| 1. |
- Stahlhut, Michelle, et al.
(författare)
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Building the repertoire of measures of walking in Rett syndrome
- 2017
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Ingår i: Disability and Rehabilitation. - : Informa UK Limited. - 0963-8288 .- 1464-5165. ; 39:19, s. 1926-1931
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Tidskriftsartikel (refereegranskat)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 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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| 2. |
- Stahlhut, Michelle, et al.
(författare)
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Discriminative Validity of the Danish Version of the Pediatric Evaluation of Disability Inventory (PEDI).
- 2011
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Ingår i: Physical and Occupational Therapy in Pediatrics. - : Informa UK Limited. - 1541-3144 .- 0194-2638. ; 31, s. 78-89
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Tidskriftsartikel (refereegranskat)abstract
- ABSTRACT The Pediatric Evaluation of Disability Inventory (PEDI) assesses functional status in children with disabilities aged 0.5-7.5 years. The purpose of this study was to examine if the Danish version of the PEDI was able to discriminate between nondisabled children and children with cerebral palsy (CP) or juvenile idiopathic arthritis (JIA). Convenience sampling was used to select the clinical samples that consisted of 22 children with CP and 14 children with JIA aged 1.5-7.5 years. An additional sample, consisting of 224 nondisabled children aged 1.0-5.9 years, was obtained on the basis of random sampling. The PEDI was administered as a questionnaire completed by the parents. Results from receiver operating characteristics (ROC) analysis showed that in the case of discrimination between children with CP and nondisabled children, the sensitivity and specificity were 77.3 and 97.8%, respectively. In the discrimination between children with JIA and nondisabled children, the sensitivity and specificity were 99.8 and 81.7%, respectively. Study results indicate that the Danish version of the PEDI shows good discriminative validity. Further studies of the discriminative validity are recommended.
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| 3. |
- Stahlhut, Michelle, et al.
(författare)
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Facilitators and Barriers of Participation in “Uptime” Activities in Girls and Women With Rett Syndrome : Perspectives From Parents and Professionals
- 2019
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Ingår i: Qualitative Health Research. - : SAGE Publications. - 1049-7323 .- 1552-7557. ; 29:4, s. 609-619
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Tidskriftsartikel (refereegranskat)abstract
- Rett syndrome (RTT) is a rare neurodevelopmental disorder usually affecting females. It is associated with intellectual and multiple disabilities leading to a high level of dependency in all aspects of daily living including participation in physical activities. This study explored facilitators and barriers to “uptime” (non-sedentary) activities in Danish girls and women with RTT as perceived by parents and professionals using focus groups. Through thematic analysis, one central theme emerged: a constant balance to do the best thing for the girl or woman. Within the central theme, five subthemes of facilitators and barriers were identified relating to the individual and the physical, organizational, social, and attitudinal environments. Environmental barriers can be reduced through policy and management-level changes in health promotion and strong advocacy of physical activity by health professionals. Targeting both facilitators and barriers of “uptime” activities enables the planning and implementing of health-promoting interventions in individuals with RTT.
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| 4. |
- Stahlhut, Michelle, et al.
(författare)
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Feasibility and Effectiveness of an Individualized 12-Week "Uptime" Participation (U-PART) Intervention in Girls and Women With Rett Syndrome
- 2020
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Ingår i: Physical Therapy. - : Oxford University Press (OUP). - 0031-9023 .- 1538-6724. ; 100:1, s. 168-179
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Tidskriftsartikel (refereegranskat)abstract
- BACKGROUND: Girls and women with Rett Syndrome (RTT) have low levels of daily physical activity and high levels of sedentary time. Reducing sedentary time and enhancing "uptime" activities, such as standing and walking, could be an important focus for interventions to address long-term health and quality of life in RTT. OBJECTIVE: The aim of the study was to evaluate the feasibility and health-related effects of an individualized 12-week uptime participation (U-PART) intervention in girls and women with RTT. DESIGN: The study used a single-group pretest-posttest design with 4 assessments (2 baseline, postintervention, and follow-up). METHODS: A participation-based intervention employing a whole-day approach was used. During a 12-week intervention period, individualized programs focused on participation in enjoyable uptime activities in home, school/day center, and community settings. Feasibility was assessed with a study-specific questionnaire. Primary outcome measures were sedentary time and daily step count. Secondary outcomes were gross motor skills, walking capacity, quality of life, and goal attainment scaling. RESULTS: Fourteen girls and women who were 5 to 48 years old and had RTT participated. The U-PART intervention was perceived as feasible by caregivers. Similar scores were observed at baseline assessments in all outcomes. Positive effects with small to medium effect sizes (0.27-0.54) were seen in sedentary time (- 4%), daily step count (+ 689 steps/d), walking capacity (+ 18.8 m), quality of life (+ 2.75 points), and goal attainment scaling after the intervention. Positive effects were maintained in sedentary time (- 3.2%) and walking capacity (+ 12.1 m) at short-term follow-up. LIMITATIONS: This study was limited by the lack of a control group. However, participants acted as their own control, and the stable baseline period partially mitigated this issue. CONCLUSIONS: The U-PART intervention was found to be feasible and effective in the short term in girls and women with RTT.
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| 5. |
- Stahlhut, Michelle
(författare)
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Health-enhancing participation in girls and women with Rett syndrome - a balancing act
- 2018
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Doktorsavhandling (övrigt vetenskapligt/konstnärligt)abstract
- Rett syndrome (RTT) is a rare neurodevelopmental disorder usually affecting females. It is associated with multiple disabilities including intellectual disability leading to a high level of dependency in all aspects of daily living including participation in physical activities. The overall aim of this thesis was to investigate physical activity behaviors by developing measures of walking, describing patterns of physical behavior and influencing factors, and evaluating the effects of an intervention that focuses on participation in standing and walking activities (i.e. ‘uptime’ activities) in Danish girls and women with RTT. This thesis comprises four studies. In study I, measurement properties of a modified two-minute walk test (2MWT) and a modified RTT-specific functional mobility scale (FMS-RS) were determined. Forty-two girls and women with RTT aged 2.4–60.9 years were included. Comparison measures were the Clinical Severity Score (CSS), Rett Syndrome Gross Motor Scale (RSGMS) and the mobility domain in the Pediatric Evaluation of Disability Inventory (PEDI-m). In study II, patterns of sedentary behavior and physical activity were described in a population-based sample including 48 girls and women with RTT aged 5.5–60.5 years. Participants wore the activPAL and StepWatch Activity Monitor (SAM) for at least four days. In study III, facilitators and barriers to participation in ‘up-time’ were explored from the perspectives of parents and professionals using focus groups. Data was analyzed using thematic analysis. In study IV, the feasibility and effectiveness of an individualized 12-wk ‘uptime’ participation intervention (U-PART) were evaluated in 14 girls and women with RTT aged 5.6-48.3 years. Each individual program focused on participation in enjoyable activities to promote ‘uptime’ in home, school/day center and community settings. Feasibility was assessed using a study-specific questionnaire. Primary outcomes were sedentary time (activPAL) and daily steps (SAM). Secondary outcomes were gross motor skills (RSGMS), walking capacity (2MWT), quality of life (Quality of Life Inventory-Disability, QI-Disability) and participation-level goals (Goal Attainment Scaling, GAS). Outcomes were evaluated on four occasions: at baseline and after a 6-week interval, immediately following the 12-week intervention program and 12 weeks after the intervention program. Results showed low-moderate and moderate-high correlations between comparison measures and the 2MWT and FMS-RS, respectively. Intraclass correlation coefficients (ICC) were high for both the 2MWT (ICC=0.86-0.98) and FMS-RS (ICC=0.94-0.99) test values. In the 2MWT standard error of measurement (SEM) was 13.8m and minimal detectable difference (MDD) was calculated to be 38m (study I). On average 83.3% (SD 13.9%) of waking hours were spent in sedentary behaviors (n=48) and the median (IQR) daily step count was 5128 (2829–7704) (n=28). Advancing age and poorer walking skills were associated with higher levels of sedentary time (study II). Several facilitators and barriers of ‘uptime’ activities were identified within five subthemes relating to the individual girl/woman and her physical, organizational, social and attitudinal environment. The resources within each of the five areas needed to be balanced to enable optimal participation in ‘uptime’ activities (study III). Stakeholders perceived the U-PART intervention as feasible and significant positive effects were seen after the intervention in sedentary time (-4.1%), daily steps (+708 steps), 2MWT (+18.9m), QI-Disability (+2.8) and for individually determined goals. At follow-up, effects on sedentary time (-3.4%) and 2MWT (+12.4m) were maintained (study IV).In conclusion both walking measures showed good concurrent validity and test-retest reliability and have the potential to be used in both clinical practice and research (study I). High levels of sedentary time and low daily step counts were demonstrated in RTT (study II). Parents and professionals described how opportunity for participation in ‘uptime’ activities depended on a balance of facilitators and barriers within the individual with RTT and the environment (study III). Knowledge from study I-III enabled the planning and implementing of a health-promoting intervention. The U-PART intervention was considered feasible with regards to acceptability and practicality, and positive effects were seen in the outcomes of sedentary time, daily step count, walking capacity, quality of life and participation-level goals, some maintained after a further 12 weeks (study IV).This thesis contributes important knowledge to disability research in Denmark and internationally by focusing on the availability of valid outcome measures and health-promoting strategies in girls and women with RTT.
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| 6. |
- Stahlhut, Michelle, et al.
(författare)
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Patterns of sedentary time and ambulatory physical activity in a Danish population of girls and women with Rett syndrome
- 2019
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Ingår i: Disability and Rehabilitation. - : Informa UK Limited. - 0963-8288 .- 1464-5165. ; 41:2, s. 133-141
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Tidskriftsartikel (refereegranskat)abstract
- Background: Rett syndrome (RTT) is a rare neurodevelopmental disorder leading to multiple disabilities and high dependency on caregivers. This study aimed to: (1) describe the patterns of sedentary time and daily steps and (2) identify the association of individual and environmental characteristics with sedentary time. Methods: All Danish females with RTT older than 5 years of age and with a MECP2 mutation were invited to participate. The activPAL and StepWatch Activity Monitor (SAM) were worn by participants for at least four days. Sedentary time and step counts were plotted by time to examine daily activity patterns. Associations between sedentary time and individual and environmental covariates were assessed with linear regression models. Results: The median (interquartile range) age of participants was 22.0 (14.3–36.5) years. On average 83.3% (standard deviation 13.9%) of waking hours were spent in sedentary behaviours (n = 48) and the median (interquartile range) daily step count was 5128 (2829–7704) (n = 28). Females older than 33.5 years, and those unable to walk independently were more sedentary. Conclusions: This study demonstrated high levels of sedentary time and low daily step counts in a Danish population of females with RTT. Advancing age and lower walking skills were associated with higher levels of sedentary time.Implications for RehabilitationSedentary lifestyles in individuals with disabilities have a negative impact on health and quality of life.High levels of sedentary time and low daily step counts were demonstrated in a Danish population of females with Rett syndrome.Advancing age and inability to walk independently were strongly associated with higher levels of sedentary time in females with Rett syndrome.Understanding patterns of sedentary behaviour and physical activity can aid health care professionals in developing health-promoting physical activity interventions.
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