Clinical and Genetic Examination of Usher Syndrome Patients' Cohort in Europe

NCT ID: NCT01954953

Last Updated: 2015-02-04

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

UNKNOWN

Total Enrollment

100 participants

Study Classification

OBSERVATIONAL

Study Start Date

2013-09-30

Brief Summary

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This study aims to characterize Usher patients in order to correlate this data with genetic information.

Tasks:

* Standardization and improvement of Usher syndrome diagnosis: refine and elaborate special tests of visual and otological function in association with genotype that enable to determine the most significant markers for Usher disease progression and therapeutic effect.
* Perform genotype and phenotype correlations in Usher syndrome patients
* Develop and maintain database for phenotypically and genotypically well-characterized patient cohorts, suitable for future therapeutic trials

Detailed Description

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Conditions

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Usher Syndrome

Study Design

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Observational Model Type

COHORT

Study Time Perspective

PROSPECTIVE

Study Groups

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no intervention

No interventions assigned to this group

Eligibility Criteria

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Inclusion Criteria

* Clinical characteristics for USH1, USH2 and USH3 as defined by the Usher syndrome consortium;
* Informed consent and agreement to participate in the study;
* Distance best corrected visual acuity ≥ 0.1.

Exclusion Criteria

* Systemic pathologies or severe ocular pathologies, systemic or topical medication usage, and/or other otolaryngology pathologies which could contaminate the results;
* Unwillingness to provide a blood sample ;
* Unwilling and/or unable to undergo the study procedures.
Minimum Eligible Age

6 Months

Maximum Eligible Age

70 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Institut National de la Santé Et de la Recherche Médicale, France

OTHER_GOV

Sponsor Role collaborator

Centre Hospitalier National d'Ophtalmologie des Quinze-Vingts

OTHER

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Locations

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CHU, Laboratoire de génétique moléculaire, INSERM

Montpellier, , France

Site Status RECRUITING

CIC of CHNO des Quinze-vingts

Paris, , France

Site Status RECRUITING

Johannes Gutenberg University of Mainz, Institute of Zoology, Dept. Cell and Matrix Biology Mainz

Mainz, , Germany

Site Status RECRUITING

Radboud University Nijmegen Medical Centre, Dept. Otorhinolaryngology

Nijmegen, , Netherlands

Site Status RECRUITING

AIBILI, 4C - Coimbra Coordinating Centre for Clinical Research

Coimbra, , Portugal

Site Status RECRUITING

IBILI- Faculty of Medicine - University of Coimbra, Center for Hereditary Diseases and Visual Neurosciences Laboratory

Coimbra, , Portugal

Site Status RECRUITING

Countries

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France Germany Netherlands Portugal

Central Contacts

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Ieva Sliesoraityte, MD PhD

Role: CONTACT

Facility Contacts

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Christel Vaché, PhD

Role: primary

Laurent Vinet

Role: primary

+33140021126

Kerstin Nagel-Wolfrum, PhD

Role: primary

Erwin van Wijk, PhD

Role: primary

Leal Sérgio Casimiro da Silva

Role: primary

Eduardo José Gil Duarte Silva, MD PhD

Role: primary

References

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Sliesoraityte I, Troeger E, Bernd A, Kurtenbach A, Zrenner E. Correlation between spectral domain OCT retinal nerve fibre layer thickness and multifocal pattern electroretinogram in advanced retinitis pigmentosa. Adv Exp Med Biol. 2012;723:471-8. doi: 10.1007/978-1-4614-0631-0_59. No abstract available.

Reference Type BACKGROUND
PMID: 22183366 (View on PubMed)

Overlack N, Goldmann T, Wolfrum U, Nagel-Wolfrum K. Gene repair of an Usher syndrome causing mutation by zinc-finger nuclease mediated homologous recombination. Invest Ophthalmol Vis Sci. 2012 Jun 26;53(7):4140-6. doi: 10.1167/iovs.12-9812.

Reference Type BACKGROUND
PMID: 22661463 (View on PubMed)

Goldmann T, Rebibo-Sabbah A, Overlack N, Nudelman I, Belakhov V, Baasov T, Ben-Yosef T, Wolfrum U, Nagel-Wolfrum K. Beneficial read-through of a USH1C nonsense mutation by designed aminoglycoside NB30 in the retina. Invest Ophthalmol Vis Sci. 2010 Dec;51(12):6671-80. doi: 10.1167/iovs.10-5741. Epub 2010 Jul 29.

Reference Type BACKGROUND
PMID: 20671281 (View on PubMed)

Kersten FF, van Wijk E, Hetterschijt L, Baubeta K, Peters TA, Aslanyan MG, van der Zwaag B, Wolfrum U, Keunen JE, Roepman R, Kremer H. The mitotic spindle protein SPAG5/Astrin connects to the Usher protein network postmitotically. Cilia. 2012 Apr 25;1(1):2. doi: 10.1186/2046-2530-1-2.

Reference Type BACKGROUND
PMID: 23351521 (View on PubMed)

Overlack N, Kilic D, Bauss K, Marker T, Kremer H, van Wijk E, Wolfrum U. Direct interaction of the Usher syndrome 1G protein SANS and myomegalin in the retina. Biochim Biophys Acta. 2011 Oct;1813(10):1883-92. doi: 10.1016/j.bbamcr.2011.05.015. Epub 2011 Jul 13.

Reference Type BACKGROUND
PMID: 21767579 (View on PubMed)

Estrada-Cuzcano A, Koenekoop RK, Senechal A, De Baere EB, de Ravel T, Banfi S, Kohl S, Ayuso C, Sharon D, Hoyng CB, Hamel CP, Leroy BP, Ziviello C, Lopez I, Bazinet A, Wissinger B, Sliesoraityte I, Avila-Fernandez A, Littink KW, Vingolo EM, Signorini S, Banin E, Mizrahi-Meissonnier L, Zrenner E, Kellner U, Collin RW, den Hollander AI, Cremers FP, Klevering BJ. BBS1 mutations in a wide spectrum of phenotypes ranging from nonsyndromic retinitis pigmentosa to Bardet-Biedl syndrome. Arch Ophthalmol. 2012 Nov;130(11):1425-32. doi: 10.1001/archophthalmol.2012.2434.

Reference Type BACKGROUND
PMID: 23143442 (View on PubMed)

Garcia-Garcia G, Besnard T, Baux D, Vache C, Aller E, Malcolm S, Claustres M, Millan JM, Roux AF. The contribution of GPR98 and DFNB31 genes to a Spanish Usher syndrome type 2 cohort. Mol Vis. 2013;19:367-73. Epub 2013 Feb 13.

Reference Type BACKGROUND
PMID: 23441107 (View on PubMed)

Vache C, Besnard T, le Berre P, Garcia-Garcia G, Baux D, Larrieu L, Abadie C, Blanchet C, Bolz HJ, Millan J, Hamel C, Malcolm S, Claustres M, Roux AF. Usher syndrome type 2 caused by activation of an USH2A pseudoexon: implications for diagnosis and therapy. Hum Mutat. 2012 Jan;33(1):104-8. doi: 10.1002/humu.21634. Epub 2011 Nov 16.

Reference Type BACKGROUND
PMID: 22009552 (View on PubMed)

Stoetzel C, Laurier V, Davis EE, Muller J, Rix S, Badano JL, Leitch CC, Salem N, Chouery E, Corbani S, Jalk N, Vicaire S, Sarda P, Hamel C, Lacombe D, Holder M, Odent S, Holder S, Brooks AS, Elcioglu NH, Silva ED, Rossillion B, Sigaudy S, de Ravel TJ, Lewis RA, Leheup B, Verloes A, Amati-Bonneau P, Megarbane A, Poch O, Bonneau D, Beales PL, Mandel JL, Katsanis N, Dollfus H. BBS10 encodes a vertebrate-specific chaperonin-like protein and is a major BBS locus. Nat Genet. 2006 May;38(5):521-4. doi: 10.1038/ng1771. Epub 2006 Apr 2.

Reference Type BACKGROUND
PMID: 16582908 (View on PubMed)

Castelo-Branco M, Mendes M, Sebastiao AR, Reis A, Soares M, Saraiva J, Bernardes R, Flores R, Perez-Jurado L, Silva E. Visual phenotype in Williams-Beuren syndrome challenges magnocellular theories explaining human neurodevelopmental visual cortical disorders. J Clin Invest. 2007 Dec;117(12):3720-9. doi: 10.1172/JCI32556.

Reference Type BACKGROUND
PMID: 18037993 (View on PubMed)

Goldmann T, Overlack N, Wolfrum U, Nagel-Wolfrum K. PTC124-mediated translational readthrough of a nonsense mutation causing Usher syndrome type 1C. Hum Gene Ther. 2011 May;22(5):537-47. doi: 10.1089/hum.2010.067. Epub 2011 Mar 25.

Reference Type BACKGROUND
PMID: 21235327 (View on PubMed)

Related Links

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Other Identifiers

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P13-02

Identifier Type: -

Identifier Source: org_study_id

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