Physical activity of children and adolescents with Charcot-Marie-Tooth neuropathies: A cross-sectional case-controlled study.
Journal
PloS one
ISSN: 1932-6203
Titre abrégé: PLoS One
Pays: United States
ID NLM: 101285081
Informations de publication
Date de publication:
2019
2019
Historique:
received:
06
12
2018
accepted:
20
05
2019
entrez:
13
6
2019
pubmed:
13
6
2019
medline:
6
2
2020
Statut:
epublish
Résumé
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.
Sections du résumé
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.
Identifiants
pubmed: 31188833
doi: 10.1371/journal.pone.0209628
pii: PONE-D-18-34992
pmc: PMC6561632
doi:
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
e0209628Déclaration de conflit d'intérêts
The authors have declared that no competing interests exist.
Références
J Biomed Inform. 2009 Apr;42(2):377-81
pubmed: 18929686
Ann Neurol. 2012 May;71(5):642-52
pubmed: 22522479
Muscle Nerve. 2009 Feb;39(2):158-66
pubmed: 19145658
Arch Dis Child. 2019 Jun;104(6):535-540
pubmed: 30104392
Res Dev Disabil. 2016 Feb-Mar;49-50:268-76
pubmed: 26741263
Dev Med Child Neurol. 2015 Feb;57(2):137-48
pubmed: 25403649
Clin Biomech (Bristol, Avon). 2006 Jan;21(1):89-98
pubmed: 16182419
J Child Neurol. 2010 Apr;25(4):463-8
pubmed: 19671887
Arch Pediatr Adolesc Med. 2011 Jan;165(1):68-76
pubmed: 21199983
Arch Phys Med Rehabil. 2004 Aug;85(8):1217-23
pubmed: 15295743
Biomed Environ Sci. 2016 Mar;29(3):177-86
pubmed: 27109128
Spinal Cord. 2008 Apr;46(4):246-54
pubmed: 17923844
Lancet Child Adolesc Health. 2017 Oct;1(2):106-113
pubmed: 30169201
Res Dev Disabil. 2015 Dec;47:165-74
pubmed: 26431806
Health Qual Life Outcomes. 2014 Aug 30;12:132
pubmed: 25176595
J Sch Health. 2016 Sep;86(9):677-85
pubmed: 27492937
Gait Posture. 2018 May;62:262-267
pubmed: 29579702
BMC Public Health. 2014 May 16;14:461
pubmed: 24886625
Arch Public Health. 2014 Dec 24;72(1):47
pubmed: 25671114
Ann Neurol. 2017 Sep;82(3):353-359
pubmed: 28796392
J Neurol. 2006 Dec;253(12):1594-602
pubmed: 16924398
BMC Pediatr. 2016 Jan 19;16:9
pubmed: 26786677
Arch Phys Med Rehabil. 2015 Feb;96(2):260-8
pubmed: 25286435
Physiotherapy. 2013 Mar;99(1):63-70
pubmed: 23219645
Med Sci Sports Exerc. 1997 Oct;29(10):1344-9
pubmed: 9346166
Pediatr Phys Ther. 2013 Winter;25(4):415-20
pubmed: 23995671
Gait Posture. 2001 May;13(3):193-201
pubmed: 11323225
J Sci Med Sport. 2009 Sep;12(5):518-25
pubmed: 19038579
Disabil Rehabil. 2017 Aug;39(17):1753-1758
pubmed: 27684376
Med Sci Sports Exerc. 2013 May;45(5):997-1003
pubmed: 23190596
Sports Med. 2001;31(6):439-54
pubmed: 11394563
JAMA Neurol. 2016 Jun 1;73(6):645-51
pubmed: 27043305
Med Sci Sports Exerc. 2008 Apr;40(4):767-72
pubmed: 18317366
Arch Phys Med Rehabil. 2014 Jan;95(1):117-128.e11
pubmed: 23820298
J Neurol Neurosurg Psychiatry. 2006 Aug;77(8):977-9
pubmed: 16574732