NCT04288128

Brief Summary

One of the main objectives of this project is to validate potential biological, clinical and/or imaging biomarkers in SCA patients through a multimodal assessment, for future ASOs trials.

Trial Health

87
On Track

Trial Health Score

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

Enrollment
40

participants targeted

Target at P25-P50 for all trials

Timeline
Completed

Started May 2020

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

First Submitted

Initial submission to the registry

February 17, 2020

Completed
11 days until next milestone

First Posted

Study publicly available on registry

February 28, 2020

Completed
3 months until next milestone

Study Start

First participant enrolled

May 28, 2020

Completed
2 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

May 20, 2022

Completed
12 days until next milestone

Study Completion

Last participant's last visit for all outcomes

June 1, 2022

Completed
Last Updated

November 18, 2025

Status Verified

January 1, 2025

Enrollment Period

2 years

First QC Date

February 17, 2020

Last Update Submit

November 14, 2025

Conditions

Keywords

Spinocerebellar ataxiaMultimodal approachBiomarkerAntisense oligonucleotides (ASOs) therapy

Outcome Measures

Primary Outcomes (1)

  • Identification of biological, clinical and/or imaging biomarkers in SCA2 and SCA7 patients mutations carriers and patients through a multimodal assessment over one year to prepare therapeutic trials

    Over one year

Secondary Outcomes (17)

  • To determine the cross-sectional and longitudinal variability of SARA (Scale for the Assessment and Rating of Ataxia) and CCFS (Composite Cerebellar Functional Score) scores in SCA 2 and SCA 7 gene mutation carriers and healthy controls over one

    Over one year

  • Determine the cross-sectional and longitudinal variability of volumetric MRI and NMR-proto spectroscopy in SCA 2 and SCA 7 gene mutation carriers and healthy controls

    Over one year

  • Delineate a specific pattern of frontal-like cognitive deficit in SCAs gene carriers

    Over one year

  • To determine the cross-sectional and longitudinal variability of CSF, blood and urine biomarkers in SCAs gene mutation carriers and controls

    Over one year

  • To explore the relationship of CSF, blood and urine biomarker levels in relation to clinical and imaging markers of disease progression

    Over one year

  • +12 more secondary outcomes

Study Arms (2)

SCA early-manifest and premanifest patients

This cohort is defined by individuals with a SARA score between 0 and 15 (both values included).

Procedure: Lumbar punctureOther: Magnetic Resonance Imaging (MRI)

Control participants

This cohort is defined by individuals with a SARA score less than 5 and no significant neurological symptoms.

Procedure: Lumbar punctureOther: Magnetic Resonance Imaging (MRI)

Interventions

Each participant will undergo lumbar puncture at first visit (M0) and last visit (M12)

Control participantsSCA early-manifest and premanifest patients

Each participant will undergo scanning at 3 visits (M0, M6 and M12)

Control participantsSCA early-manifest and premanifest patients

Eligibility Criteria

Age18 Years+
Sexall
Healthy VolunteersYes
Age GroupsAdult (18-64), Older Adult (65+)
Sampling MethodProbability Sample
Study Population

1. Early stage subjects and premanifest mutation carriers refer to individuals who tested positive for the SCA 2 or 7 gene mutation and SARA score between 0 and 15 (both values included) 2. Control participants refer to individuals with non-mutation carriers.

You may qualify if:

  • Ability to walk independently 30 foot without an assistive device
  • Able to stand unassisted for 30 seconds
  • Affiliated with the French social security, or a social security equivalent, if they are not French.
  • Capacity to consent
  • Signed Informed Consent by the subject
  • Ability to undergo MRI scanning
  • Genetic diagnosis of SCA 2 or 7 (available CAG repeat length)
  • SARA score ≤15
  • Negative Genetic diagnosis of SCA2/SCA7 available
  • No significant neurological symptoms
  • SARA score \< 5
  • Ability to undergo a lumbar puncture

You may not qualify if:

  • Subjects currently receiving, or having received within 2 months prior to enrolment into this study, any investigational drug
  • Pregnancy or breastfeeding
  • Genotype consistent with other inherited ataxias
  • Changes in coordinative physical and occupational therapy for ataxia 2 months prior to study participation
  • Concomitant disorder(s) or condition(s) that affects assessment of ataxia or severity of ataxia during this study
  • Contra-indications to MRI examination
  • Person deprived of their liberty by judicial or administrative decision

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (1)

Institut du Cerveau - Paris Brain Institute

Paris, 75013, France

Location

Related Publications (2)

  • Coarelli G, Dubec-Fleury C, Petit E, Sayah S, Fischer C, Nassisi M, Gatignol P, Dorgham K, Daghsen L, Daye P, Cunha P, Kacher R, Hilab R, Hurmic H, Lamaziere A, Lamy JC, Welter ML, Chupin M, Mangin JF, Lane R, Gaymard B, Pouget P, Audo I, Brice A, Tezenas du Montcel S, Durr A. Longitudinal Changes of Clinical, Imaging, and Fluid Biomarkers in Preataxic and Early Ataxic Spinocerebellar Ataxia Type 2 and 7 Carriers. Neurology. 2024 Sep 10;103(5):e209749. doi: 10.1212/WNL.0000000000209749. Epub 2024 Aug 12.

  • Nassisi M, Coarelli G, Blanchard B, Dubec-Fleury C, Drine K, Kitic N, Sancho S, Hilab R, Tezenas du Montcel S, Junge C, Lane R, Arnold HM, Durr A, Audo I. ATXN7-Related Cone-Rod Dystrophy: The Integrated Functional Evaluation of the Cerebellum (CERMOI) Study. JAMA Ophthalmol. 2024 Apr 1;142(4):301-308. doi: 10.1001/jamaophthalmol.2024.0001.

Biospecimen

Retention: SAMPLES WITH DNA

Whole blood collected for DNA analysis

MeSH Terms

Conditions

Spinocerebellar Ataxias

Interventions

Spinal PunctureMagnetic Resonance Spectroscopy

Condition Hierarchy (Ancestors)

Cerebellar AtaxiaCerebellar DiseasesBrain DiseasesCentral Nervous System DiseasesNervous System DiseasesSpinocerebellar DegenerationsSpinal Cord DiseasesHeredodegenerative Disorders, Nervous SystemNeurodegenerative DiseasesAtaxiaDyskinesiasNeurologic ManifestationsGenetic Diseases, InbornCongenital, Hereditary, and Neonatal Diseases and Abnormalities

Intervention Hierarchy (Ancestors)

BiopsySpecimen HandlingClinical Laboratory TechniquesDiagnostic Techniques and ProceduresDiagnosisDiagnostic Techniques, NeurologicalPuncturesTherapeuticsSurgical Procedures, OperativeInvestigative TechniquesSpectrum AnalysisChemistry Techniques, Analytical

Study Officials

  • Alexandra DURR

    Institut du Cerveau - Paris Brain Institute

    PRINCIPAL INVESTIGATOR

Study Design

Study Type
observational
Observational Model
COHORT
Time Perspective
PROSPECTIVE
Sponsor Type
OTHER GOV
Responsible Party
SPONSOR

Study Record Dates

First Submitted

February 17, 2020

First Posted

February 28, 2020

Study Start

May 28, 2020

Primary Completion

May 20, 2022

Study Completion

June 1, 2022

Last Updated

November 18, 2025

Record last verified: 2025-01

Locations