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
Investigator-initiated study of intravitreal gene therapy with AAV-RC-C08 expressing RS1 for X-linked juvenile retinoschisis patients
Author Affiliations & Notes
  • Junran Sun
    Shanghai General Hospital, Shanghai, Shanghai, China
  • Yang Liu
    Shanghai General Hospital, Shanghai, Shanghai, China
  • Yidong Wu
    Shanghai General Hospital, Shanghai, Shanghai, China
  • Tong Li
    Shanghai General Hospital, Shanghai, Shanghai, China
  • Fenghua Wang
    Shanghai General Hospital, Shanghai, Shanghai, China
  • Xiaodong Sun
    Shanghai General Hospital, Shanghai, Shanghai, China
  • Footnotes
    Commercial Relationships   Junran Sun None; Yang Liu None; Yidong Wu None; Tong Li None; Fenghua Wang None; Xiaodong Sun None
  • Footnotes
    Support  None
Investigative Ophthalmology & Visual Science June 2024, Vol.65, 5332. doi:
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      Junran Sun, Yang Liu, Yidong Wu, Tong Li, Fenghua Wang, Xiaodong Sun; Investigator-initiated study of intravitreal gene therapy with AAV-RC-C08 expressing RS1 for X-linked juvenile retinoschisis patients. Invest. Ophthalmol. Vis. Sci. 2024;65(7):5332.

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

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Abstract

Purpose : To evaluate the efficacy and safety of intravitreal injection of AAV-RC-C08, an adeno-associated viral vector expressing the RS1 gene, as a potential therapeutic intervention for X-linked juvenile retinoschisis.

Methods : A cohort of XLRS patients(n=5) was enrolled in a single-arm, open-label IIT trial. Fundus examinations, visual acuity, visual fields, electroretinography (ERG), and optical coherence tomography (OCT) were employed to monitor both safety and efficacy outcomes.

Results : Following intravitreal injection of AAV-RC-C08, one-month analysis demonstrated a mean increase in ETDRS BCVA of +4.2 letters (p<0.001) and a mean enlargement in dynamic visual field of +22.4% (p<0.001) were observed. Sixty percent of subjects experienced a decrease in CST and structural ameliorations in the macular EZ intensity, with improvement in BCVA and b-wave amplitude. No serious ocular or systemic AEs were encountered.

Conclusions : The IIT study highlights the promising potential of intravitreal AAV-RC-C08 - RS1 as a novel treatment approach for XLRS. Further research and clinical studies are warranted to validate these preliminary results.

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

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