Functional outcome measures in young, steroid-naïve boys with Duchenne muscular dystrophy.

Mayhew, Anna G, Moat, Dionne, McDermott, Michael P, Eagle, Michelle, Griggs, Robert C, King, Wendy M, James, Meredith K, Muni-Lofra, Robert, Shillington, Alison, Gregson, Sarah, Pallant, Lindsey, Skura, Christy, Staudt, Loretta A, Eichinger, Katy, McMurchie, Heather, Rabb, Rosanna, Di Marco, Marina, Brown, Sarah, Zanin, Riccardo, Arnoldi, Maria Teresa, McIntyre, Melissa, Wilson, Amelia, Alfano, Lindsay N, Lowes, Linda P, Blomgren, Colleen, Milev, Evelin, Iodice, Mario, Pasternak, Amy, Chiu, Angela, Lehnert, Ilka, Claus, Nicole, Dieruf, Kathy A, Rolle, Enrica, Nicorici, Alina, Andres, Barbara, Hobbiebrunken, Elke, Roetmann, Gerda, Kern, Victoria, Civitello, Matthew, Vogt, Sibylle, Hayes, Melissa J, Scholtes, Cheryl, Lacroix, Catherine, Gunn, Tara, Warner, Sinead, Newman, Jennifer, Barp, Andrea, Kundrat, Katherine, Kovelman, Staci, Powers, Penny J and Guglieri, Michela (2022) Functional outcome measures in young, steroid-naïve boys with Duchenne muscular dystrophy. Neuromuscular disorders : NMD. ISSN 1873-2364. This article is available to all UHB staff and students login using UHB Athens Account. Register for Athens here - https://openathens.nice.org.uk/

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Official URL: https://www.nmd-journal.com/article/S0960-8966(22)...

Abstract

The purpose of this study was to quantitate motor performance in 196 genetically confirmed steroid-naïve boys with Duchenne muscular dystrophy (DMD), to evaluate the test-retest reliability of measures of motor performance in young DMD boys, and to assess correlations among the different functional outcomes including timed tests. Boys aged 4-7 years were recruited in the FOR-DMD study, a comparative effectiveness study of different steroid regimens in DMD. Eligible boys had to be able to rise from the floor independently and to perform pulmonary function testing consistently. The boys were evaluated with standardized assessments at the screening and baseline visits at 32 sites in 5 countries (US, UK, Canada, Italy, Germany). Assessments included timed rise from floor, timed 10 m walk/run, six-minute walk distance, North Star Ambulatory Assessment (NSAA) and forced vital capacity (FVC). Mean age at baseline was 5.9 years (range 4.1-8.1 years). Test-retest reliability was high for functional assessments, regardless of time lag between assessments (up to 90 days) and for the majority of age groups. Correlations were strong among the functional measures and timed tests, less so with FVC. Physiotherapy measures are reliable in a young, steroid-naïve population and rise from floor velocity appears to be a sensitive measure of strength in this population.

Item Type: Article
Additional Information: This article is available to all UHB staff and students login using UHB Athens Account. Register for Athens here - https://openathens.nice.org.uk/
Subjects: WS Paediatrics. Child health
Divisions (November 2021 Onwards): Paediatrics
Physiotherapy
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Depositing User: Ms Willow Baylis
Date Deposited: 14 Jun 2022 15:11
Last Modified: 14 Jun 2022 15:11
URI: http://www.repository.uhblibrary.co.uk/id/eprint/5646

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