Status:

ACTIVE_NOT_RECRUITING

Gyrate Atrophy Ocular and Systemic Study

Lead Sponsor:

Jaeb Center for Health Research

Collaborating Sponsors:

Conquering Gyrate Atrophy Foundation

Foundation Fighting Blindness

Conditions:

Gyrate Atrophy

Gyrata of Choroid and Retina; Atrophy

Eligibility:

All Genders

12+ years

Brief Summary

The Gyrate Atrophy Ocular and Systemic Study characterizes the natural history of ornithine levels and retinal degeneration (RD) associated with disease-causing OAT variants in the presence of standar...

Detailed Description

Gyrate atrophy is a rare inherited chorioretinal degeneration that is associated with hyperornithinemia, an inborn error of metabolism caused by autosomal recessive mutations in the ornithine aminotra...

Eligibility Criteria

Inclusion

  • Participants must meet all the following inclusion criteria at the Screening Visit in order to be eligible to enroll into the genetic screening phase.
  • Willing to participate in the study and able to communicate consent during the consent process.
  • Willing and able to complete all study visit assessments at each visit over the forty-eight (48) month study period. Age ≥ 12 years.
  • Must meet one (1) of the Genetic Screening Criteria below:
  • At least 2 disease-causing variants in the OAT gene which are homozygous or heterozygous in trans, based on a report from a clinically certified lab, or a report from a research lab that has been pre-approved by the study Genetics Committee.
  • At least 2 disease-causing variants in the OAT gene with unknown phase, based on a report from a clinically certified lab, or a report from a research lab that has been pre-approved by the study Genetics Committee, AND must meet both of the following phenotype criteria: .Classic fundus appearance of gyrate atrophy (based on investigator discretion) AND Elevated ornithine levels \>300 μmol/L (documented on any prior lab report).
  • Note: if a participant has a variant(s) of unknown significance, they will still qualify if they meet the Genetic Screening Criteria above. Ocular Inclusion Criteria Participant must meet the following criteria at the Screening Visit to enroll into the genetic screening phase.
  • Both eyes must have a clinical diagnosis of retinal dystrophy. Both eyes must permit good quality photographic imaging (e.g., but not limited to, clear ocular media, adequate pupil dilation, stable fixation).

Exclusion

  • Participants must not meet any of the following exclusion criteria at the Screening Visit in order to be eligible to enroll into the genetic screening phase.
  • Single pathogenic or likely pathogenic genetic variants known to be associated with autosomal dominant retinitis pigmentosa/retinal dystrophy (AD, heterozygous), X-374 linked retinitis pigmentosa/retinal dystrophy (XL, hemizygous), or mitochondrial inheritance.
  • Expected to enter experimental treatment trial at any time during this study. History of more than 1 year of cumulative treatment, at any time, with an agent associated with pigmentary retinopathy, including hydroxychloroquine, chloroquine, thioridazine, and deferoxamine. Note: Since this is a Natural History Study collecting data on the progression of Gyrate Atrophy, pregnant women will not be specifically excluded from participation.
  • Ocular

Key Trial Info

Start Date :

November 21 2023

Trial Type :

OBSERVATIONAL

Allocation :

ACTUAL

End Date :

December 31 2028

Estimated Enrollment :

46 Patients enrolled

Trial Details

Trial ID

NCT05312736

Start Date

November 21 2023

End Date

December 31 2028

Last Update

September 9 2025

Active Locations (11)

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Page 1 of 3 (11 locations)

1

University of California San Francisco

San Francisco, California, United States, 94158

2

Johns Hopkins University, Wilmer Eye Institute

Baltimore, Maryland, United States, 21287

3

Harvard Univ., Massachusetts Eye and Ear Infirmary

Boston, Massachusetts, United States, 02114

4

University of Pennsylvania

Philadelphia, Pennsylvania, United States, 19104