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Upper limb function in Duchenne muscular dystrophy: 24 month longitudinal data.


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.

SUBMITTER: Pane M 

PROVIDER: S-EPMC6010252 | biostudies-literature | 2018

REPOSITORIES: biostudies-literature

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Upper limb function in Duchenne muscular dystrophy: 24 month longitudinal data.

Pane Marika M   Coratti Giorgia G   Brogna Claudia C   Mazzone Elena Stacy ES   Mayhew Anna A   Fanelli Lavinia L   Messina Sonia S   D'Amico Adele A   Catteruccia Michela M   Scutifero Marianna M   Frosini Silvia S   Lanzillotta Valentina V   Colia Giulia G   Cavallaro Filippo F   Rolle Enrica E   De Sanctis Roberto R   Forcina Nicola N   Petillo Roberta R   Barp Andrea A   Gardani Alice A   Pini Antonella A   Monaco Giulia G   D'Angelo Maria Grazia MG   Zanin Riccardo R   Vita Gian Luca GL   Bruno Claudio C   Mongini Tiziana T   Ricci Federica F   Pegoraro Elena E   Bello Luca L   Berardinelli Angela A   Battini Roberta R   Sansone Valeria V   Albamonte Emilio E   Baranello Giovanni G   Bertini Enrico E   Politano Luisa L   Sormani Maria Pia MP   Mercuri Eugenio E  

PloS one 20180620 6


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 t  ...[more]

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