Muscle ultrasound is a sensitive biomarker in oculopharyngeal muscular dystrophy.

biomarker clinical measures muscle ultrasound oculopharyngeal muscular dystrophy orofacial and limb muscles

Journal

Muscle & nerve
ISSN: 1097-4598
Titre abrégé: Muscle Nerve
Pays: United States
ID NLM: 7803146

Informations de publication

Date de publication:
10 2022
Historique:
revised: 15 07 2022
received: 18 04 2021
accepted: 17 07 2022
pubmed: 22 7 2022
medline: 21 9 2022
entrez: 21 7 2022
Statut: ppublish

Résumé

Oculopharyngeal muscular dystrophy (OPMD) is a late-onset, progressive muscle disease. Quantitative muscle ultrasound (QMUS) assesses structural changes in muscles and is a sensitive biomarker in neuromuscular disorders. Our aim of this study was to determine whether QMUS can detect muscle pathology and can be used as longitudinal imaging biomarker in OPMD. Genetically confirmed OPMD patients, recruited by their treating physicians or from the national neuromuscular database, were examined twice, 20 months apart, using QMUS of orofacial and limb muscles, and measurements of functional capacity and muscle strength. Absolute echo intensity (AEI) and muscle thickness of all muscles were analyzed and correlated with clinical data. The tongue, deltoid, iliopsoas, rectus femoris, and soleus muscles showed increased AEI at baseline compared with normal values in 43 OPMD patients, with the rectus femoris being most often affected (51%).The AEI and muscle thickness of 9 of 11 muscles correlated significantly with the motor function measure, 10-step stair test, swallowing capacity, dynamometry, Medical Research Council grade, tongue strength, and bite force (r = 0.302 to -0.711). Between baseline and follow-up, deterioration in AEI was found for the temporalis, tongue, and deltoid muscles, and decreased muscle thickness was detected for the temporalis, masseter, digastric, tongue, deltoid, iliopsoas, and soleus muscles (P < .05). No relation was found between the change in AEI and repeat length or disease duration. QMUS detected muscle pathology and disease progression in OPMD over 20 months. We conclude that QMUS should be considered as a biomarker in treatment trials.

Identifiants

pubmed: 35859342
doi: 10.1002/mus.27679
doi:

Substances chimiques

Biomarkers 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

453-461

Informations de copyright

© 2022 The Authors. Muscle & Nerve published by Wiley Periodicals LLC.

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Auteurs

Rosemarie H M J M Kroon (RHMJM)

Radboud university medical center, Donders Institute for Brain, Cognition and Behaviour, Department of Rehabilitation, Nijmegen, The Netherlands.

Johanna G Kalf (JG)

Radboud university medical center, Donders Institute for Brain, Cognition and Behaviour, Department of Rehabilitation, Nijmegen, The Netherlands.

Rutger L Meijers (RL)

Radboud university medical center, Donders Institute of Brain, Cognition and Behaviour, Department of Neurology, Nijmegen, the Netherlands.

Bert J M de Swart (BJM)

Radboud university medical center, Donders Institute for Brain, Cognition and Behaviour, Department of Rehabilitation, Nijmegen, The Netherlands.

Ian G M Cameron (IGM)

Radboud University, Donders Institute for Brain, Cognition and Behaviour, Centre for Neuroscience, Nijmegen, The Netherlands.
University of Twente, Biomedical Signals and Systems, Faculty of Electrical Engineering, Mathematics and Computer, Science, Enschede, The Netherlands.

Jonne Doorduin (J)

Radboud university medical center, Donders Institute of Brain, Cognition and Behaviour, Department of Neurology, Nijmegen, the Netherlands.

Nens van Alfen (N)

Radboud university medical center, Donders Institute of Brain, Cognition and Behaviour, Department of Neurology, Nijmegen, the Netherlands.

Baziel G M van Engelen (BGM)

Radboud university medical center, Donders Institute of Brain, Cognition and Behaviour, Department of Neurology, Nijmegen, the Netherlands.

Corinne G C Horlings (CGC)

Radboud university medical center, Donders Institute of Brain, Cognition and Behaviour, Department of Neurology, Nijmegen, the Netherlands.
Medical University of Innsbruck, Department of Neurology, Innsbruck, Austria.

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