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

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 ph...

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Published in:PloS one 2019-06, Vol.14 (6), p.e0209628-e0209628
Main Authors: Kennedy, Rachel A, Carroll, Kate, Paterson, Kade L, Ryan, Monique M, Burns, Joshua, Rose, Kristy, McGinley, Jennifer L
Format: Article
Language:English
Subjects:
Active control
Adolescent
Adolescents
Age
Archives & records
Biology and Life Sciences
Care and treatment
Case-Control Studies
Charcot-Marie-Tooth disease
Charcot-Marie-Tooth Disease - physiopathology
Child
Children
Cross-Sectional Studies
Disability
Exercise
Gait
Gait Disorders, Neurologic - etiology
Hospitals
Humans
Informatics
Male
Medicine
Medicine and health sciences
Neurodegenerative Diseases
Neurology
Neuropathy
Pediatrics
People and Places
Physical activity
Physical education
Physical fitness
Quality of life
Risk factors
Studies
Surveys and Questionnaires
Systematic review
Teenagers
Walk Test
Walking
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cited_by cdi_FETCH-LOGICAL-c692t-e2b05b6de07bc297a6cf67dd65ffd881d4643442edebb20977bcd40cea85131d3
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creator Kennedy, Rachel A
Carroll, Kate
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Ryan, Monique M
Burns, Joshua
Rose, Kristy
McGinley, Jennifer L
description 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. 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. 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). 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.
doi_str_mv 10.1371/journal.pone.0209628
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The relationship between CMT-related disability, ambulatory function and physical activity in children and adolescents with CMT is unknown. 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. 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 &lt; 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 &lt; 0.001]. Physical activity correlated with greater disability (ρ = -0.56, p &lt; 0.001) and normalised six-minute walk distance (ρ = 0.74, p &lt; 0.001). 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.</abstract><cop>United States</cop><pub>Public Library of Science</pub><pmid>31188833</pmid><doi>10.1371/journal.pone.0209628</doi><tpages>e0209628</tpages><orcidid>https://orcid.org/0000-0003-1805-6937</orcidid><oa>free_for_read</oa></addata></record>
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identifier ISSN: 1932-6203
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issn 1932-6203
1932-6203
language eng
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source NCBI_PubMed Central（免费）; Publicly Available Content Database
subjects Active control
Adolescent
Adolescents
Age
Archives & records
Biology and Life Sciences
Care and treatment
Case-Control Studies
Charcot-Marie-Tooth disease
Charcot-Marie-Tooth Disease - physiopathology
Child
Children
Cross-Sectional Studies
Disability
Exercise
Gait
Gait Disorders, Neurologic - etiology
Hospitals
Humans
Informatics
Male
Medicine
Medicine and health sciences
Neurodegenerative Diseases
Neurology
Neuropathy
Pediatrics
People and Places
Physical activity
Physical education
Physical fitness
Quality of life
Risk factors
Studies
Surveys and Questionnaires
Systematic review
Teenagers
Walk Test
Walking
title Physical activity of children and adolescents with Charcot-Marie-Tooth neuropathies: A cross-sectional case-controlled study
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