The Rett Syndrome Gross Motor Scale - Dutch Version (RSGMS-NL) Can Reliably Assess Gross Motor Skills in Dutch Individuals with Rett Syndrome

被引:2
作者
Borst, Hanneke [1 ,2 ,3 ]
Weeda, Josianne [4 ]
Downs, Jennyn [5 ,6 ]
Curfs, Leopold [7 ]
de Bie, Rob [8 ]
机构
[1] Maastricht Univ, Med Ctr, Rett Expertise Ctr, Maastricht, Netherlands
[2] Maastricht Univ, Sch Mental Hlth & Neurosci, Maastricht, Netherlands
[3] Gemiva SVG Groep, Gouda, Netherlands
[4] Maastricht Univ, Dept Epidemiol, Maastricht, Netherlands
[5] Telethon Kids Inst, Nedlands, WA, Australia
[6] Curtin Univ, Sch Physiotherapy & Exercise Sci, Bentley, WA, Australia
[7] Maastricht Univ, Rett Expertise Ctr, Maastricht, Netherlands
[8] Maastricht Univ, Care & Publ Hlth Res Inst, Dept Epidemiol, Maastricht, Netherlands
关键词
Rett syndrome; motor skills; validation studies; reproducibility of results; RELIABILITY;
D O I
10.1080/17518423.2021.1960920
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Aim The Rett Syndrome Gross Motor Scale (RSGMS) is an observational measurement, assessing gross motor skills in individuals with Rett syndrome. A Dutch version is lacking. The current study aims to translate and cross-culturally adapt the original RSMGS to Dutch and assess its inter-rater and intra-rater reliability. Design Translation and cross-cultural adaptation were performed in concordance with internationally accepted guidelines. A pretest was performed, and expert validation was assured. Video data of three girls with Rett syndrome was independently assessed by 27 physiotherapists via an online webinar to measure inter-rater reliability. Additionally, videos of 17 individuals with Rett syndrome were scored twice by two raters to assess intra-rater reliability. The reliability of the total score, the three subscale scores and the new items were analyzed using Intraclass Correlation Coefficients (ICC). Results Good comprehensibility and expert validation of the RSMGS-NL was achieved, and four items were added to the original scale. Inter-rater reliability for the total score was excellent (ICC 0.97, 95% CI 0.89-0.99), and good to perfect inter-rater reliability for the three subscales; Sitting, Standing & walking and Challenge was found (ICC values 1.0 (95% CI 0), 0.98 (95% CI 0.91-0.99) and 0.82 (95% CI 0.93-0.99) respectively). The intra-rater reliability was excellent for the total test score (ICC 0.98, 95% CI 0.97-0.99) and good to excellent for the subscale scores (ICC values 0.87 (95% CI 0.75-0.94), 0.99 (95% CI 0.98-1.0) and 0.97 (95% CI 0.95-0.99) respectively). The four new items (Standing to sitting, walking down a slope, ascending the stairs, descending the stairs) showed good to excellent intra-rater reliability. Conclusion The RSGMS-NL is a reliable measure of gross motor skills in Dutch individuals with Rett syndrome.
引用
收藏
页码:133 / 139
页数:7
相关论文
共 21 条
  • [1] Rett syndrome is caused by mutations in X-linked MECP2, encoding methyl-CpG-binding protein 2
    Amir, RE
    Van den Veyver, IB
    Wan, M
    Tran, CQ
    Francke, U
    Zoghbi, HY
    [J]. NATURE GENETICS, 1999, 23 (02) : 185 - 188
  • [2] [Anonymous], 2020, WAT IS RETT SYNDROOM
  • [3] Guidelines for the process of cross-cultural adaptation of self-report measures
    Beaton, DE
    Bombardier, C
    Guillemin, F
    Ferraz, MB
    [J]. SPINE, 2000, 25 (24) : 3186 - 3191
  • [4] Investigating genotype-phenotype relationships in Rett syndrome using an international data set
    Bebbington, A.
    Anderson, A.
    Ravine, D.
    Fyfe, S.
    Pineda, M.
    de Klerk, N.
    Ben-Zeev, B.
    Yatawara, N.
    Percy, A.
    Kaufmann, W. E.
    Leonard, H.
    [J]. NEUROLOGY, 2008, 70 (11) : 868 - 875
  • [5] Neurophysiological Signatures of Motor Impairment in Patients with Rett Syndrome
    Bernardo, Pia
    Cobb, Stuart
    Coppola, Antonietta
    Tomasevic, Leo
    Di Lazzaro, Vincenzo
    Bravaccio, Carmela
    Manganelli, Fiore
    Dubbioso, Raffaele
    [J]. ANNALS OF NEUROLOGY, 2020, 87 (05) : 763 - 773
  • [6] The incidence of Rett syndrome in France
    Bienvenu, Thierry
    Philippe, Christophe
    De Roux, Nicolas
    Raynaud, Martine
    Bonnefond, Jean Paul
    Pasquier, Laurent
    Lesca, Gaetan
    Mancini, Josette
    Jonveaux, Philippe
    Moncla, Anne
    Feingold, Josue
    Chelly, Jamel
    Villard, Laurent
    [J]. PEDIATRIC NEUROLOGY, 2006, 34 (05) : 372 - 375
  • [7] Gross Motor Profile in Rett Syndrome as Determined by Video Analysis
    Downs, J. A.
    Bebbington, A.
    Jacoby, P.
    Msall, M. E.
    McIlroy, O.
    Fyfe, S.
    Bahi-Buisson, N.
    Kaufmann, W. E.
    Leonard, H.
    [J]. NEUROPEDIATRICS, 2008, 39 (04) : 205 - 210
  • [8] The Natural History of Scoliosis in Females With Rett Syndrome
    Downs, Jennepher
    Torode, Ian
    Wong, Kingsley
    Ellaway, Carolyn
    Elliott, Elizabeth J.
    Christodoulou, John
    Jacoby, Peter
    Thomson, Margaret R.
    Izatt, Maree T.
    Askin, Geoffrey N.
    McPhee, Bruce I.
    Bridge, Corinne
    Cundy, Peter
    Leonard, Helen
    [J]. SPINE, 2016, 41 (10) : 856 - 863
  • [9] Environmental enrichment intervention for Rett syndrome: an individually randomised stepped wedge trial
    Downs, Jenny
    Rodger, Jenny
    Li, Chen
    Tan, Xuesong
    Hu, Nan
    Wong, Kingsley
    de Klerk, Nicholas
    Leonard, Helen
    [J]. ORPHANET JOURNAL OF RARE DISEASES, 2018, 13
  • [10] Validating the Rett Syndrome Gross Motor Scale
    Downs, Jenny
    Stahlhut, Michelle
    Wong, Kingsley
    Syhler, Birgit
    Bisgaard, Anne-Marie
    Jacoby, Peter
    Leonard, Helen
    [J]. PLOS ONE, 2016, 11 (01):