Arthrogryposis Multiplex Congenita in Pediatric Age: Correlation Between MUScular MRI and Functional Evaluation

NCT ID: NCT05393375

Last Updated: 2023-01-04

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

97 participants

Study Classification

OBSERVATIONAL

Study Start Date

2022-05-01

Study Completion Date

2022-11-01

Brief Summary

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The aim of our study is to evaluate the correlation between quantified fibro-adipous infiltration of muscles on MRI assessed by MERCURI Score and the functional abilities (deficiencies, activity limitations and social participation of pediatric patients with arthrogryposis.

Detailed Description

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Arthrogryposis multiplex congenita (AMC) is a group of rare diseases characterized by joint contractures at two or more distinct joint levels at birth. More than 400 causes are currently known. The prevalence is estimated at 1/3000 to 1/2000. Joint limitations are not progressive, but the functional consequences have a lifelong impact on patients.

Correlations between fibro-adipous infiltration and muscular deficits have already been studied in other neuromuscular diseases such as Pompe disease or Duchenne muscular dystrophy. These studies concluded on a good correlation between disease progression and severity, muscle strength, and fatty infiltration assessed by Mercuri score. The investigators concluded that muscular MRI can be used as a non-invasive biomarker and a tool to follow the progression of neuromuscular disease and help evaluate the potential effects of therapies.

The aim of this project is to define whether MRI could also be used as a tool, through the assessment of muscle fatty infiltration, to help predict the functional abilities of our patients with AMC, and more specifically the two most frequent types, Amyoplasia and distal arthrogryposis.

The investigators will analyze fatty infiltration measured by muscle MRI and quantify it using the MERCURI score which was developed by Mercuri et al. (Mercuri et al. 2002) as a semi-quantitative score to quantify fibro-adipose muscle infiltration on muscle MRI.

The investigators will evaluate musculoskeletal deficiencies and limitations of activity from data obtained from patients followed in our AMC reference center at the University Hospital Grenoble Alpes (CHUGA).

The investigators will include data from 220 children, 97 of whom have had a complete check-up including a muscle MRI. The investigators will study the existence of a correlation between this score and the deficiencies and limitations of activity.

The investigators will then evaluate the potential predictive role of fatty infiltration measured on MRI in the functional abilities of the patients.

This characterization will help to better determine and anticipate the functional capacities of our patients from the beginning of their treatment to improve their functional prognosis.

This initial work could enhance the reflection on the biomechanical modeling of joints and key functions such as standing and the acquisition of walking, in order to pave the way for rehabilitation and compensation methods that are better adapted to patient's needs.

Conditions

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Arthrogryposis Multiplex Congenita AMC-Arthrogryposis Multiplex Congenita Pediatric ALL

Study Design

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Observational Model Type

COHORT

Study Time Perspective

RETROSPECTIVE

Study Groups

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Group 1 Amyoplasia

Patients with diagnosis of Amyoplasia

No intervention

Intervention Type OTHER

No intervention

Group 2 Distal Arthrogryposis

Patients with diagnosis of Distal Arthrogryposis

No intervention

Intervention Type OTHER

No intervention

Group 3 Other

Patients with diagnosis of other form of AMC

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

* age \< 18 at the first clinical evaluation or follow up exclusively in CHUGA pediatric rehabilitation unit
* Clinical or molecular diagnosis of AMC established by geneticists at CHUGA
* functional evaluation during consultation and follow-up in CHUGA pediatric rehabilitation unit
* Muscle MRI of upper limbs, lower limbs and trunk performed at CHUGA pediatric radiology unit

Exclusion Criteria

* Contra indication MRI.
* MRI non exploitable.
* Patient under guardianship or deprived of liberty
Minimum Eligible Age

0 Years

Maximum Eligible Age

18 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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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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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)

Quijano-Roy S, Avila-Smirnow D, Carlier RY; WB-MRI muscle study group. Whole body muscle MRI protocol: pattern recognition in early onset NM disorders. Neuromuscul Disord. 2012 Oct 1;22 Suppl 2:S68-84. doi: 10.1016/j.nmd.2012.08.003.

Reference Type RESULT
PMID: 22980770 (View on PubMed)

Dieterich K, Le Tanno P, Kimber E, Jouk PS, Hall J, Giampietro P. The diagnostic workup in a patient with AMC: Overview of the clinical evaluation and paraclinical analyses with review of the literature. Am J Med Genet C Semin Med Genet. 2019 Sep;181(3):337-344. doi: 10.1002/ajmg.c.31730. Epub 2019 Aug 1.

Reference Type RESULT
PMID: 31368648 (View on PubMed)

Other Identifiers

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2022-AMUSE

Identifier Type: -

Identifier Source: org_study_id

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