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NCT Number: NCT05809635

Study of BEST1 Vitelliform Macular Dystrophy

The purpose of this study is to establish the natural history of of participants with BESTROPHIN 1 Vitelliform Macular Dystrophy.

The blinding disorder Best Vitelliform Macular Dystrophy (VMD) is caused by any one of more than 250 different mutations in the BEST1 gene.

As new treatments are developed, a clear understanding of the natural history of disease progression of BEST1 VMD is necessary. The goals of this natural history study are to:

1. Report the natural history of retinal degeneration in participants with a clinical diagnosis of VMD with molecular confirmation of a pathogenic BEST1 mutation(s). 2. Identify sensitive structural and functional outcome measures to use for future multicenter clinical trials for the treatment of BESTROPHIN 1 VMD. 3. Compare progression of the identified structural and functional measures between the two eyes to judge the suitability of the second untreated eye as a control for a future clinical trial involving unilateral treatment 4. Identify well-defined patient populations for future clinical trials of investigative treatments for BEST1 VMD.

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Key information

Sex eligibility

All sexes

Study type

Observational

Primary location

Institut de la Vision/Centre de maladies rares du Centre Hospitalier National Ophtalmologique des Quinze-Vingts, Paris, France

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Who can participate

Healthy volunteers accepted: No

Only the study team can determine whether someone qualifies for participation.

Inclusion criteria

  • Ability to provide informed consent
  • Diagnosis of BEST1-associated VMD by study physician, who are trained retinal specialists in the university clinic Must be able to commit to 4 follow-up study visits (3 years)

Exclusion criteria

  • Systemic condition that prevents the participant from undergoing the exams

Treatment and study plan

Natural History Study

Other

Longitudinal assessment of participants with BEST1 Vitelliform Macular Dystrophy

Primary outcomes

  1. Medmont Dark Adapted Chromatic (DAC) Automated Perimeter

    Time frame: Up to 3 years

  2. Full-field electroretinogram (ERG)

    Time frame: Up to 3 years

    ERG conducted under International Society for Clinical Electrophysiology of Vision (ISCEV) Protocol.

  3. Electroocoulogram (EOG)

    Time frame: Up to 3 years

    EOG conducted under International Society for Clinical Electrophysiology of Vision (ISCEV) Protocol

  4. Optical Coherence Tomography (OCT)

    Time frame: Up to 3 years

  5. Fundus Autofluorescence (FAF)

    Time frame: Up to 3 years

  6. Near-infrared fundus autofluorescence (NIR-AF)

    Time frame: Up to 3 years

  7. Quantitative Fundus Autofluorescence (qAF)

    Time frame: Up to 3 years

Secondary outcomes

  1. Best-corrected Visual Acuity (BCVA)

    Time frame: Up to 3 years

  2. Color Fundus Photos

    Time frame: Up to 3 years

  3. Macular Integrity Assessment (MAIA) Microperimetry

    Time frame: Up to 3 years

  4. Goldman Kinetic Visual Field

    Time frame: Up to 3 years

  5. Light-adapted Static Perimetry

    Time frame: Up to 3 years

  6. Dark-adapted Chromatic Perimetry

    Time frame: Up to 3 years

  7. Full-field Stimulus Testing

    Time frame: Up to 3 years

Study contacts

Contact information is provided by the study sponsor or research team.

Stephen H Tsang, MD, PhD

CONTACT

[email protected]

212-342-1186

Sponsors and collaborators

Lead sponsor

Columbia University

Other

Collaborators

  • Centre Hospitalier National d'Ophtalmologie des Quinze-Vingts
  • National Eye Institute (NEI)
  • Universität Tübingen

Registry information

Official study title

Natural History Study in Retinitis Pigmentosa Caused by Mutations in the BEST1 Gene

Important dates

Study start
2021
Primary completion
2026
Study completion
2026
First posted
Apr 12, 2023
Registry last updated
Jul 30, 2025

OpenTrials presents study information sourced from ClinicalTrials.gov. The official registry record should be consulted for the latest information.

View the official ClinicalTrials.gov record (opens in a new tab)

This listing is for discovery and informational purposes only. It is not medical advice, does not guarantee that a study is recruiting, and does not determine eligibility. Contact the study team and a qualified healthcare professional when considering participation.

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