NCT00001671

Brief Summary

Leukodystrophy is a disease of the white matter of the brain. White matter is the portion of the brain responsible for conducting electrical impulses from one area of the brain to the other. Insulating cells called myelin cover the brain and nerve cells in the white matter. If myelin becomes damaged electrical information cannot be transferred properly. Many patients suffering from leukodystrophies do not fit the description of any of the defined types of leukodystrophies and are therefore considered to have a leukodystrophy of unknown cause. The purpose of this study is to define groups of patients with leukodystrophies and to work toward finding the cause of the disorders. In order to do this, researchers will analyze patients with leukodystrophies of unknown causes. Patients will undergo clinical, neurophysiologic, biochemical, and genetic examinations and tests. Researchers believe that by studying these patients and their disorders they will be able to better understand the causes of myelin destruction, and eventually lead to effective treatments for these disorders.

Trial Health

90
On Track

Trial Health Score

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

Enrollment
400

participants targeted

Target at P75+ for all trials

Timeline
Completed

Started Sep 1997

Longer than P75 for all trials

Geographic Reach
4 countries

5 active sites

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

September 9, 1997

Completed
2.2 years until next milestone

First Submitted

Initial submission to the registry

November 3, 1999

Completed
1 day until next milestone

First Posted

Study publicly available on registry

November 4, 1999

Completed
9.1 years until next milestone

Study Completion

Last participant's last visit for all outcomes

December 15, 2008

Completed
Last Updated

July 2, 2017

Status Verified

December 15, 2008

First QC Date

November 3, 1999

Last Update Submit

June 30, 2017

Conditions

Keywords

White MatterMyelinDegenerative DiseasesGeneticOligodendrocytesLeukodystrophy

Eligibility Criteria

Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64), Older Adult (65+)

You may qualify if:

  • Candidates for participation in the protocol will be patients of all ages with clinical and radiographic signs of leukodystrophy who do not have a specific etiology despite a previous comprehensive workup. The preceding investigation would have excluded the following: adrenoleukodystrophy, adrenomyeloneuropathy, metachromatic leukodystrophy, Krabbe disease, Canavan disease, a well-defined amino acid organic acid disorder, or a systemic mitochondrial cytopathy.
  • First -degree relatives of patients with leukodystrophies of unknown etiology (father, mother, siblings, or sons and daughters of the patients)

You may not qualify if:

  • Refusal to sign the protocol consent form.
  • Candidates who are unable to travel to the National Institutes of Health Clinical Center.

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (5)

University of California, San Francisco

San Francisco, California, 94143, United States

Location

Childrens National Medical Center

Washington D.C., District of Columbia, United States

Location

Institut National de la Sante' et de la Recherche Medicale

Clermont-Ferrand, Cedex, 63001, France

Location

Tel Aviv University

Tel Aviv, Israel

Location

Academiseh Ziuekenhuis Vrije Universiteit

Amsterdam, Netherlands

Location

Related Publications (4)

  • Aicardi J. The inherited leukodystrophies: a clinical overview. J Inherit Metab Dis. 1993;16(4):733-43. doi: 10.1007/BF00711905.

    PMID: 7692130BACKGROUND
  • Schiffmann R, Moller JR, Trapp BD, Shih HH, Farrer RG, Katz DA, Alger JR, Parker CC, Hauer PE, Kaneski CR, et al. Childhood ataxia with diffuse central nervous system hypomyelination. Ann Neurol. 1994 Mar;35(3):331-40. doi: 10.1002/ana.410350314.

    PMID: 8122885BACKGROUND
  • Schiffmann R, Tedeschi G, Kinkel RP, Trapp BD, Frank JA, Kaneski CR, Brady RO, Barton NW, Nelson L, Yanovski JA. Leukodystrophy in patients with ovarian dysgenesis. Ann Neurol. 1997 May;41(5):654-61. doi: 10.1002/ana.410410515.

    PMID: 9153528BACKGROUND
  • McNeill N, Nasca A, Reyes A, Lemoine B, Cantarel B, Vanderver A, Schiffmann R, Ghezzi D. Functionally pathogenic EARS2 variants in vitro may not manifest a phenotype in vivo. Neurol Genet. 2017 Jul 14;3(4):e162. doi: 10.1212/NXG.0000000000000162. eCollection 2017 Aug.

MeSH Terms

Conditions

Lysosomal Storage Diseases

Condition Hierarchy (Ancestors)

Metabolism, Inborn ErrorsGenetic Diseases, InbornCongenital, Hereditary, and Neonatal Diseases and AbnormalitiesMetabolic DiseasesNutritional and Metabolic Diseases

Study Design

Study Type
observational
Sponsor Type
NIH

Study Record Dates

First Submitted

November 3, 1999

First Posted

November 4, 1999

Study Start

September 9, 1997

Study Completion

December 15, 2008

Last Updated

July 2, 2017

Record last verified: 2008-12-15

Locations