CC BY 4.0 · Arq Neuropsiquiatr 2024; 82(07): s00441788587
DOI: 10.1055/s-0044-1788587
Original Article

Brazilian version of the Hammersmith Functional Motor Scale Expanded: cross-cultural adaptation and validation

Versão brasileira da Hammersmith Functional Motor Scale Expanded: adaptação transcultural e validação
1   Universidade Federal de Minas Gerais, Departamento de Pediatria, Belo Horizonte MG, Brazil.
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1   Universidade Federal de Minas Gerais, Departamento de Pediatria, Belo Horizonte MG, Brazil.
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1   Universidade Federal de Minas Gerais, Departamento de Pediatria, Belo Horizonte MG, Brazil.
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1   Universidade Federal de Minas Gerais, Departamento de Pediatria, Belo Horizonte MG, Brazil.
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2   Columbia University Irving Medical Center, Department of Rehabilitation and Regenerative Medicine, New York, United States.
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1   Universidade Federal de Minas Gerais, Departamento de Pediatria, Belo Horizonte MG, Brazil.
› Institutsangaben

Abstract

Background The Hammersmith Functional Motor Scale Expanded (HFMSE) has been widely used to assess the motor function of patients with spinal muscular atrophy (SMA) older than 2 years, with the ability to sit and/or walk.

Objective To translate, cross-culturally adapt and validate the HFMSE to Brazilian Portuguese.

Methods The translation process and cross-cultural adaptation followed international guidelines recommendations. The reliability and applicability of the Brazilian version consisted of the application of the HFMSE (in Brazilian Portuguese) to 20 patients with types 2 and 3 SMA. Two examiners assessed the participants for interrater reliability, through the analysis of Kappa reliability agreement (k) and intraclass correlation coefficient (ICC).

Results The HFMSE was successfully translated and cross culturally adapted to Brazilian Portuguese. Twenty participants with types 2 and 3 SMA were enrolled in the study (type 2 = 6; type 3 = 14). The ICC for the total score showed very high reliability (ICC =1.00), and the reliability of each of the items individually was considered excellent (Kappa > 0.80).

Conclusion The Brazilian version of the HFMSE proved to be valid and reliable for the evaluation of SMA patients older than 2 years with the ability to sit and/or walk.

Resumo

Antecedentes A Hammersmith Functional Motor Scale Expanded (HFMSE) tem sido amplamente utilizada para avaliar a função motora de pacientes com atrofia muscular espinhal (AME) maiores de dois anos, com capacidade de sentar e/ou andar.

Objetivo Traduzir, adaptar transculturalmente e validar a HFMSE para o português brasileiro.

Métodos A tradução e a adaptação transcultural seguiram as diretrizes internacionais. A confiabilidade e a aplicabilidade da versão brasileira consistiram na aplicação da HFMSE (em português brasileiro) em 20 pacientes com AME tipos 2 e 3. Dois examinadores avaliaram os participantes quanto à confiabilidade interexaminadores, por meio da análise da concordância de confiabilidade Kappa (k) e do coeficiente de correlação intraclasse (intraclass correlation coefficient [ICC]).

Resultados O processo de tradução e adaptação transcultural da HFMSE para o português brasileiro foi concluído com sucesso. Vinte participantes com AME tipos 2 e 3 foram incluídos no estudo (tipo 2 = 6; tipo 3 = 14). O ICC para o escore total apresentou confiabilidade alta (ICC = 1.00) e a confiabilidade de cada um dos itens individualmente foi considerada excelente (K > 0,80).

Conclusão A HFMSE (PT-BR) mostrou-se válida e confiável para a avaliação de pacientes com AME, com mais de dois anos de idade e com capacidade de sentar-se independentemente e/ou andar.

Authors' Contributions

ACMLM: conceptualization, methodology, project administration, data collection, formal analysis, writing-original draft, writing-review and editing. MBC, GPCS, CCP: methodology, data collection, critical review. JM: critical review and approval of the final version. JGG: conceptualization, methodology, formal analysis, drafting of manuscript, critical review. All authors discussed the results and contributed to the final manuscript.




Publikationsverlauf

Eingereicht: 25. Februar 2024

Angenommen: 06. April 2024

Artikel online veröffentlicht:
29. Juli 2024

© 2024. The Author(s). 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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Bibliographical Record
Ana Carolina Monteiro Lessa de Moura, Marina Belisário Carvalhais, Gabriela Palhares Campolina Sampaio, Clara Catharino Pinhati, Jacqueline Montes, Juliana Gurgel-Giannetti. Brazilian version of the Hammersmith Functional Motor Scale Expanded: cross-cultural adaptation and validation. Arq Neuropsiquiatr 2024; 82: s00441788587.
DOI: 10.1055/s-0044-1788587
 
  • References

  • 1 Farrar MA, Park SB, Vucic S. et al. Emerging therapies and challenges in spinal muscular atrophy. Ann Neurol 2017; 81 (03) 355-368
  • 2 Lefebvre S, Bürglen L, Reboullet S. et al. Identification and characterization of a spinal muscular atrophy-determining gene. Cell 1995; 80 (01) 155-165
  • 3 Mercuri E, Sumner CJ, Muntoni F, Darras BT, Finkel RS. Spinal muscular atrophy. Nat Rev Dis Primers 2022; 8 (01) 52
  • 4 Annoussamy M, Seferian AM, Daron A. et al; NatHis-SMA study group. Natural history of Type 2 and 3 spinal muscular atrophy: 2-year NatHis-SMA study. Ann Clin Transl Neurol 2021; 8 (02) 359-373
  • 5 Kaufmann P, McDermott MP, Darras BT. et al; Muscle Study Group (MSG), Pediatric Neuromuscular Clinical Research Network for Spinal Muscular Atrophy (PNCR). Prospective cohort study of spinal muscular atrophy types 2 and 3. Neurology 2012; 79 (18) 1889-1897
  • 6 Tizzano EF, Finkel RS. Spinal muscular atrophy: A changing phenotype beyond the clinical trials. Neuromuscul Disord 2017; 27 (10) 883-889
  • 7 Bérard C, Fermanian J, Payan C. Outcome measure for SMA II and III patients. Neuromuscul Disord 2008; 18 (07) 593-594 , author reply 594–595
  • 8 Vuillerot C. State of the art for motor function assessment tools in spinal muscular atrophy (SMA). Arch Pediatr 2020; 27 (7S): S40-S44
  • 9 Iannaccone ST. American Spinal Muscular Atrophy Randomized Trials (AmSMART) Group. Outcome measures for pediatric spinal muscular atrophy. Arch Neurol 2002; 59 (09) 1445-1450
  • 10 Ramsey D, Scoto M, Mayhew A. et al. Revised Hammersmith Scale for spinal muscular atrophy: A SMA specific clinical outcome assessment tool. PLoS One 2017; 12 (02) e0172346
  • 11 Erdos J, Wild C. Mid- and long-term (at least 12 months) follow-up of patients with spinal muscular atrophy (SMA) treated with nusinersen, onasemnogene abeparvovec, risdiplam or combination therapies: A systematic review of real-world study data. Eur J Paediatr Neurol 2022; 39: 1-10
  • 12 Main M, Kairon H, Mercuri E, Muntoni F. The Hammersmith functional motor scale for children with spinal muscular atrophy: a scale to test ability and monitor progress in children with limited ambulation. Eur J Paediatr Neurol 2003; 7 (04) 155-159
  • 13 O'Hagen JM, Glanzman AM, McDermott MP. et al. An expanded version of the Hammersmith Functional Motor Scale for SMA II and III patients. Neuromuscul Disord 2007; 17 (9-10): 693-697
  • 14 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
  • 15 Pera MC, Coratti G, Forcina N. et al. Content validity and clinical meaningfulness of the HFMSE in spinal muscular atrophy. BMC Neurol 2017; 17 (01) 39
  • 16 Mercuri E, Messina S, Battini R. et al. Reliability of the Hammersmith functional motor scale for spinal muscular atrophy in a multicentric study. Neuromuscul Disord 2006; 16 (02) 93-98
  • 17 Beaton DE, Bombardier C, Guillemin F, Ferraz MB. Guidelines for the process of cross-cultural adaptation of self-report measures. Spine 2000; 25 (24) 3186-3191
  • 18 Fortes CPDD, Araújo APQC. Check list for healthcare questionnaires cross-cultural translation and adaptation. Cad Saude Colet 2019; 27 (02) 202-209 . Doi: 10.1590/1414-462. Doi: × 201900020002
  • 19 Portney LG, Watkins MP. Foundations of clinical research-Applications to Practice. 2015 ;3a edição.
  • 20 Glanzman AM, O'Hagen JM, McDermott MP. et al; Pediatric Neuromuscular Clinical Research Network for Spinal Muscular Atrophy (PNCR), Muscle Study Group (MSG). Validation of the Expanded Hammersmith Functional Motor Scale in spinal muscular atrophy type II and III. J Child Neurol 2011; 26 (12) 1499-1507
  • 21 Johnson R. & Bhattacharyya G. Statistics Principles and Methods. New York:: John Wiley & Sons.; 1986. . 578p.
  • 22 CONOVER. . W. J., Practical Nonparametric Statistics,. New York:: John Wiley & Sons,; 1980. , 493 p.
  • 23 McGraw KO, Wong SP. Forming inferences about some intraclass correlation coefficients. Psychol Methods 1996; 1: 30-46
  • 24 Bloch DA, Kraemer HC. 2 × 2 kappa coefficients: measures of agreement or association. Biometrics 1989; 45 (01) 269-287
  • 25 de Groot IJ, de Witte LP. Physical complaints in ageing persons with spinal muscular atrophy. J Rehabil Med 2005; 37 (04) 258-262
  • 26 Mercuri E, Darras BT, Chiriboga CA. et al; CHERISH Study Group. Nusinersen versus sham control in later-onset spinal muscular atrophy. N Engl J Med 2018; 378 (07) 625-635
  • 27 Mercuri E, Baranello G, Boespflug-Tanguy O. et al; SUNFISH Working Group. Risdiplam in types 2 and 3 spinal muscular atrophy: A randomised, placebo-controlled, dose-finding trial followed by 24 months of treatment. Eur J Neurol 2023; 30 (07) 1945-1956
  • 28 Pechmann A, König K, Bernert G. et al. SMArtCARE - A platform to collect real-life outcome data of patients with spinal muscular atrophy. Orphanet J Rare Dis 2019; 14 (01) 18
  • 29 Alves RMR, Calado APM, Van Der Linden V, Bello MAFC, Andrade LB. Brazilian version of the CHOP INTEND scale: cross-cultural adaptation and validation. Arq Neuropsiquiatr 2023; 81 (09) 816-824
  • 30 Rodriguez-Torres RS, Uher D, Gay EL. et al. Measuring Fatigue and Fatigability in Spinal Muscular Atrophy (SMA): Challenges and Opportunities. J Clin Med 2023; 12 (10) 3458