NCT00873678

Brief Summary

Primary objective:

  • assessment of the prevalence of AHI1 mutations in Joubert syndrome and cerebello-oculo-renal syndromes (JS/CORS) Secondary objective:
  • assessment of the prevalence of CEP290 mutations and NPHP1 homozygous deletions in JS/CORS
  • caracterization of mutational spectrum of AHI1, NPHP1, CEP290 genes in JS/CORS.
  • evaluation of genotype-phenotype correlation in JS/CORS.

Trial Health

87
On Track

Trial Health Score

Automated assessment based on enrollment pace, timeline, and geographic reach

Enrollment
80

participants targeted

Target at P50-P75 for all trials

Timeline
Completed

Started Mar 2007

Typical duration for all trials

Geographic Reach
1 country

1 active site

Status
completed

Health score is calculated from publicly available data and should be used for screening purposes only.

Trial Relationships

Click on a node to explore related trials.

Study Timeline

Key milestones and dates

Study Start

First participant enrolled

March 1, 2007

Completed
2.1 years until next milestone

First Submitted

Initial submission to the registry

March 31, 2009

Completed
1 day until next milestone

First Posted

Study publicly available on registry

April 1, 2009

Completed
9 months until next milestone

Primary Completion

Last participant's last visit for primary outcome

January 1, 2010

Completed
Same day until next milestone

Study Completion

Last participant's last visit for all outcomes

January 1, 2010

Completed
Last Updated

June 3, 2010

Status Verified

March 1, 2010

Enrollment Period

2.8 years

First QC Date

March 31, 2009

Last Update Submit

June 2, 2010

Conditions

Keywords

Molar toothAHI1 geneCEP290 geneciliopathies

Outcome Measures

Primary Outcomes (1)

  • Assessment of the prevalence of AHI1 mutations in Joubert syndrome and cerebello-oculo-renal syndromes (JS/CORS)

    At the inclusion visit

Secondary Outcomes (1)

  • Assessment of the prevalence of CEP290 mutations and NPHP1 homozygous deletions in JS/CORS ; Caracterization of mutational spectrum of AHI1, NPHP1, CEP290 genes in JS/CORS ; Evaluation of genotype-phenotype correlation in JS/CORS.

    At the inclusion visit

Study Arms (1)

1

Children or adult patients affected with JS/CORS

Biological: Whole blood sample

Interventions

Whole blood sample (10 ml)

1

Eligibility Criteria

Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64), Older Adult (65+)
Sampling MethodNon-Probability Sample
Study Population

Children or adult patients affected with JS/CORS

You may qualify if:

  • Child or adult patients without age maximum
  • Affected with JS/CORS défined by neurologic disease with at least one of the following symptoms :
  • neonatal hypotonia or developmental delay (before age 3) or mental retardation (QD\<70) (after age 3).
  • Ataxia
  • Oculomotor apraxia
  • and on MRI :
  • vermis hypoplasia/agenesia defined by insufficient development of cerebellar vermis.
  • And molar tooth defined by thickened, elongated and mal-orientated superior cerebellar peduncles on axial sections.

You may not qualify if:

  • Chromosomal anomalies identified by caryotype
  • Absence of signature of informed consent.
  • Absence of affiliation to social security

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (1)

Hopital Trousseau

Paris, 75012, France

Location

Biospecimen

Retention: SAMPLES WITH DNA

Whole blood sample (10 ml)

MeSH Terms

Conditions

Agenesis of Cerebellar VermisMeckel Syndrome, Type 4Ciliopathies

Condition Hierarchy (Ancestors)

Abnormalities, MultipleCongenital AbnormalitiesCongenital, Hereditary, and Neonatal Diseases and AbnormalitiesGenetic Diseases, Inborn

Study Officials

  • Lydie BURGLEN, MD

    Assistance Publique - Hôpitaux de Paris

    PRINCIPAL INVESTIGATOR

Study Design

Study Type
observational
Observational Model
FAMILY BASED
Time Perspective
CROSS SECTIONAL
Sponsor Type
OTHER

Study Record Dates

First Submitted

March 31, 2009

First Posted

April 1, 2009

Study Start

March 1, 2007

Primary Completion

January 1, 2010

Study Completion

January 1, 2010

Last Updated

June 3, 2010

Record last verified: 2010-03

Locations