Research Into the Molecular Bases of a New Phenotype Combining Premature White Hair, Polycystic Kidney Disease, Aortic Dilation/Dissection and Lymphopenia

NCT ID: NCT03651388

Last Updated: 2018-08-29

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

COMPLETED

Total Enrollment

4 participants

Study Classification

OBSERVATIONAL

Study Start Date

2010-06-01

Study Completion Date

2012-12-31

Brief Summary

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This study involves a single family, including 1 patient, father, mother and sister. The patient presented with a new phenotype associating premature white hair, renal polycystosis, aortic dilation/dissection and lymphopenia. Samples were taken in order to identify the origin of the symptomatology highlighted in the index case.

In addition, it was observed that mice invalidated for bcl-2, normal at birth and indistinguishable from control mice, showed, after one week, a phenotype similar to that observed in this patient.

The overlap between the patient's main clinical signs (lymphopenia, white hair and polycystic renal disease) and the manifestations presented by the invalidated murine model for BCL2 suggests that its phenotype may be secondary to a Bcl-2 expression defect.

Detailed Description

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Conditions

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New Phenotype (Combining Premature White Hair, Polycystic Kidney Disease, Aortic Dilation/Dissection and Lymphopenia)

Study Design

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Observational Model Type

FAMILY_BASED

Study Time Perspective

OTHER

Study Groups

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Patient

Patient with a new phenotype combining premature white hair, renal polycystosis, aortic dilation/dissection and lymphopenia

No interventions assigned to this group

Related parties of the 1st degree

1st degree related family of Group A patient

No interventions assigned to this group

Eligibility Criteria

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Inclusion Criteria

This study involves a single family, including 1 patient, father, mother and sister.
Eligible Sex

ALL

Accepts Healthy Volunteers

Yes

Sponsors

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Centre Hospitalier Universitaire Dijon

OTHER

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Locations

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Chu Dijon Bourogne

Dijon, , France

Site Status

Countries

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France

Other Identifiers

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OLIVIER-FAIVRE AOI 2010

Identifier Type: -

Identifier Source: org_study_id

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