Muscular DystrophiesCYTOKINE-FSHNCT04694456
Pro-inflammatory Cytokines in Facioscapulohumeral Muscular Dystrophy (CYTOKINE-FSH)
A study testing cytokines dosage, test of walk and Manual muscular test in people with Muscular Dystrophies, run by Centre Hospitalier Universitaire de Nice at 1 site in France.
Trial summary
The facial-glenohumeral muscular dystrophy type 1 (DMFSH1) is characterized by a selective and asymmetrical involvement of the facial muscles, the shoulder girdle and the anterolateral lodge legs. Read more about this study
- Who is it for?
- Ages 18 to 75 years
- Conditions the study is for
- Muscular Dystrophies
- What is being tested?
- cytokines dosage, test of walk and Manual muscular test
- Type of treatment
- Surgery or procedure
- Could I receive a placebo?
- No, everyone receives an active treatment
- How many people
- 30 participants
- Where
- 1 siteFrance
- Study dates
- 30 Jan 2018to 18 May 2021
- Phase
- Interventional
- Are there results?
- None posted in the registry
Key requirements
Who can take part, as listed by the sponsor. Only the study team can confirm whether you are eligible, so take this list to your doctor.
- Ages 18 to 75 years
- All sexes
- No healthy volunteers
- Adult, Older adult
Who may be able to join
- male or female age 18 to 75
- suffering from genetically confirmed FSHD1 (<11 D4Z4 repeat units on permissive chromosome 4 allele)
- ambulant or walking with assistance
- Manual Muscle Testing ≥4 for 1 of lower limb muscles
Who cannot join
- pregnancy or breast feeding
- stay in tropical/subtropical country within 3 months
- physical exercice within 10 hours
- specific diet (e.g. hypocaloric or cholesterol lowering diet)
- regular alcohol consumption; drug consumption within 3 months
- immunosuppressive or immonumodulating drug within 2 weeks or for more than 3 months withing last 6 months
Show all 13Show fewer
- vaccination, blood transfusion of immunoglobulin treatment within 3 months
- infection within 3 weeks; HIV, HBV, HCV seropositivity
- chronic inflammatory and/or autoimmune or allergic disease from the gut (Crohn disease, ulcerative colitis), skin (psoriasis, atopic dermatitis), joints (rhumatoid arthritis), nervous system (multiple sclerosis), diabetes type I and II
- neurodegenerative disorders (Alzheimer's or Parkinson's diseases)
- diagnosed cancer not under remission for at least 5 years
- participation in the last 3 months in a research clinical trial with exposure to a pharmaceutical product or a medical device
- muscular MRI contraindication
Trial locations and contacts
1 site in 1 country.
About this study
Official title: Pro-inflammatory Cytokines as Potential Therapeutic Target in Type 1 Facioscapulohumeral Muscular Dystrophy: Pilot Study
The facial-glenohumeral muscular dystrophy type 1 (DMFSH1) is characterized by a selective and asymmetrical involvement of the facial muscles, the shoulder girdle and the anterolateral lodge legs. Genetically, the disease is transmitted in an autosomal dominant manner and is caused by a pathogen contraction of repeat units (UR) say D4Z4 localized to the telomeric portion of chromosome 4qA. The loss of UR causes hypomethylation of DNA and chromatin relaxation of the region that lead to inappropriate expression of DUX4 retrogene highly toxic. The inappropriate expression induces a T cell reaction inflammatory response that participate and increase muscle damage. In favor of this hypothesis, several muscle MRI studies have shown that atrophy and fibro-adipose degeneration (hyper signal in T1) were preceded by the appearance of muscle inflammation (hyper signal T2STIR) confirmed on histologically and dysregulation of genes involved in adaptive and innate immunity. scientific hypothesis and potential benefits: the investigateur hypothesize that in patients of DMFSH1, the immune system cells may participate in the pathophysiology of the disease through changes in serum secretion of one or more cytokines and / or a modification of the response of inflammatory cells in some cell damage stimuli. Design: this is a single-center pilot study, interventional. In this study, the investigator will assay the serum cytokines and changes in peripheral blood cells of the expression of cytokines in response to some stimuli in 20 patients with Type 1 DMFSH genetically confirmed at an intermediate stage of clinical disease (kept walking, but at least one muscle of lower limbs reached) and compare with controls from the CYTOKINAGE study. The investigator will also carry patients clinical testing (MMT sum score) and functional (6minute test march MFM) and a MRI not injected whole body (T1 sequences + and T2STIR) to study the relationship between these parameters and secretion cytokines or serum in response to certain stimuli Main objective: to compare serum levels of IL-6 in patients with DMFSH and controls.
- Conditions
- Muscular Dystrophies
- Being tested
- cytokines dosage, test of walk, Manual muscular test, Motrice fonction mesurement
- Medical subject headings
- Muscular Dystrophies
What participants receive
What participants receive. In a randomised study each person is assigned to one group by chance.
cytokines dosageOther
Also called biological analysis
Mesure of cytokines concentration in serum
test of walkProcedure or surgery
patient must walk during 6 minutes on a flat surface
Manual muscular testProcedure or surgery
test performed to evaluate the patient muscular weakness
Motrice fonction mesurementProcedure or surgery
scale allowing the evaluation of patient posture and upper body movements
| Study group | Role | What they receive |
|---|---|---|
| Facioscapulohumeral muscular dystrophy | Experimental | Adult ambulant patients with facioscapulohumeral muscular dystrophy type 1 (FSHD1) Receives: Other: cytokines dosage; Procedure: test of walk; Procedure: Manual muscular test; Procedure: Motrice fonction mesurement |
What is being measured
The primary outcome is the main question the study is designed to answer.
Comparison of serum IL-6 levels between FSHD1 patients and control subjects (from other previous studies) comparable in terms of age and sex
Serum IL-6 levels will be measured using v-plex technology (MSD) in FSHD1 patients and compared with results obtained in paired control subjects from 2 cohorts (NCT00998231 and Cytokinage study NCT02660723)
Measured: 21 months
Secondary outcomes (4)
Comparison of serum levels of 28 oher pro-inflammatory cytokines between FSHD1 patients and control subjects
Serum levels of 28 other cytokines will be measured using v-plex technology (MSD) in FSHD1 patients and compared with results obtained in paired control subjects
Measured: 21 months
Comparison of cytokines produced upon in vitro stimulation of blood cells in FSHD1 patients and control subjects
Cytokine production will be induced in blood cells by non-specific stimulation with LPS, ATP+LPS-EB or poly (I:C) in FSHD1 patients and compared with results obtained in control subjects
Measured: 21 months
Evaluation of potential correlations between cytokines levels (either in the serum or produced upon in vitro stimulation) and clinical severity in FSHD1 patients
Clinical severity and muscle impairment will be evaluated using standardized clinical scales (Manual Muscle Testing, Motor Function Measure-32, 6-minute walk test, age-corrected Clinical Severity Score)
Measured: 21 months
Evaluation of potential correlations between cytokines levels (either in the serum or produced upon in vitro stimulation) and muscle MRI caracteristics in FSHD1 patients
Whole body MRI will be recorded (T1 and T2STIR sequences) in order to evaluate muscle oedema, inflammation and degeneration
Measured: 21 months
Timeline
From the start of the study to its completion; the mark is today. Estimated dates are the sponsor’s plan.
- First posted5 Jan 2021
- Study start30 Jan 2018
- Primary completion18 May 2021
- Study completion18 May 2021
Resources
- Official record on ClinicalTrials.govThe registry entry this page is built from, with its full change history.
- Publications (2)Papers the sponsor has linked to this study.
- Other trials for Muscular DystrophiesStudies of the same condition, open and under way.
- More trials run by Centre Hospitalier Universitaire de NiceThe sponsor's other registered studies.
- What the trial phases meanHow a treatment moves from first tests in people to approval.
More details
For doctors and researchers: how the study is designed, who runs it, and its registry IDs.
Study design
- Study type
- Interventional
- Phase
- Phase N/A
- Primary purpose
- Other
- Allocation
- Not applicable
- Intervention model
- Single group
- Model description
- Interventional pilot study
- Masking
- None (open label)
- Enrolment
- 30 participants (actual)
Sponsor and investigators
- Lead sponsor
- Centre Hospitalier Universitaire de Nice
- Sponsor type
- University, hospital or other
- Responsible party
- Sponsor
IDs, dates and publications
- ClinicalTrials.gov ID
- NCT04694456
- Sponsor's protocol ID
- 16-AOI-13
- Acronym
- CYTOKINE-FSH
- Status last verified
- Mar 2026
- First submitted
- 31 Dec 2020
- Last updated
- 24 Mar 2026
- Results posted
- No
- Data monitoring committee
- No
- FDA-regulated drug
- No
- FDA-regulated device
- No
- Individual data sharing
- No
Publications
Sanson B, Slioui A, Garcia J, Klouvi L, Lejeune J, Stalens C, Guien C, Rabarimeriarijaona S, Bernard R, Nectoux J, Attarian S, Bedat-Millet AL, Bouhour F, Boyer FC, Chanson JB, Choumert A, Cintas P, De La Cruz E, Feasson L, Fournier M, Ghorab K, Jacquin-Piques A, Laforet P, Magot A, Michaud M, Noury JB, Sole G, Spinazzi M, Stojkovic T, Tard C, Villa L, Beroud C, Sacconi S; French FSHD registry collaboration group. Prevalence and predictors of uncommon features in FSHD1 patients: insights from the French FSHD registry. Orphanet J Rare Dis. 2025 Sep 2;20(1):470. doi: 10.1186/s13023-025-03877-z.
PubMed 40898356Pini J, Martinuzzi E, Dhifallah S, Slioui A, Puma A, Villa L, Cavalli M, Ezaru A, Garcia J, Gambella M, Torre F, Pavan LJ, Glaichenhaus N, Sacconi S. Interleukin-6 as a Key Biomarker in Facioscapulohumeral Dystrophy: Evidence From Longitudinal Analyses. Ann Clin Transl Neurol. 2026 Feb;13(2):310-323. doi: 10.1002/acn3.70210. Epub 2025 Oct 7.
PubMed 41058127
Study at a glance
From the registry record, last updated by the sponsor on 24 Mar 2026. Recruitment status is as the sponsor reported it: confirm with the study team.
- Official title
- Pro-inflammatory Cytokines as Potential Therapeutic Target in Type 1 Facioscapulohumeral Muscular Dystrophy: Pilot Study
- ClinicalTrials.gov ID
- NCT04694456
- Acronym / sponsor ID
- CYTOKINE-FSH / 16-AOI-13
- Conditions
- Muscular Dystrophies
- Study type
- Interventional
- Recruitment status
- Completed
- Enrolment
- 30 participants (actual)
- Lead sponsor
- Centre Hospitalier Universitaire de NiceUniversity, hospital or other
- Study start
- 30 Jan 2018 (actual)
- Primary completion
- 18 May 2021 (actual)
- Study completion
- 18 May 2021 (actual)
- First posted
- 5 Jan 2021 (actual)
- Results
- Not posted in the source record
Clinical summary
The research question as the sponsor registered it. This describes what the study plans to test, not what it found.
The facial-glenohumeral muscular dystrophy type 1 (DMFSH1) is characterized by a selective and asymmetrical involvement of the facial muscles, the shoulder girdle and the anterolateral lodge legs. Genetically, the disease is transmitted in an autosomal dominant manner and is caused by a pathogen contraction of repeat units (UR) say D4Z4 localized to the telomeric portion of chromosome 4qA. The loss of UR causes hypomethylation of DNA and chromatin relaxation of the region that lead to inappropriate expression of DUX4 retrogene highly toxic. The inappropriate expression induces a T cell reaction inflammatory response that participate and increase muscle damage. In favor of this hypothesis, several muscle MRI studies have shown that atrophy and fibro-adipose degeneration (hyper signal in T1) were preceded by the appearance of muscle inflammation (hyper signal T2STIR) confirmed on histologically and dysregulation of genes involved in adaptive and innate immunity. scientific hypothesis and potential benefits: the investigateur hypothesize that in patients of DMFSH1, the immune system cells may participate in the pathophysiology of the disease through changes in serum secretion of one or more cytokines and / or a modification of the response of inflammatory cells in some cell damage stimuli. Design: this is a single-center pilot study, interventional. In this study, the investigator will assay the serum cytokines and changes in peripheral blood cells of the expression of cytokines in response to some stimuli in 20 patients with Type 1 DMFSH genetically confirmed at an intermediate stage of clinical disease (kept walking, but at least one muscle of lower limbs reached) and compare with controls from the CYTOKINAGE study. The investigator will also carry patients clinical testing (MMT sum score) and functional (6minute test march MFM) and a MRI not injected whole body (T1 sequences + and T2STIR) to study the relationship between these parameters and secretion cytokines or serum in response to certain stimuli Main objective: to compare serum levels of IL-6 in patients with DMFSH and controls.
Study design
- Primary purpose
- Other
- Allocation
- Not applicable
- Intervention model
- Single group
- Masking
- None (open label)
- Number of arms
- 1
- Model description
- Interventional pilot study
| Arm | Type | Receives |
|---|---|---|
| Facioscapulohumeral muscular dystrophy | Experimental | Other: cytokines dosage, Procedure: test of walk, Procedure: Manual muscular test, Procedure: Motrice fonction mesurementAdult ambulant patients with facioscapulohumeral muscular dystrophy type 1 (FSHD1) |
Who may qualify
- Ages
- 18 to 75 years
- Sex
- All
- Healthy volunteers
- Not accepted
- Age groups
- Adult, Older adult
Key inclusion criteria (4 listed)
- male or female age 18 to 75
- suffering from genetically confirmed FSHD1 (<11 D4Z4 repeat units on permissive chromosome 4 allele)
- ambulant or walking with assistance
- Manual Muscle Testing ≥4 for 1 of lower limb muscles
Key exclusion criteria (13 listed)
- pregnancy or breast feeding
- stay in tropical/subtropical country within 3 months
- physical exercice within 10 hours
- specific diet (e.g. hypocaloric or cholesterol lowering diet)
- regular alcohol consumption; drug consumption within 3 months
Eligibility is decided by the study team after screening; this overview does not establish that a patient qualifies. Full inclusion and exclusion criteria.
Interventions
| Intervention | Type | Arms |
|---|---|---|
| cytokines dosageAlso: biological analysisMesure of cytokines concentration in serum | Other | Facioscapulohumeral muscular dystrophy |
| test of walkpatient must walk during 6 minutes on a flat surface | Procedure or surgery | Facioscapulohumeral muscular dystrophy |
| Manual muscular testtest performed to evaluate the patient muscular weakness | Procedure or surgery | Facioscapulohumeral muscular dystrophy |
| Motrice fonction mesurementscale allowing the evaluation of patient posture and upper body movements | Procedure or surgery | Facioscapulohumeral muscular dystrophy |
Primary outcome
Comparison of serum IL-6 levels between FSHD1 patients and control subjects (from other previous studies) comparable in terms of age and sex
Time frame: 21 months
Serum IL-6 levels will be measured using v-plex technology (MSD) in FSHD1 patients and compared with results obtained in paired control subjects from 2 cohorts (NCT00998231 and Cytokinage study NCT02660723)
4 secondary outcomes
Comparison of serum levels of 28 oher pro-inflammatory cytokines between FSHD1 patients and control subjects
Time frame: 21 months
Comparison of cytokines produced upon in vitro stimulation of blood cells in FSHD1 patients and control subjects
Time frame: 21 months
Evaluation of potential correlations between cytokines levels (either in the serum or produced upon in vitro stimulation) and clinical severity in FSHD1 patients
Time frame: 21 months
Evaluation of potential correlations between cytokines levels (either in the serum or produced upon in vitro stimulation) and muscle MRI caracteristics in FSHD1 patients
Time frame: 21 months
Results and safety
The study is completed, but results are not available in the source record. That does not mean the treatment did or did not work.
Research links
- Official record on ClinicalTrials.govLast updated by the sponsor 24 Mar 2026.
- Results publication: PubMed 40898356Sanson B, Slioui A, Garcia J, Klouvi L, Lejeune J, Stalens C, Guien C, Rabarimeriarijaona S, Bernard R, Nectoux J, Attarian S, Bedat-Millet AL, Bouhour F, Boyer FC, Chanson JB, Choumert A, Cintas P, De La Cruz E, Feasson L, Fournier M, Ghorab K, Jacquin-Piques A, Laforet P, Magot A, Michaud M, Noury JB, Sole G, Spinazzi M, Stojkovic T, Tard C, Villa L, Beroud C, Sacconi S; French FSHD registry collaboration group. Prevalence and predictors of uncommon features in FSHD1 patients: insights from the French FSHD registry. Orphanet J Rare Dis. 2025 Sep 2;20(1):470. doi: 10.1186/s13023-025-03877-z.
- Derived from this study: PubMed 41058127Pini J, Martinuzzi E, Dhifallah S, Slioui A, Puma A, Villa L, Cavalli M, Ezaru A, Garcia J, Gambella M, Torre F, Pavan LJ, Glaichenhaus N, Sacconi S. Interleukin-6 as a Key Biomarker in Facioscapulohumeral Dystrophy: Evidence From Longitudinal Analyses. Ann Clin Transl Neurol. 2026 Feb;13(2):310-323. doi: 10.1002/acn3.70210. Epub 2025 Oct 7.
- Individual participant dataSharing planned: No
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Referral and contact
AzTrials does not enrol participants or take referrals. Contact the study team or a site listed below; they decide who can take part. This study is not recruiting at the moment.
The record lists no contact. Use the official record to reach the sponsor.
1 site in 1 country: see every site and its status.
Questions about this trial
What is this clinical trial studying?
A study testing cytokines dosage, test of walk and Manual muscular test in people with Muscular Dystrophies, run by Centre Hospitalier Universitaire de Nice at 1 site in France.
The full registered description is in the About this study section and the official record.
Who may be eligible to take part?
The sponsor lists 4 inclusion and 13 exclusion criteria. Eligibility is decided by the study team after screening, so read the full criteria and contact a listed site.
Is this clinical trial currently recruiting?
The record says: finished, as of the sponsor's last update on 24 Mar 2026. Status can change between updates, so confirm with the study contact before making plans.
Where is the study taking place?
The record lists 1 site in France. Whether a site is taking part right now should be confirmed with that site. See every site.
What are the possible benefits and risks?
They depend on the study and the treatment, and no study can promise a benefit. The study team explains the known risks during informed consent, before anyone joins; the record itself does not list them.
Are results available for this study?
The study is completed, but results are not available in the source record. That does not mean the treatment did or did not work.
How can a doctor refer a patient or contact the research team?
The record lists no contact. The official record names the sponsor, who can be asked directly. AzTrials does not enrol participants or take referrals.
What does the primary outcome measure?
Comparison of serum IL-6 levels between FSHD1 patients and control subjects (from other previous studies) comparable in terms of age and sex (time frame: 21 months). It is what the study was designed to measure, not a finding.
Where can I read the original study information?
On the ClinicalTrials.gov record for NCT04694456, which also lists 2 linked publications.
Does a listing mean the treatment is approved or proven to work?
No. A listing means the study is registered. It does not show that the treatment is safe, works, or is approved by a regulator.
How often is this information updated?
AzTrials copies changed records from ClinicalTrials.gov every night and reloads the whole registry every week. The sponsor last updated this record on 24 Mar 2026.