specific cells by breeding pair OPTNfl/fl mice with cell specific Cre mice (Vglut2-Cre)

Results : Astrogliosis was observed post one month after intravitreal injection of AAV2-Cre compared to intravitreal injection of AAV2-PLAP. Also, slightly RGCs loss was observed post one-month AAV2-Cre injection.


GFAP fluorescence
Cre STDEV: 11.420944, SEM: 4.66258088
PLAP STDEV: 2.25240612, SEM: 1.12620306
P<0.001

Conclusions : In general, OPTN knock out through intravitreal injection among most of the cells in the retina showed astrogliosis and RGC loss, which demonstrated that OPTN knock out would have a change to the mouse retina, and OPTN could be potentially used as a therapeutic target in normal tension glaucoma.

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

×
×

This PDF is available to Subscribers Only

Sign in or purchase a subscription to access this content. ×

You must be signed into an individual account to use this feature.

×