Pane, M;
Coratti, G;
Brogna, C;
Mazzone, ES;
Mayhew, A;
Fanelli, L;
Messina, S;
... Mercuri, E; + view all
(2018)
Upper limb function in Duchenne muscular dystrophy: 24 month longitudinal data.
PLoS One
, 13
(6)
, Article e0199223. 10.1371/journal.pone.0199223.
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Abstract
The aim of the study was to establish 24 month changes in upper limb function using a revised version of the performance of upper limb test (PUL 2.0) in a large cohort of ambulant and non-ambulant boys with Duchenne muscular dystrophy and to identify possible trajectories of progression. Of the 187 patients studied, 87 were ambulant (age range: 7–15.8 years), and 90 non-ambulant (age range: 9.08–24.78). The total scores changed significantly over time (p<0.001). Non-ambulant patients had lower total scores at baseline (mean 19.7) when compared to the ambulant ones (mean 38.4). They also had also a bigger decrease in total scores over 24 months compared to the ambulant boys (4.36 vs 2.07 points). Multivariate model analysis showed that the Performance of Upper Limb changes reflected the entry level and ambulation status, that were independently associated to the slope of Performance of Upper Limb changes. This information will be of help both in clinical practice and at the time of designing clinical trials.
| Type: | Article |
|---|---|
| Title: | Upper limb function in Duchenne muscular dystrophy: 24 month longitudinal data |
| Open access status: | An open access version is available from UCL Discovery |
| DOI: | 10.1371/journal.pone.0199223 |
| Publisher version: | https://doi.org/10.1371/journal.pone.0199223 |
| Language: | English |
| Additional information: | © 2018 Pane et al. This is an open access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/). |
| Keywords: | Walking, Duchenne muscular dystrophy, Shoulders, Patient advocacy, Steroids, Clinical trials, Data acquisition, Psychometrics |
| UCL classification: | UCL UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health > Developmental Neurosciences Dept |
| URI: | https://discovery.ucl.ac.uk/id/eprint/10066592 |
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