Published in Pipeline

FDA clears Opus Genetics' IND for BEST1 IRD gene therapy

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5 min read

Opus Genetics has received FDA clearance for its investigational new drug (IND) application of the gene therapy OPGx-BEST1, indicated to treat bestrophine-1 (BEST1)-related inherited retinal disease (IRD).

Let’s start with a look at BEST1.

The BEST1 gene is responsible for providing instruction on making the bestrophin protein, which acts as a channel to manage charged (chlorine ion) atoms’ movement in and out of cells within the retina.

  • Variants (mutations) in the BEST1 gene (as well as the PRHP2 gene) can result in the development of abnormally-shaped channels unable to control chloride flow.
  • Also to keep in mind: BEST1 plays a key role in the retinal pigment epithelium (RPE), which is critical for vision.

As a result of these BEST1 mutations: BEST1-related IRDs (bestrophinopathies) can develop, impacting an estimated 9,000 patients across the United States.

A notable IRD included in this: Vitelliform macular dystrophy (VMD), a rare genetic eye disorder affecting the macula that can lead to worsening vision loss.

  • Breaking this disease down, VMD can manifest into one of two forms: early-onset and adult-onset.
    • The early-onset form is known as Best disease, which typically develops in childhood (though it can also appear later in life).

And how does OPGx-BEST1 target this macular degeneration?

The BEST1 gene therapy delivers a functional copy of the BEST1 gene to RPE cells to create a BEST1 protein and stabilize homeostasis between RPE cells and photoreceptors.

  • How it accomplishes this: By leveraging an adeno-associated virus (AAV) vector via Opus Genetics’ proprietary AAV-based gene therapy platform.

The intent: To return RPE cells to their normal function and enable them to properly support photoreceptors—restoring vision.

Duly noted. Now, what does this IND mean?

A step forward in OPGx-BEST1’s clinical evaluation, to say the least.

In general: An IND is submitted to the FDA by a company or investigator in order to move from the preclinical stage of a proposed investigational drug on to human-based clinical trials.

  • Included in this submission:
    • Pharmacology and toxicology data from preclinical research
    • Proposed clinical trial protocols
    • Manufacturing and quality control info

Notably: The FDA will generally review a submission to ensure the safety and rights of research subjects as well as the quality of the scientific evaluation pertaining to the proposed drug.

And for OPGx-BEST1’s case?

With its IND clearance in place, Opus Genetics intends to initiate a phase 1/2 clinical trial.

Nice! Before we talk more about that, what did its preclinical performance look like?

Promising … and this is based on 2024 research presented at the Association for Research and Vision in Ophthalmology (ARVO) annual meeting.

The IND-enabling study: Evaluated a unilateral subretinal injection of OPGx-BEST1 (in three varying doses) administered (versus vehicle) in a group of canines diagnosed with bestrophinopathy (canine multifocal retinopathy).

The results: OPGx-BEST1 was well tolerated and resulted in no significant ophthalmic and systemic toxicity at two of its doses (low- and mid-doses)—with subtle clinical (funduscopic) signs of potential retinal toxicity noted among patients treated with a high-dose version.

Interesting … so what do we know about this upcoming study?

  • The design: A multicenter, open-label trial
  • The purpose: To evaluate the safety, tolerability, and preliminary efficacy of a single subretinal injection of OPGx-BEST1 in a to-be-determined number of patients
  • The participants: Patients diagnosed with genetically-confirmed BEST1-related IRD
  • The outcome measures: Exploring the biological activity via functional and anatomical endpoints—including changes in visual function and retinal structure

And the timeframe for its initiation?

Per the company: Before the end of 2025 (second half [H2]).

As for the big-picture significance: With no treatment currently approved for BEST1-related IRDs, OPG-BEST1 represents a potential groundbreaking opportunity for this patient base.


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