NCT07038239

Brief Summary

The Microrchidia CW-type zinc finger 2 (MORC2) gene encodes a protein expressed in all tissues and enriched in the brain. It is involved in Charcot-Marie-Tooth disease, with mire than 30 families presenting MORC2 mutations. Recently, MORC2 mutation have been shown to be responsible for more complex phenotypes like DIFGAN: developmental delay, impaired growth, dysmorphic facies and axonal neuropathy. Different mutations are responsible from a diverse spectrum of phenotype, from CMT to DIFGAN. MORC2 is involved, through its ATPase activity, in DNA repair, chromatin remodeling and epigenetic silencing via the Human silencing hub (HUSH) complex. Our hypothesis is that the hypo- or hyper-activation of the HUSH complex by different MORC2 mutations could be responsible for different phenotypes in patients. The aim of this study is to perform a genotype-phenotype correlation study in patients presenting MORC2 mutations.

Trial Health

77
On Track

Trial Health Score

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

Enrollment
45

participants targeted

Target at P25-P50 for all trials

Timeline
16mo left

Started Sep 2025

Geographic Reach
1 country

12 active sites

Status
recruiting

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 Progress34%
Sep 2025Sep 2027

First Submitted

Initial submission to the registry

May 27, 2025

Completed
1 month until next milestone

First Posted

Study publicly available on registry

June 26, 2025

Completed
2 months until next milestone

Study Start

First participant enrolled

September 1, 2025

Completed
1 year until next milestone

Primary Completion

Last participant's last visit for primary outcome

September 1, 2026

Expected
1 year until next milestone

Study Completion

Last participant's last visit for all outcomes

September 1, 2027

Last Updated

July 4, 2025

Status Verified

July 1, 2025

Enrollment Period

1 year

First QC Date

May 27, 2025

Last Update Submit

July 3, 2025

Conditions

Keywords

Charcot-Marie-ToothDIFGANHuman silencing hub complexgenotype-phenotype correlationMORC2DNA dalage repairtranscriptional modulationEPIGENETIC

Outcome Measures

Primary Outcomes (1)

  • Epigenetic biomarker levels

    Quantification of epigenetic biomarkers in patient and control-derived cells (number of reads in patients vs controls)

    At inclusion

Secondary Outcomes (5)

  • RNA-seq

    At inclusion

  • Detection and quantification of specific nucleic acid biomarkers in patient's serum

    At inclusion

  • Quantification of nucleic acid biomarkers in patient's cerebrospinal fluid

    At inclusion

  • Quantification of proteic biomarkers in patient's serum

    At inclusion

  • Quantification of proteic biomarkers in patient's cerebrospinal fluid (CSF)

    At inclusion

Study Arms (3)

Charcot-Marie-Tooth patients : Patients presenting with length-dependent sensitive-motor axonal neur

Patients presenting with length-dependent sensitive-motor axonal neuropathy

Diagnostic Test: Skin biopsyDiagnostic Test: Blood sample

DIFGAN patients

Patients presenting with DIFGAN syndrome : developmental delay, impaired growth, dysmorphic facies and axonal neuropathy

Diagnostic Test: Skin biopsyDiagnostic Test: Blood sample

Control

Control group without any neurological disorder

Interventions

Skin biopsyDIAGNOSTIC_TEST

under the arm using a 3 mm punch, with local anaesthesia, in the investigating centres.

Charcot-Marie-Tooth patients : Patients presenting with length-dependent sensitive-motor axonal neurDIFGAN patients
Blood sampleDIAGNOSTIC_TEST

3 classical 4ml tubes samples per patients, using the routine blood sampling technique, in the investigating centres. For children, blood sampling volume will be adapted to the patient's weight according to L.1121-1 of the French public health code.

Charcot-Marie-Tooth patients : Patients presenting with length-dependent sensitive-motor axonal neurDIFGAN patients

Eligibility Criteria

Age4 Years+
Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64), Older Adult (65+)
Sampling MethodNon-Probability Sample
Study Population

This study will focus on children and adults suffering from a MORC2 gene mutation, and presenting a CMT or DIFGAN phenotype.

You may qualify if:

  • Presence of a mutation in the MORC2 gene, identified during an evaluation for peripheral neuropathy or intellectual disability
  • Affiliation with the national health insurance system
  • Informed consent from the patient if an adult, or from parents/legal guardians if the patient is a minor

You may not qualify if:

  • Presence of another mutation responsible for peripheral neuropathy or intellectual disability
  • Refusal to undergo biological sample collection
  • Pregnant, postpartum, or breastfeeding women
  • Individuals deprived of liberty by judicial or administrative decision
  • Individuals not affiliated with a social security system or not benefiting from an equivalent health coverage scheme

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (12)

CHU de Besançon

Besançon, 25030, France

RECRUITING

CHRU Brest

Brest, 29200, France

RECRUITING

CHU Grenoble

Grenoble, 38700, France

RECRUITING

CH de Versailles

Le Chesnay, 78150, France

RECRUITING

Service de Génétique moléculaire, pharmacogénétique, hormologie Hôpital Bicêtre

Le Kremlin-Bicêtre, 94270, France

RECRUITING

Hospices Civils de Lyon

Lyon, 69317, France

RECRUITING

CHU Marseille

Marseille, 13005, France

RECRUITING

CHU de Nantes

Nantes, 44000, France

RECRUITING

CH Pitié Salpêtrière

Paris, 75013, France

RECRUITING

Hôpital Necker

Paris, 75015, France

RECRUITING

CHU de Saint-Etienne

Saint-Etienne, 42270, France

RECRUITING

CHU Strasbourg

Strasbourg, 67000, France

RECRUITING

MeSH Terms

Conditions

Charcot-Marie-Tooth DiseaseDevelopmental Disabilities

Interventions

Blood Specimen Collection

Condition Hierarchy (Ancestors)

Hereditary Sensory and Motor NeuropathyNervous System MalformationsNervous System DiseasesHeredodegenerative Disorders, Nervous SystemNeurodegenerative DiseasesPolyneuropathiesPeripheral Nervous System DiseasesNeuromuscular DiseasesCongenital AbnormalitiesCongenital, Hereditary, and Neonatal Diseases and AbnormalitiesGenetic Diseases, InbornNeurodevelopmental DisordersMental Disorders

Intervention Hierarchy (Ancestors)

Specimen HandlingClinical Laboratory TechniquesDiagnostic Techniques and ProceduresDiagnosisPuncturesSurgical Procedures, OperativeInvestigative Techniques

Central Study Contacts

Study Design

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

Study Record Dates

First Submitted

May 27, 2025

First Posted

June 26, 2025

Study Start

September 1, 2025

Primary Completion (Estimated)

September 1, 2026

Study Completion (Estimated)

September 1, 2027

Last Updated

July 4, 2025

Record last verified: 2025-07

Data Sharing

IPD Sharing
Will not share

Locations