Status:

UNKNOWN

Characterizing Rate of Progression in USHer Syndrome (CRUSH) Study

Lead Sponsor:

Radboud University Medical Center

Collaborating Sponsors:

Stichting Ushersyndroom

Conditions:

Usher Syndrome, Type 2A

Retinitis Pigmentosa

Eligibility:

All Genders

16-55 years

Brief Summary

Mutations in USH2A give rise to two phenotypes: Usher syndrome type 2a (USH2A) and nonsyndromic RP (USH2A associated nsRP). Usher syndrome is the most common form of congenital deafblindness. Patients...

Eligibility Criteria

Inclusion

  • Clinically diagnosed with rod-cone degeneration and at least two; pathogenic or likely pathogenic mutations in one of the Usher type 2 genes;
  • Willing and able to complete the informed consent process;
  • Ability to return for all study visits over 48 months;
  • Age ≥ 16 years.
  • Both eyes must meet all of the following:
  • Clinical diagnosis of a rod-cone degeneration;
  • Clear ocular media and adequate pupil dilation to permit good quality photographic imaging;
  • Ability to perform kinetic and static perimetry reliably;
  • Baseline visual acuity ETDRS letter score of 54 or more \[approximate Snellen equivalent 20/80 or better\];
  • Stable fixation;
  • Clinically determined \[on Octopus 900 Pro\] kinetic visual field III4e area 7,5°, or more in the study eye.

Exclusion

  • Mutations in genes that cause autosomal dominant RP, X-linked RP, or presence of biallelic mutations in autosomal recessive RP/retinal dystrophy genes other than Usher genes;
  • 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).
  • If either eye has any of the following, the patient is not eligible:
  • Current vitreous hemorrhage;
  • Current or any history of rhegmatogenous retinal detachment;
  • Current or any history of (e.g., prior to cataract or refractive surgery) spherical equivalent of the refractive error worse than -8 Diopters of myopia;
  • History of intraocular surgery (e.g., cataract surgery, vitrectomy, penetrating keratoplasty, or LASIK) within the last 3 months;
  • Current or any history of confirmed diagnosis of glaucoma (e.g., based on glaucoma visual field, nerve changes, or glaucoma filtering surgery);
  • Current or any history of retinal vascular occlusion or proliferative diabetic retinopathy;
  • Expected to have cataract removal surgery during the study;
  • History or current evidence of ocular disease that, in the opinion of the investigator, may confound assessment of visual function;
  • History of treatment for retinitis pigmentosa that could affect the progression of retinal degeneration (including participation in a clinical trial within the last year or a retained drug delivery device).
  • If either ear has any of the following, the patient is not eligible:
  • The audiometric PTA(1-2-4kHz) for the best hearing ear should not exceed 75dB HL;
  • Patients with bilateral cochlear implants cannot participate in the study;
  • A planned, second, cochlear implantation during the study.

Key Trial Info

Start Date :

February 11 2019

Trial Type :

OBSERVATIONAL

Allocation :

ACTUAL

End Date :

March 2 2024

Estimated Enrollment :

36 Patients enrolled

Trial Details

Trial ID

NCT04820244

Start Date

February 11 2019

End Date

March 2 2024

Last Update

April 29 2022

Active Locations (1)

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Radboud universitair medisch centrum

Nijmegen, Gelderland, Netherlands, 6525 GA