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DOI: 10.1055/s-0043-1772832
Brazilian version of the CHOP INTEND scale: cross-cultural adaptation and validation
Versão brasileira da escala CHOP INTEND: adaptação transcultural e validaçãoAbstract
Background Spinal muscular atrophy (SMA) is a rare genetic disease that causes progressive muscle weakness and impacts motor function. The type I is the most severe presentation and affects infants before 6 months old. In addition, the instruments available for assessing motor function have limitations when applied to infants with neuromuscular diseases and significant muscle weakness.
Objective To translate, cross-culturally adapt, and validate the Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND) to Brazilian Portuguese.
Methods The present study comprised the translation, synthesis of translations, backtranslation, consolidation by a committee of experts, and test of the final version of the CHOP INTEND in 13 patients with SMA type I. We also assessed the content validity and reliability of the translated version.
Results The scale was translated considering semantic, structural, idiomatic, and cultural aspects. All agreement rates were > 0.8, the overall content validity index of the instrument was 0.98, and inter-rater reliability using the intraclass correlation coefficient was 0.998.
Conclusion The Brazilian version of the CHOP INTEND met semantic and technical equivalence criteria with the original version and was valid and reliable for patients with SMA type I.
Resumo
Antecedentes A atrofia muscular espinhal (AME) é uma doença genética rara que provoca fraqueza muscular progressiva com impacto sobre a motricidade dos pacientes. A AME tipo I é considerada o tipo mais grave e acomete lactentes antes dos 6 meses de idade. As escalas disponíveis para avaliação das aquisições motoras mostram limitações para uso com crianças pequenas com doenças neuromusculares e fraqueza importante.
Objetivo Realizar a tradução, adaptação transcultural e validação para a língua portuguesa do Brasil da Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND, na sigla em inglês).
Métodos O presente estudo seguiu as etapas de tradução, síntese das traduções, retrotradução, consolidação por comitê de especialistas e teste com 13 pacientes com AME tipo 1. Foi avaliada a validade de conteúdo e a confiabilidade do instrumento.
Resultados A escala foi traduzida considerando os aspectos semânticos, estruturais, idiomáticos e culturais. Todas as taxas de concordância foram > 0,8. O índice de validade de conteúdo geral do instrumento foi de 0,98. A confiabilidade interavaliadores analisada através do coeficiente de correlação intraclasse (ICC, na sigla em inglês) demonstrou um valor de ICC = 0,998.
Conclusão A versão da CHOP INTEND em português atende aos critérios de equivalência semântica e técnica em relação à versão original e apresenta validade de conteúdo e confiabilidade para seu uso na população de pacientes com AME tipo I.
Authors' Contributions
RMRA: conceptualization, methodology, project administration, data collection, formal analysis, writing-original draft, writing-review and editing; APMC, VVDL, MAFCB: data acquisition, drafting of manuscript, critical review; LBA: conceptualization, methodology, formal analysis, writing-original draft, writing-review and editing. All authors discussed the results and contributed to the final manuscript.
Publikationsverlauf
Eingereicht: 03. Dezember 2022
Angenommen: 01. Mai 2023
Artikel online veröffentlicht:
04. Oktober 2023
© 2023. Academia Brasileira de Neurologia. This is an open access article published by Thieme under the terms of the Creative Commons Attribution 4.0 International License, permitting copying and reproduction so long as the original work is given appropriate credit (https://creativecommons.org/licenses/by/4.0/)
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References
- 1 Mercuri E. Spinal muscular atrophy: from rags to riches. Neuromuscul Disord 2021; 31 (10) 998-1003
- 2 Farrar MA, Park SB, Vucic S. et al. Emerging therapies and challenges in spinal muscular atrophy. Ann Neurol 2017; 81 (03) 355-368
- 3 Mercuri E, Lucibello S, Perulli M. et al. Longitudinal natural history of type I spinal muscular atrophy: a critical review. Orphanet J Rare Dis 2020; 15 (01) 84
- 4 Farrar MA, Kiernan MC. The genetics of spinal muscular atrophy: progress and challenges. Neurotherapeutics 2015; 12 (02) 290-302
- 5 Schorling DC, Pechmann A, Kirschner J. Advances in treatment of spinal muscular atrophy - new phenotypes, new challenges, new implications for care. J Neuromuscul Dis 2020; 7 (01) 1-13
- 6 Pane M, Coratti G, Sansone VA. et al; Italian EAP Working Group. Type I SMA “new natural history”: long-term data in nusinersen-treated patients. Ann Clin Transl Neurol 2021; 8 (03) 548-557
- 7 Darras BT, Masson R, Mazurkiewicz-Bełdzińska M. et al; FIREFISH Working Group. Risdiplam-treated infants with Type 1 spinal muscular atrophy versus historical controls. N Engl J Med 2021; 385 (05) 427-435
- 8 Mercuri E, Finkel RS, Muntoni F. et al; SMA Care Group. Diagnosis and management of spinal muscular atrophy: Part 1: Recommendations for diagnosis, rehabilitation, orthopedic and nutritional care. Neuromuscul Disord 2018; 28 (02) 103-115
- 9 Finkel RS, Mercuri E, Meyer OH. et al; SMA Care group. Diagnosis and management of spinal muscular atrophy: Part 2: Pulmonary and acute care; medications, supplements and immunizations; other organ systems; and ethics. Neuromuscul Disord 2018; 28 (03) 197-207
- 10 Pierzchlewicz K, Kępa I, Podogrodzki J, Kotulska K. Spinal muscular atrophy: The use of functional motor scales in the era of disease-modifying treatment. Child Neurol Open 2021;8:2329048X211008725
- 11 Bach JR, Saltstein K, Sinquee D, Weaver B, Komaroff E. Long-term survival in Werdnig-Hoffmann disease. Am J Phys Med Rehabil 2007; 86 (05) 339-345 , quiz 346–348, 379
- 12 Bach JR, Gupta K, Reyna M, Hon A. Spinal muscular atrophy type 1: Prolongation of survival by noninvasive respiratory aids. Pediatr Asthma Allergy Immunol 2009; 22 (04) 151-161
- 13 Glanzman AM, Mazzone E, Main M. et al. The Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND): test development and reliability. Neuromuscul Disord 2010; 20 (03) 155-161
- 14 Glanzman AM, McDermott MP, Montes J. et al; Pediatric Neuromuscular Clinical Research Network for Spinal Muscular Atrophy (PNCR), Muscle Study Group (MSG). Validation of the Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND). Pediatr Phys Ther 2011; 23 (04) 322-326
- 15 Finkel R, Bertini E, Muntoni F, Mercuri E. 209th ENMC International Workshop: Outcome measures and clinical trial readiness in spinal muscular atrophy 7–9 November 2014, Heemskerk, The Netherlands. Neuromuscul Dis 2015; 25 (07) 593-602
- 16 Glanzman AM, Mazzone ES, Young SD. et al. Evaluator Training and Reliability for SMA Global Nusinersen Trials1. J Neuromuscul Dis 2018; 5 (02) 159-166
- 17 Iwabe C, Miranda-Pfeilsticker BH, Nucci A. Medida da função motora: versão da escala para o português e estudo de confiabilidade. Rev Bras Fisioter. 2008; 12 (05) 417-424
- 18 Corre MT. Tradução e adaptação cultural dos instrumentos: Hammersmith Neonatal Neurological Assessment (HNNE) e Hammersmith Infant Neurological Assessment (HINE) e validação do instrumento HNNE para lactentes brasileiros com risco de paralisia cerebral [Thesis]. São Paulo: Universidade de Medicina de Ribeirão Preto; 2020
- 19 Reichenheim ME, Moraes CL. [Operationalizing the cross-cultural adaptation of epidemiological measurement instruments]. Rev Saude Publica 2007; 41 (04) 665-673
- 20 Fortes CPDD, Araújo APQC. Check list for healthcare questionnaires cross-cultural translation and adaptation. Cad Saude Colet 2019; 27 (02) 202-209
- 21 Mokkink LB, Terwee CB, Patrick DL. et al. The COSMIN study reached international consensus on taxonomy, terminology, and definitions of measurement properties for health-related patient-reported outcomes. J Clin Epidemiol 2010; 63 (07) 737-745
- 22 Souza AC, Alexandre NMC, Guirardello EB. Psychometric properties in instruments evaluation of reliability and validity. Epidemiol Serv Saude 2017; 26 (03) 649-659
- 23 Borsa JC, Damásio BF, Bandeira DR. Cross-cultural adaptation and validation of psychological instruments: Some considerations. Paideia. 2012; Sep; 22 (53) 423-432
- 24 Alexandre NMC, Coluci MZO. [Content validity in the development and adaptation processes of measurement instruments]. Cien Saude Colet 2011; 16 (07) 3061-3068
- 25 Alexandre NMC, Gallasch CH, Lima MHM, Rodrigues RCM. Reliability in the development and evaluation of measurement instruments in the health field. Rev Eletr Enf 2013; 15 (03) 802-809
- 26 Koo TK, Li MY. A guideline of selecting and reporting intraclass correlation coefficients for reliability research. J Chiropr Med 2016; 15 (02) 155-163
- 27 Hjartarson HT, Nathorst-Böös K, Sejersen T. Disease modifying therapies for the management of children with Spinal Muscular Atrophy (5q SMA): An update on the emerging evidence. Drug Des Devel Ther 2022; 16: 1865-1883
- 28 Russman BS, Iannaccone ST, Samaha FJ. A phase 1 trial of riluzole in spinal muscular atrophy. Arch Neurol 2003; 60 (11) 1601-1603
- 29 Blaschek A, Hesse N, Warken B. et al. Quantitative motion measurements based on markerless 3D full-body tracking in children with SMA highly correlate with standardized motor assessments. J Neuromuscul Dis 2022; 9 (01) 121-128
- 30 Edel L, Grime C, Robinson V. et al. A new respiratory scoring system for evaluation of respiratory outcomes in children with spinal muscular atrophy type1 (SMA1) on SMN enhancing drugs. Neuromuscul Disord 2021; 31 (04) 300-309
- 31 Bach JR, Gonçalves MR, Hamdani I, Winck JC. Extubation of patients with neuromuscular weakness: a new management paradigm. Chest 2010; 137 (05) 1033-1039
- 32 Fauroux B, Khirani S. Neuromuscular disease and respiratory physiology in children: putting lung function into perspective. Respirology 2014; 19 (06) 782-791