title: Measuring Ankle Instability in Pediatric Charcot-Marie-Tooth Disease.
abstract: Children with Charcot-Marie-Tooth disease frequently suffer ankle sprain and experience chronic ankle instability; however, no pediatric self-reported measures of chronic ankle instability exist. The aim was to modify and validate the most reliable measure of chronic ankle instability in adults: the Cumberland Ankle Instability Tool. The Cumberland Ankle Instability Tool-Youth was tested for reliability, construct validity, and sensitivity to discriminate between 104 children aged 8 to 16 years: 31 children with Charcot-Marie-Tooth disease, 31 unaffected children with a history of ankle sprains, and 42 controls. Children with Charcot-Marie-Tooth disease had lower scores compared to unaffected children with a history of sprains (χ2= 15.10; P < .001) and controls (χ2= 33.69; P < .001). Scores moderately correlated to visual analog scale scores of ankle steadiness (rs= 0.684; P < .001), and "good" test-retest reliability was identified (ICC2,1= 0.73). The Cumberland Ankle Instability Tool-Youth demonstrated excellent sensitivity and construct validity, identifying chronic ankle instability as a common problem for children with Charcot-Marie-Tooth disease.
authors.affiliation: 1Arthritis and Musculoskeletal Research Group, Faculty of Health Sciences, The University of Sydney, NSW, Australia.
authors.forename: Melissa
authors.initials: M
authors.lastname: Mandarakas
authors.forename: Claire E
authors.initials: CE
authors.lastname: Hiller
authors.forename: Kristy J
authors.initials: KJ
authors.lastname: Rose
authors.forename: Joshua
authors.initials: J
authors.lastname: Burns
keywords: Charcot-Marie-Tooth disease; ankle instability; outcome research; peripheral neuropathy