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Physical activity of children and adolescents with Charcot-Marie-Tooth neuropathies: A cross-sectional case-controlled study

BACKGROUND: Disability related to the progressive and degenerative neuropathies known collectively as Charcot-Marie-Tooth disease (CMT) affects gait and function, increasing with age and influencing physical activity in adults with CMT. The relationship between CMT-related disability, ambulatory fun...

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Autores principales: Kennedy, Rachel A., Carroll, Kate, Paterson, Kade L., Ryan, Monique M., Burns, Joshua, Rose, Kristy, McGinley, Jennifer L.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Public Library of Science 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6561632/
https://www.ncbi.nlm.nih.gov/pubmed/31188833
http://dx.doi.org/10.1371/journal.pone.0209628
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author Kennedy, Rachel A.
Carroll, Kate
Paterson, Kade L.
Ryan, Monique M.
Burns, Joshua
Rose, Kristy
McGinley, Jennifer L.
author_facet Kennedy, Rachel A.
Carroll, Kate
Paterson, Kade L.
Ryan, Monique M.
Burns, Joshua
Rose, Kristy
McGinley, Jennifer L.
author_sort Kennedy, Rachel A.
collection PubMed
description BACKGROUND: Disability related to the progressive and degenerative neuropathies known collectively as Charcot-Marie-Tooth disease (CMT) affects gait and function, increasing with age and influencing physical activity in adults with CMT. The relationship between CMT-related disability, ambulatory function and physical activity in children and adolescents with CMT is unknown. METHOD: A cross-sectional case-controlled study of physical activity in 50 children with CMT and age- and gender-matched typically developing (TD) controls [mean age 12.5 (SD 3.9) years]. A 7-day recall questionnaire assessed physical activity; CMT-related disability and gait-related function were measured to explore factors associated with physical activity. RESULTS: Children with CMT were less active than TD controls (estimated weekly moderate to vigorous physical activity CMT 283.6 (SD 211.6) minutes, TD 315.8 (SD 204.0) minutes; p < 0.001). The children with CMT had moderate disability [CMT Pediatric Scale mean score 17 (SD 8) /44] and reduced ambulatory capacity in a six-minute walk test [CMT 507.7 (SD 137.3) metres, TD 643.3 (74.6) metres; p < 0.001]. Physical activity correlated with greater disability (ρ = -0.56, p < 0.001) and normalised six-minute walk distance (ρ = 0.74, p < 0.001). CONCLUSIONS: CMT-related disability affects physical activity and gait-related function in children and adolescents with CMT compared to TD peers. Reduced physical activity adversely affects function across the timespan of childhood and adolescence into adulthood in people with CMT.
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spelling pubmed-65616322019-06-20 Physical activity of children and adolescents with Charcot-Marie-Tooth neuropathies: A cross-sectional case-controlled study Kennedy, Rachel A. Carroll, Kate Paterson, Kade L. Ryan, Monique M. Burns, Joshua Rose, Kristy McGinley, Jennifer L. PLoS One Research Article BACKGROUND: Disability related to the progressive and degenerative neuropathies known collectively as Charcot-Marie-Tooth disease (CMT) affects gait and function, increasing with age and influencing physical activity in adults with CMT. The relationship between CMT-related disability, ambulatory function and physical activity in children and adolescents with CMT is unknown. METHOD: A cross-sectional case-controlled study of physical activity in 50 children with CMT and age- and gender-matched typically developing (TD) controls [mean age 12.5 (SD 3.9) years]. A 7-day recall questionnaire assessed physical activity; CMT-related disability and gait-related function were measured to explore factors associated with physical activity. RESULTS: Children with CMT were less active than TD controls (estimated weekly moderate to vigorous physical activity CMT 283.6 (SD 211.6) minutes, TD 315.8 (SD 204.0) minutes; p < 0.001). The children with CMT had moderate disability [CMT Pediatric Scale mean score 17 (SD 8) /44] and reduced ambulatory capacity in a six-minute walk test [CMT 507.7 (SD 137.3) metres, TD 643.3 (74.6) metres; p < 0.001]. Physical activity correlated with greater disability (ρ = -0.56, p < 0.001) and normalised six-minute walk distance (ρ = 0.74, p < 0.001). CONCLUSIONS: CMT-related disability affects physical activity and gait-related function in children and adolescents with CMT compared to TD peers. Reduced physical activity adversely affects function across the timespan of childhood and adolescence into adulthood in people with CMT. Public Library of Science 2019-06-12 /pmc/articles/PMC6561632/ /pubmed/31188833 http://dx.doi.org/10.1371/journal.pone.0209628 Text en © 2019 Kennedy et al http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
spellingShingle Research Article
Kennedy, Rachel A.
Carroll, Kate
Paterson, Kade L.
Ryan, Monique M.
Burns, Joshua
Rose, Kristy
McGinley, Jennifer L.
Physical activity of children and adolescents with Charcot-Marie-Tooth neuropathies: A cross-sectional case-controlled study
title Physical activity of children and adolescents with Charcot-Marie-Tooth neuropathies: A cross-sectional case-controlled study
title_full Physical activity of children and adolescents with Charcot-Marie-Tooth neuropathies: A cross-sectional case-controlled study
title_fullStr Physical activity of children and adolescents with Charcot-Marie-Tooth neuropathies: A cross-sectional case-controlled study
title_full_unstemmed Physical activity of children and adolescents with Charcot-Marie-Tooth neuropathies: A cross-sectional case-controlled study
title_short Physical activity of children and adolescents with Charcot-Marie-Tooth neuropathies: A cross-sectional case-controlled study
title_sort physical activity of children and adolescents with charcot-marie-tooth neuropathies: a cross-sectional case-controlled study
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6561632/
https://www.ncbi.nlm.nih.gov/pubmed/31188833
http://dx.doi.org/10.1371/journal.pone.0209628
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