Mercuri Analysis Contribution on Handicap Evaluation in ArthrogypOsis, a Congenital Neuromuscular Disease

NCT ID: NCT05137756

Last Updated: 2021-11-30

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

53 participants

Study Classification

OBSERVATIONAL

Study Start Date

2019-12-01

Study Completion Date

2021-10-01

Brief Summary

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The aim is to evaluate the correlation of quantified fibro-adipous infiltration of muscles, using the MRI-based Mercuri score, with deficiencies, activity limitations and social participation in patients with arthrogryposis multiplex congenita.

Detailed Description

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Arthrogryposis multiplex congenita (AMC) refers to a rare disease spectrum characterized by the presence of joint contractures at birth in at least two different body areas. Causes are multiple. Amyoplasia and distal arthrogryposes are the most frequent causes. Phenotypes consist of muscle weakness and atrophy of variant severity and joint deformities. The impact on mobility, activities or daily living and participation is variable (Dai et al, 2018).

Mercuri et al. (Mercuri et al, 2002) have developed a semi-quantitative score to mesure fibro-adipous muscle infiltration on muscle MRI.

Correlations between fibro-adipous infiltration and muscular deficits have already been studied in other neuromusclar diseases such as Pompe disease (Figueroa-Bonaparte et al, 2016) or Duchenne muscular dystrophy (Brogan et al, 2018). These studies revealed good correlation between disease onset, muscle strength and the degree of muscle fibro-adipous infiltration, calculated using the Mercuri score. The authors concluded that muscular MRI can be used to follow the neuromuscular disease progression as it is correlated with muscle function.

Contrary to these progressive diseases, AMC is a congenital non progressive condition. It is therefore tempting to hypothesize that the muscular fibro-adipous infiltration in these patients could be of prognostic value for future capacities.

Our aim is to evaluate the fibro-adipous muscle infiltration in a thoroughly phenotyped cohort of adult patients with Amyoplasia and Distal Arthrogryposes using the Mercuri score, based on muscular MRI, in order to evaluate the correlation with deficiencies, activity limitations and social participation.

Conditions

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Diagnosis of Arthrogryposis Amyoplasia or Distal Arthrogryposis 5 Day Multidisciplinary Evaluation in AMC Clinic of the National Reference Center With Physical Medecin, Medical Genetic and Imaging Departments at the Hospital Grenoble Alpes

Keywords

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Arthrogryposis Multiplex Congenita MRI mercuri score fibro-adipous infiltration fonctionnal capacity rehabilitation retrospective observationnal study

Study Design

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Observational Model Type

COHORT

Study Time Perspective

RETROSPECTIVE

Study Groups

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Amyoplasia

patient with diagnosis of Amyoplasia

no intervention

Intervention Type OTHER

no intervention

Distal arthrogryposis

patient with diagnosis of Distal arthrogryposis

no intervention

Intervention Type OTHER

no intervention

Interventions

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no intervention

no intervention

Intervention Type OTHER

Eligibility Criteria

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Inclusion Criteria

* adults
* with a diagnostic of Amyoplasia or distal arthrogryposis
* evaluated by a all body MRI
* functionnal evaluation during day hospitalisation in neurorehabilitation department
* between 2010 and october 2020

Exclusion Criteria

* other aetiology of Arthrogryposis multiplex congenita (AMC)
* incomplete MRI or medical files
* interaction with other disease
Minimum Eligible Age

17 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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University Hospital, Grenoble

OTHER

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Dominic PERENNOU, MDPHD

Role: STUDY_DIRECTOR

CHU Grenoble Alpes

Klaus DIETERICH, MD

Role: STUDY_DIRECTOR

CHU Grenoble Alpes

Locations

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Chu Grenoble Alpes

La Tronche, , France

Site Status

Countries

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France

References

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Liu CY, Yao J, Kovacs WC, Shrader JA, Joe G, Ouwerkerk R, Mankodi AK, Gahl WA, Summers RM, Carrillo N. Skeletal Muscle Magnetic Resonance Biomarkers in GNE Myopathy. Neurology. 2021 Feb 2;96(5):e798-e808. doi: 10.1212/WNL.0000000000011231. Epub 2020 Nov 20.

Reference Type BACKGROUND
PMID: 33219145 (View on PubMed)

Hall JG. Arthrogryposis multiplex congenita: etiology, genetics, classification, diagnostic approach, and general aspects. J Pediatr Orthop B. 1997 Jul;6(3):159-66.

Reference Type RESULT
PMID: 9260643 (View on PubMed)

Mercuri E, Pichiecchio A, Counsell S, Allsop J, Cini C, Jungbluth H, Uggetti C, Bydder G. A short protocol for muscle MRI in children with muscular dystrophies. Eur J Paediatr Neurol. 2002;6(6):305-7. doi: 10.1016/s1090-3798(02)90617-3.

Reference Type RESULT
PMID: 12401454 (View on PubMed)

Figueroa-Bonaparte S, Segovia S, Llauger J, Belmonte I, Pedrosa I, Alejaldre A, Mayos M, Suarez-Cuartin G, Gallardo E, Illa I, Diaz-Manera J; Spanish Pompe Study Group. Muscle MRI Findings in Childhood/Adult Onset Pompe Disease Correlate with Muscle Function. PLoS One. 2016 Oct 6;11(10):e0163493. doi: 10.1371/journal.pone.0163493. eCollection 2016.

Reference Type RESULT
PMID: 27711114 (View on PubMed)

Dai S, Dieterich K, Jaeger M, Wuyam B, Jouk PS, Perennou D. Disability in adults with arthrogryposis is severe, partly invisible, and varies by genotype. Neurology. 2018 May 1;90(18):e1596-e1604. doi: 10.1212/WNL.0000000000005418. Epub 2018 Apr 6.

Reference Type RESULT
PMID: 29626181 (View on PubMed)

Other Identifiers

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2021-MACHAON

Identifier Type: -

Identifier Source: org_study_id