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Natural History Study of Usher Syndrome ( Light4Deaf )

Recruiting
Conditions
Usher Syndromes
Registration Number
NCT04665726
Lead Sponsor
Centre Hospitalier National d'Ophtalmologie des Quinze-Vingts
Brief Summary

Clinical centres in the LIGHT4DEAF consortium have developed and will continue to improve a reliable, early molecular diagnosis and protocols for full clinical characterisation of Usher syndrome, which will be valuable for the foreseen USH clinical trials. The clinical arm of the project aims at performing a deep-phenotyping of retinal degeneration, hearing loss, vestibular dysfunction, neurocognitive ability of subects with a molecular diagnosis of any Usher syndrome. Functional and structural parameters for retinal, auditory, and vestibular impairments are followed overtime to document the natural history of the disease and establish relevant clinical endpoint for disease progression that may be useful for future clinical trials.

Detailed Description

Our cohort study aims at precisely documenting ophthalmic, auditory, vestibular, cogninitive alterations over time with phenotype/genotype correlation Ophthalmological assessment; Best corrected visual acuity, kynetic perimetry, microperimetry, colour contrast sensitivity, retinal multimodal imaging (fundus photograph, fundus autofluorescence, SD-OCT, OCTA, adaptive optics)

ENT assessment:

Tone and voice audiometry, Distortion product otoacoustic emissions Language assessment for children

Vestibular assessment:

Complete assessment of vestibular, canal and otolithic function Neuro-cognitive and visio spatial assessment Genetic: deep-genotyping using next generation sequencing

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
400
Inclusion Criteria
  • Patient with a molecular diagnosis of Usher syndrome type I, II or III or a clinical diagnosis of Usher syndrome type I, II or III which will then be confirmed by a molecular diagnosis
  • Health insurance beneficiary
  • Informed consent signed by the patient or their legal representatives
Exclusion Criteria

• Patient or his/her legal representatives unable to understand the study and for whom informed consent cannot be obtained

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Primary Outcome Measures
NameTimeMethod
5-year natural history of Usher syndromeFrom date of inclusion until the date of last documented progression , assessed up to 5 years

Phenotype/genotype correlation, structure function correlation and progression of structural and functional parameters

Secondary Outcome Measures
NameTimeMethod

Trial Locations

Locations (4)

Centre Hospitalier National d'Ophtalmologie des Quinze-Vingts

🇫🇷

Paris, France

CHU Necker

🇫🇷

Paris, France

CHU Robert Debré

🇫🇷

Paris, France

CHU Pitié Salpêtrière

🇫🇷

Paris, France

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