Karoliny Lisandra Teixeira Cruz1, Ana Cristina Resende Camargos2, Juliana Cardoso3, Cyntia Rogean de Jesus Alves de Baptista3, Aline Duprat Ramos4, Ana Claudia Mattiello-Sverzut3, Joshua Burns5, Hércules Ribeiro Leite6. 1. Programa de Pós Graduação em Reabilitação e Desempenho Funcional (PPGReab), Universidade Federal dos Vales do Jequitinhonha e Mucuri (UFVJM), Campus JK, Alto da Jacuba, MG, Brazil. 2. Programa de Pós Graduação em Reabilitação e Desempenho Funcional (PPGReab), Universidade Federal dos Vales do Jequitinhonha e Mucuri (UFVJM), Campus JK, Alto da Jacuba, MG, Brazil; Programa de Pós Graduação em Ciências da Reabilitação (PPGCr), Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, MG, Brazil. 3. Department of Health Sciences, Ribeirão Preto Medical School, University of Sao Paulo, Sao Paulo, SP, Brazil. 4. Hospital Foundation of the State of Minas Gerais - FHEMIG - João XXIII Hospital, Physical Therapy Department, Belo Horizonte, MG, Brazil. 5. University of Sydney School of Health Sciences, Faculty of Medicine and Health & Children's Hospital at Westmead, New South Wales, Sydney, Australia. 6. Programa de Pós Graduação em Reabilitação e Desempenho Funcional (PPGReab), Universidade Federal dos Vales do Jequitinhonha e Mucuri (UFVJM), Campus JK, Alto da Jacuba, MG, Brazil; Programa de Pós Graduação em Ciências da Reabilitação (PPGCr), Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, MG, Brazil. Electronic address: hercules@ufmg.br.
Abstract
BACKGROUND: The Charcot-Marie-Tooth disease Pediatric Scale (CMTPedS) has been used to measure aspects of disability in children with all types of Charcot-Marie-Tooth disease (CMT). OBJECTIVE: To translate and cross-culturally adapt the CMTPedS into Brazilian-Portuguese and determine its reliability and validity. METHODS: The translation and cross-cultural adaptation followed international guidelines recommendations. Twenty individuals with CMT were assessed. Two examiners assessed the participants for inter-rater reliability. Face validity was assessed by eight physical therapists that judged the relevance of each test item. The Bland-Altman analysis (bias) and standard error of measurement (SEM) complemented the analysis. Furthermore, intraclass correlation coefficients (ICC), weighted kappa (k), and internal consistency (Cronbach's alpha) was determined. RESULTS: The CMTPedS was successfully translated and cross-culturally adapted. Twenty children/youth were enrolled in the study. Of these, the majority (55%) were girls with a mean age of 13.9 (range: from 6 to 18) years. Regarding face validity, the CMTPedS-Br showed relevant items for assessing children and youth with CMT. The ICC for the total score showed excellent reliability (ICC2.1 = 0.93, 95% CI = 0.84, 0.97). The most reliable items were grip, dorsiflexion and plantar flexion strength while the least reliable items were pinprick, vibration, and gait. The internal consistency was excellent (α = 0.96, 95% CI = 0.91, 0.99) and the agreement showed small variability (bias = 0.15, 95% CI= -4.28, 4.60). CONCLUSION: The CMTPedS-Br showed adequate reliability and face validity to measure disability in individuals with CMT. This tool will allow Brazil to be part of multicentered studies on such a rare but debilitating condition.
BACKGROUND: The Charcot-Marie-Tooth disease Pediatric Scale (CMTPedS) has been used to measure aspects of disability in children with all types of Charcot-Marie-Tooth disease (CMT). OBJECTIVE: To translate and cross-culturally adapt the CMTPedS into Brazilian-Portuguese and determine its reliability and validity. METHODS: The translation and cross-cultural adaptation followed international guidelines recommendations. Twenty individuals with CMT were assessed. Two examiners assessed the participants for inter-rater reliability. Face validity was assessed by eight physical therapists that judged the relevance of each test item. The Bland-Altman analysis (bias) and standard error of measurement (SEM) complemented the analysis. Furthermore, intraclass correlation coefficients (ICC), weighted kappa (k), and internal consistency (Cronbach's alpha) was determined. RESULTS: The CMTPedS was successfully translated and cross-culturally adapted. Twenty children/youth were enrolled in the study. Of these, the majority (55%) were girls with a mean age of 13.9 (range: from 6 to 18) years. Regarding face validity, the CMTPedS-Br showed relevant items for assessing children and youth with CMT. The ICC for the total score showed excellent reliability (ICC2.1 = 0.93, 95% CI = 0.84, 0.97). The most reliable items were grip, dorsiflexion and plantar flexion strength while the least reliable items were pinprick, vibration, and gait. The internal consistency was excellent (α = 0.96, 95% CI = 0.91, 0.99) and the agreement showed small variability (bias = 0.15, 95% CI= -4.28, 4.60). CONCLUSION: The CMTPedS-Br showed adequate reliability and face validity to measure disability in individuals with CMT. This tool will allow Brazil to be part of multicentered studies on such a rare but debilitating condition.
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