title: Handwriting difficulties of children with Charcot-Marie-Tooth disease type 1A.
abstract: Hand weakness and impaired manual dexterity have been reported in children with Charcot-Marie-Tooth disease type 1A (CMT1A). This early onset of upper limb involvement might explain frequent clinical referrals for assessment and treatment of impaired handwriting performance. The aim of this study was to examine the impact of CMT1A on handwriting speed and legibility, and identify demographic, anthropometric, and physical measures that might relate to handwriting performance. Handwriting speed (Handwriting Speed Test), handwriting legibility (Evaluation Tool of Children's Handwriting-Cursive), and hand strength (hand-held dynamometry of tip pinch, lateral pinch and grip) were assessed in 30 children with CMT1A (aged 8-17 years) and 30 age- and sex-matched controls. Children with CMT1A exhibited 34% slower handwriting speed (p < 0.0001) with 4% reduced legibility (p = 0.001) and 37-48% lower hand strength (p < 0.0001). All measures of strength, age, height, and weight were positively associated with handwriting speed (r = 0.39-0.79, p < 0.01). None of these factors related to handwriting legibility (p > 0.05). Regression modelling identified a diagnosis of CMT1A, lateral pinch weakness and younger age as significant independent predictors of slower handwriting speed, explaining 78% of the variance. Children with CMT1A have considerable handwriting difficulties, primarily with speed, and substantial associated hand and finger weakness. Understanding the cause-effect relationship between strength and function might provide modifiable targets for upper limb intervention.
authors.affiliation: Faculty of Health Sciences, University of Sydney, Sydney, NSW, Australia.
authors.forename: Daniel
authors.initials: D
authors.lastname: Kunovsky
authors.affiliation: Faculty of Health Sciences, Curtin University, Perth, WA, Australia.
authors.forename: Reinie
authors.initials: R
authors.lastname: Cordier
authors.affiliation: Faculty of Health Sciences, University of Sydney, Sydney, NSW, Australia.|Sydney Children's Hospitals Network (Randwick and Westmead), Sydney, NSW, Australia.
authors.forename: Paula
authors.initials: P
authors.lastname: Bray
authors.affiliation: Faculty of Health Sciences, University of Sydney, Sydney, NSW, Australia.|Sydney Children's Hospitals Network (Randwick and Westmead), Sydney, NSW, Australia.
authors.forename: Joshua
authors.initials: J
authors.lastname: Burns
mesh_terms: D000293:Adolescent; D016022:Case-Control Studies; D002607:Charcot-Marie-Tooth Disease / Q000150:complications* / Q000534:rehabilitation; D002648:Child; D005260:Female; D018737:Hand Strength / Q000502:physiology*; D006236:Handwriting; D006801:Humans; D007806:Language Disorders / Q000209:etiology*; D008297:Male; D009788:Occupational Therapy; D011596:Psychomotor Disorders / Q000209:etiology*
keywords: handwriting; musculoskeletal; neuropathy; paediatric; upper extremity