Quantitative muscle MRI as an assessment tool for monitoring disease progression in LGMD2I: a multicentre longitudinal study

Tracey A Willis; Kieren G Hollingsworth; Anna Coombs; Marie-Louise Sveen; Søren Peter Andersen; Tanya Stojkovic; Michelle Eagle; Anna Mayhew; Paulo L de Sousa; Liz Dewar; Jasper M Morrow; Christopher D J Sinclair; John S Thornton; Kate Bushby; Hanns Lochmüller; Michael G Hanna; Jean-Yves Hogrel; Pierre G Carlier; John Vissing; Volker Straub

doi:10.1371/journal.pone.0070993

Quantitative muscle MRI as an assessment tool for monitoring disease progression in LGMD2I: a multicentre longitudinal study

Tracey A Willis, Kieren G Hollingsworth, Anna Coombs, Marie-Louise Sveen, Søren Peter Andersen, Tanya Stojkovic, Michelle Eagle, Anna Mayhew, Paulo L de Sousa, Liz Dewar, Jasper M Morrow, Christopher D J Sinclair, John S Thornton, Kate Bushby, Hanns Lochmüller, Michael G Hanna, Jean-Yves Hogrel, Pierre G Carlier, John Vissing, Volker Straub

Department of Neurology

152 Citations (Scopus)

Abstract

Outcome measures for clinical trials in neuromuscular diseases are typically based on physical assessments which are dependent on patient effort, combine the effort of different muscle groups, and may not be sensitive to progression over short trial periods in slow-progressing diseases. We hypothesised that quantitative fat imaging by MRI (Dixon technique) could provide more discriminating quantitative, patient-independent measurements of the progress of muscle fat replacement within individual muscle groups.

Original language	English
Journal	P L o S One
Volume	8
Issue number	8
Pages (from-to)	e70993
ISSN	1932-6203
DOIs	https://doi.org/10.1371/journal.pone.0070993
Publication status	Published - 2013

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Willis, T. A., Hollingsworth, K. G., Coombs, A., Sveen, M.-L., Andersen, S. P., Stojkovic, T., Eagle, M., Mayhew, A., de Sousa, P. L., Dewar, L., Morrow, J. M., Sinclair, C. D. J., Thornton, J. S., Bushby, K., Lochmüller, H., Hanna, M. G., Hogrel, J.-Y., Carlier, P. G., Vissing, J., & Straub, V. (2013). Quantitative muscle MRI as an assessment tool for monitoring disease progression in LGMD2I: a multicentre longitudinal study. P L o S One, 8(8), e70993. https://doi.org/10.1371/journal.pone.0070993

Willis, TA, Hollingsworth, KG, Coombs, A, Sveen, M-L, Andersen, SP, Stojkovic, T, Eagle, M, Mayhew, A, de Sousa, PL, Dewar, L, Morrow, JM, Sinclair, CDJ, Thornton, JS, Bushby, K, Lochmüller, H, Hanna, MG, Hogrel, J-Y, Carlier, PG, Vissing, J & Straub, V 2013, 'Quantitative muscle MRI as an assessment tool for monitoring disease progression in LGMD2I: a multicentre longitudinal study', P L o S One, vol. 8, no. 8, pp. e70993. https://doi.org/10.1371/journal.pone.0070993

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abstract = "Outcome measures for clinical trials in neuromuscular diseases are typically based on physical assessments which are dependent on patient effort, combine the effort of different muscle groups, and may not be sensitive to progression over short trial periods in slow-progressing diseases. We hypothesised that quantitative fat imaging by MRI (Dixon technique) could provide more discriminating quantitative, patient-independent measurements of the progress of muscle fat replacement within individual muscle groups.",

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AU - Willis, Tracey A

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AU - Coombs, Anna

AU - Sveen, Marie-Louise

AU - Andersen, Søren Peter

AU - Stojkovic, Tanya

AU - Eagle, Michelle

AU - Mayhew, Anna

AU - de Sousa, Paulo L

AU - Dewar, Liz

AU - Morrow, Jasper M

AU - Sinclair, Christopher D J

AU - Thornton, John S

AU - Bushby, Kate

AU - Lochmüller, Hanns

AU - Hanna, Michael G

AU - Hogrel, Jean-Yves

AU - Carlier, Pierre G

AU - Vissing, John

AU - Straub, Volker

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AB - Outcome measures for clinical trials in neuromuscular diseases are typically based on physical assessments which are dependent on patient effort, combine the effort of different muscle groups, and may not be sensitive to progression over short trial periods in slow-progressing diseases. We hypothesised that quantitative fat imaging by MRI (Dixon technique) could provide more discriminating quantitative, patient-independent measurements of the progress of muscle fat replacement within individual muscle groups.

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DO - 10.1371/journal.pone.0070993

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IS - 8

ER -

Quantitative muscle MRI as an assessment tool for monitoring disease progression in LGMD2I: a multicentre longitudinal study

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