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Building the repertoire of measures of walking in Rett syndrome

Stahlhut, Michelle LU ; Downs, Jenny; Leonard, Helen; Bisgaard, Anne Marie and Nordmark, Eva LU (2016) In Disability and Rehabilitation p.1-6
Abstract

Background: The repertoire of measures of walking in Rett syndrome is limited. This study aimed to determine measurement properties of a modified two-minute walk test (2MWT) and a modified Rett syndrome-specific functional mobility scale (FMS-RS) in Rett syndrome. Methods: Forty-two girls and women with Rett syndrome (median 18.4 years, range 2.4–60.9 years) were assessed for clinical severity, gross motor skills, and mobility. To measure walking capacity, 27 of this group completed a 2MWT twice on two different assessment days. To assess walking performance, the FMS-RS was administered to the total sample of parents (n = 42) on two occasions approximately one week apart. Results: There were negative correlations between clinical... (More)

Background: The repertoire of measures of walking in Rett syndrome is limited. This study aimed to determine measurement properties of a modified two-minute walk test (2MWT) and a modified Rett syndrome-specific functional mobility scale (FMS-RS) in Rett syndrome. Methods: Forty-two girls and women with Rett syndrome (median 18.4 years, range 2.4–60.9 years) were assessed for clinical severity, gross motor skills, and mobility. To measure walking capacity, 27 of this group completed a 2MWT twice on two different assessment days. To assess walking performance, the FMS-RS was administered to the total sample of parents (n = 42) on two occasions approximately one week apart. Results: There were negative correlations between clinical severity and 2MWT (r = −0.48) and FMS-RS (r = −0.60–0.66). There were positive correlations between gross motor skills and mobility and 2MWT (r = 0.51, 0.43) and FMS-RS (r = 0.71–0.93, 0.74–0.94), respectively. Test–retest reliability for the 2MWT was good with high intraday and interday correlations (ICC = 0.86–0.98). For the 2MWT, the standard error of measurement was 13.8 m and we would be 95% confident that changes greater than 38 m would be greater than within subject error. There was good test–retest reliability for all three distances on the FMS-RS (ICC = 0.94–0.99). Conclusions: Walking capacity as measured by the 2MWT showed expected but limited relationships with measures of different constructs, providing some support for concurrent validity. Walking performance as measured with the FMS-RS was more strongly consistent with other clinical measures supporting its concurrent validity. Test–retest reliability was good for both the FMS-RS and the 2MWT. Therefore, these measures have the potential to be used in clinical practice and research. Implications for RehabilitationWalking is one of the commonest daily physical activities in ambulant girls and women with RTT. Comprehensive knowledge about the walking abilities in this population is limited.Evidence of validity and test–retest reliability have been demonstrated for the modified two-minute walk test (2MWT) and the Rett syndrome-specific functional mobility scale (FMS-RS).The 2MWT and FMS-RS offer detailed information of the capacity and performance of walking, respectively, in girls and women with RTT

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author
organization
publishing date
type
Contribution to journal
publication status
epub
subject
keywords
functional mobility scale, Rett syndrome, two-minute walk test, walking
in
Disability and Rehabilitation
pages
6 pages
publisher
Taylor & Francis
external identifiers
  • Scopus:84983372026
ISSN
0963-8288
DOI
10.1080/09638288.2016.1212280
language
English
LU publication?
yes
id
4ab5393c-0537-4856-bd61-4edf7677f404
date added to LUP
2016-09-21 22:55:38
date last changed
2017-01-17 12:47:56
@article{4ab5393c-0537-4856-bd61-4edf7677f404,
  abstract     = {<p>Background: The repertoire of measures of walking in Rett syndrome is limited. This study aimed to determine measurement properties of a modified two-minute walk test (2MWT) and a modified Rett syndrome-specific functional mobility scale (FMS-RS) in Rett syndrome. Methods: Forty-two girls and women with Rett syndrome (median 18.4 years, range 2.4–60.9 years) were assessed for clinical severity, gross motor skills, and mobility. To measure walking capacity, 27 of this group completed a 2MWT twice on two different assessment days. To assess walking performance, the FMS-RS was administered to the total sample of parents (n = 42) on two occasions approximately one week apart. Results: There were negative correlations between clinical severity and 2MWT (r = −0.48) and FMS-RS (r = −0.60–0.66). There were positive correlations between gross motor skills and mobility and 2MWT (r = 0.51, 0.43) and FMS-RS (r = 0.71–0.93, 0.74–0.94), respectively. Test–retest reliability for the 2MWT was good with high intraday and interday correlations (ICC = 0.86–0.98). For the 2MWT, the standard error of measurement was 13.8 m and we would be 95% confident that changes greater than 38 m would be greater than within subject error. There was good test–retest reliability for all three distances on the FMS-RS (ICC = 0.94–0.99). Conclusions: Walking capacity as measured by the 2MWT showed expected but limited relationships with measures of different constructs, providing some support for concurrent validity. Walking performance as measured with the FMS-RS was more strongly consistent with other clinical measures supporting its concurrent validity. Test–retest reliability was good for both the FMS-RS and the 2MWT. Therefore, these measures have the potential to be used in clinical practice and research. Implications for RehabilitationWalking is one of the commonest daily physical activities in ambulant girls and women with RTT. Comprehensive knowledge about the walking abilities in this population is limited.Evidence of validity and test–retest reliability have been demonstrated for the modified two-minute walk test (2MWT) and the Rett syndrome-specific functional mobility scale (FMS-RS).The 2MWT and FMS-RS offer detailed information of the capacity and performance of walking, respectively, in girls and women with RTT</p>},
  author       = {Stahlhut, Michelle and Downs, Jenny and Leonard, Helen and Bisgaard, Anne Marie and Nordmark, Eva},
  issn         = {0963-8288},
  keyword      = {functional mobility scale,Rett syndrome,two-minute walk test,walking},
  language     = {eng},
  month        = {08},
  pages        = {1--6},
  publisher    = {Taylor & Francis},
  series       = {Disability and Rehabilitation},
  title        = {Building the repertoire of measures of walking in Rett syndrome},
  url          = {http://dx.doi.org/10.1080/09638288.2016.1212280},
  year         = {2016},
}