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Past and Current Use of Walking Measures for Children With Spina Bifida: A Systematic Review
Accepted Manuscript Past and current use of walking measures for children with spina bifida: a systematic review Derek L. Bisaro, MPT, Julia Bidonde, PhD, Kyra J. Kane, MSc, Shane A. Bergsma, PhD, Kristin E. Musselman, PhD PII:
S0003-9993(15)00384-6
DOI:
10.1016/j.apmr.2015.04.014
Reference:
YAPMR 56185
To appear in:
ARCHIVES OF PHYSICAL MEDICINE AND REHABILITATION
Received Date: 12 February 2015 Revised Date:
16 April 2015
Accepted Date: 21 April 2015
Please cite this article as: Bisaro DL, Bidonde J, Kane KJ, Bergsma SA, Musselman KE, Past and current use of walking measures for children with spina bifida: a systematic review, ARCHIVES OF PHYSICAL MEDICINE AND REHABILITATION (2015), doi: 10.1016/j.apmr.2015.04.014. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Running Head: Walking measurement in spina bifida
review
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Past and current use of walking measures for children with spina bifida: a systematic
Derek L Bisaro1 MPT, Julia Bidonde1 PhD, Kyra J Kane1 MSc, Shane A Bergsma2 PhD, Kristin
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E Musselman1,3,4 PhD
School of Physical Therapy, College of Medicine, University of Saskatchewan, Saskatoon, SK;
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Department of Computer Science, College of Arts and Science, University of Saskatchewan,
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Saskatoon, SK; 3Toronto Rehabilitation Institute – University Health Network, Toronto, ON; Department of Physical Therapy, Faculty of Medicine, University of Toronto, Toronto, ON.
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This work has been accepted for presentation at the 2015 meeting of the Canadian Physiotherapy Association.
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This work was supported by grants from the Spina Bifida and Hydrocephalus Foundation of Canada and the College of Medicine, University of Saskatchewan, to KEM.
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Corresponding Author:
Kristin Musselman PT, PhD SCI Mobility Lab, Lyndhurst Centre – TRI 520 Sutherland Drive, Toronto, ON, M4G 3V9 [email protected] 416-597-3422 ext.6190
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Running Head: Walking measurement in spina bifida
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Past and current use of walking measures for children with spina bifida: a systematic
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ABSTRACT
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Objective: To describe walking measurement in children with spina bifida, and to identify
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patterns in the use of walking measures in this population.
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Data Sources: Seven medical databases were searched from inception until March 2014. Search
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terms encompassed three themes: 1) children, 2) spina bifida, and 3) walking.
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Study Selection: Articles were included if participants were children aged 1-17 years with spina
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bifida, and if walking was measured. Articles were excluded if the assessment was restricted to
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kinematic, kinetic or electromyographic analyses of walking. A total of 1,751 abstracts were
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screened by two authors independently, and 109 articles were included in this review.
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Data Extraction: Data were extracted using standardized forms. Extracted data included study
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and participant characteristics, and details about the walking measures used, including
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psychometric properties. Two authors evaluated the methodological quality of articles using a
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previously published framework that considers sampling method, study design, and
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psychometric properties of the measures used.
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Data Synthesis: Nineteen walking measures were identified. Ordinal-level rating scales (e.g.,
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Hoffer Functional Ambulation Scale) were most commonly used (57% of articles), followed by
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ratio-level, spatiotemporal measures, such walking speed (18% of articles). Walking was
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measured for a variety of reasons relevant to multiple health care disciplines. A machine learning
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analysis was used to identify patterns in the use of walking measures. The learned classifier
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predicted whether or not a spatiotemporal measure was used with 77.1% accuracy. A trend to use
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spatiotemporal measures in older children and those with lumbar and sacral spinal lesions was
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identified. Most articles were prospective studies that used samples of convenience and
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unblinded assessors. Few articles evaluated or considered the psychometric properties of the
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walking measures.
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Conclusions: Despite a demonstrated need to measure walking in children with spina bifida, few
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valid, reliable and responsive measures have been established for this population.
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Keywords: walking, spinal dysraphism, review
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Spina bifida is one of the most common congenital birth defects, with a current prevalence of
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about 0.3-0.4/1,000 births in Canada1 and the United States2. With this condition, a neural tube
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defect results in damage to the spinal cord, brain and/or meninges. Walking is a challenge for
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many children with spina bifida. More than half of those with neurological lesions at or below
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the thoracic level achieve walking at some point in their childhood, with the walking rate
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increasing as the lesion level decreases.3-6 As a result of their sensorimotor impairments, many
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children with spina bifida walk with abnormal gait patterns. This significantly increases the
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energy cost of walking and reduces their walking endurance.7-11 A notable proportion of children
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with spina bifida lose the ability to walk as they age.4,5
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Achieving and/or improving the ability to walk is an important goal for many children with spina
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bifida and their families, as walking enables greater participation in daily activities, recreation,
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and contributes positively to quality of life. As a result, walking is often a focus of the medical
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management and rehabilitation of those children who have the potential to walk.3 Considerable
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effort is spent investigating therapeutic approaches that may lead to improved walking outcomes
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for children with spina bifida, such as walking training11,12, surgical procedures13,14, neural
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prostheses15 and orthoses16-18.
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In order for researchers and clinicians to accurately evaluate the effects of an intervention on
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walking, and to follow a child’s walking ability over time, valid, reliable and responsive
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measures of walking must be used. Laboratory-based assessments involving sophisticated
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motion analysis systems can measure kinematic gait characteristics in children with spina
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bifida.16,19 Since most clinicians do not have access to such systems, walking measures that can
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be administered in a clinical setting are required. Furthermore, the measures must have good
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psychometric properties (i.e., validity, reliability, responsiveness) to be useful. To date, however,
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there is little guidance for clinicians and researchers as to what walking measures are useful for
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children with spina bifida.
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As a first step toward providing guidance on how to assess walking in children with spina bifida,
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we performed a systematic review and critical evaluation of the literature. Our objectives were
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to: 1) identify the walking measures used in clinical settings for children with spina bifida, 2)
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describe the circumstances under which these measures were used, and 3) evaluate these
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measures with respect to their psychometric properties reported in the literature.
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METHODS
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A systematic review was performed following the PRISMA (Preferred Reporting Items for
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Systematic Reviews and Meta-Analysis) guidelines.20 There is no protocol for this review. To
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form the review question, a modified PICOS (population, interventions or indicators,
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comparators, outcomes and study design) framework was used. The population was children
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(i.e., aged 1-17 years, inclusive) with spina bifida. The indicator was a measure of walking that
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could be used in a clinical setting. There were no comparators. The outcome of interest was
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walking ability, and there were no restrictions on study design.
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Search Strategy
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A literature search was completed in consultation with an Information Specialist at the
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University of Saskatchewan. Seven databases were searched (Medline, PubMed, Embase,
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Scopus, Web of Science, Cinahl, and Amed) from the earliest known record until March 11,
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2014. The search terms included keywords and controlled vocabulary (where applicable) and
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focused on the following themes: 1) children, 2) spina bifida, and 3) walking (see Appendix 1 for
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search example). No restrictions were placed on the language, date, or type of publication.
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Duplicate records were removed using a research management tool (REFworks, Bethesda, MD).
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All abstracts were then independently reviewed by two authors (___ and ___) to select the
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articles for full text review. The inclusion criteria were as follows:
1) Study participants were children with spina bifida (diagnoses included: spinal
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dysraphism, neural tube defects, meningomyelocele, congenital or nontraumatic spinal
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cord injuries, spina bifida, myelomeningocele, lipomyelomeningocele, lipomeningocele,
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spinal cord malformation).
2) Functional walking capacity, defined as “the ability to ambulate daily using reciprocal
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steps overground for short distances” with or without assistive devices 21, was assessed or
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reported in some way. This definition of was chosen since it reflects walking that is
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clinically relevant.
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Exclusion criteria included:
1) Studies where the participants were exclusively infants (i.e. <1 year old) or exclusively
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adults (i.e., >18 years old).
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2) Studies that did not include any individuals with spina bifida.
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3) Studies that did not examine walking function (e.g., examined gross motor function or
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wheeled mobility).
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4) Studies in which walking was evaluated only through kinematic, kinetic or electromyographic analyses (i.e., measures not routinely used in clinical settings). 5) Studies in which walking ability was given a dichotomous classification of ambulatory or non-ambulatory.
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6) Animal studies, modelling studies, narrative review articles, conference proceedings, editorials or letters to the editor.
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Data extraction
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The following data were extracted from included full text articles using a standard data
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extraction form: patient populations tested, type of neural tube defect, level of lesion, age of
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participants, number of child participants with spina bifida, method of participant recruitment,
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methods of walking assessment, purpose of walking assessment, psychometric properties of the
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walking measures used, results of walking measurement, and type of study. For articles that were
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not in English, individuals proficient in the language and/or Google Translate assisted with
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translation, and in one case the author of the article was contacted for the information in English.
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Methodological quality of the included articles was assessed by adapting the methods of Dobson
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et al.22 Nine methodological characteristics were assessed, such as the sampling method and
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psychometric properties of the walking measures used (see Table 1). These categories were rated
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as adequate /inadequate or stated/not stated depending on the question. Methodological quality
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was evaluated by a pair of reviewers independently and the final decisions reached through
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consensus.
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(Table 1 near here)
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Data Synthesis
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Descriptive summaries
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Extracted data from all included articles were summarized to describe the use of walking
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measures among children with spina bifida. As this is a descriptive review, there is no principal
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summary measure. The following analyses were completed:
1) The number of times a given measure was used was counted and expressed as a
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percentage of the total number of times a walking measure was used in the included
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articles.
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2) The number of times a given study purpose was reported was counted. For example,
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possible study purposes included investigating the relationship between walking ability
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and another variable, or examining the effects of a surgical or orthotic intervention. 3) For each walking measure identified, information concerning its validity, reliability,
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responsiveness and/or interpretability (e.g., interpretation of scores relative to normative
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data, cut-off values) was aggregated.
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Statistical machine learning analysis
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To further describe how walking measures are used in children with spina bifida, we determined
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whether there was any pattern to the selection of a measure. For example, are categorical rating
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scales chosen because of participant age, and/or because of the range of lesion levels? If such
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patterns exist, they would inform future measurement guidelines. The included studies were
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classified along two separate dimensions. First, each study was classified as to whether it
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included a spatiotemporal, ratio-level measure (e.g., walking speed or distance) or not. Second,
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each study was classified as to whether it included a categorical, ordinal-level measure (e.g.,
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Hoffer Functional Ambulation Scale) or not. While not all measures identified in this review fell
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into one of these two categories (e.g., the Walking, Running and Jumping dimension of the
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Gross Motor Function Measure), all studies included at least one measure that did. To detect
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whether there were any patterns to the selection of spatiotemporal or categorical measures, we
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performed a statistical analysis using techniques from the discipline of machine learning.
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Specifically, we trained two decision tree classifiers23 to predict whether (A) a spatiotemporal
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measure would be used or not, and (B) whether a categorical measure would be used or not. For
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both the (A) and (B) analyses, the classifiers made their predictions on the basis of the following
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inputs from each study: (1) the reason for the walking assessment, (2) the minimum and
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maximum ages of the children tested, (3) the rostral and caudal levels of spinal lesion (labeled as
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thoracic, lumbar or sacral), and (4) the publication date.
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The C4.5 decision tree algorithm was chosen as the classifier because it performs well on a
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variety of problems, and generates a learned model that is easily interpretable.23 Moreover, as
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opposed to logistic regression, C4.5 is able to consider nonlinear combinations of the inputs, and
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is therefore able to learn a more expressive predictive model. To train the C4.5 classifier, the
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Weka data mining tool was used (with default parameter settings).24
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After training the classifier, we tested its ability to predict the output class on new, previously-
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unseen data. Specifically, we determined whether the classifier discovered a non-trivial
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relationship between the inputs and the output class. A trivial relationship is one where the most
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common output is always selected, regardless of the inputs. In machine learning terminology,
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such a classifier is referred to as the majority-class baseline. For example, in our analysis, the
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majority-class baseline would always predict ‘categorical’ as the output class, since categorical
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measures were more commonly used. If the C4.5 classifier was able to predict the correct output
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at a level above the majority-class baseline, it would suggest that there was some non-trivial
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pattern to the choice of walking measure. To evaluate the classifier’s prediction accuracy on new
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data, 10-fold-cross-validation was used. McNemar’s Test was used to determine whether the
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learned classifier was more accurate than the majority-class baseline. Alpha was set at 0.05.
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Analysis of risk of bias
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Lastly, the overall risk of bias (i.e., bias of the walking measurements performed) of the included
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articles was evaluated by synthesizing the data extracted regarding methodological quality. Risk
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of bias may be high, low, or unclear (i.e., insufficient information).25 Information concerning
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sampling, blinding, and the selection of a walking measurement were considered. Sampling bias
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was considered high if the majority of studies used samples of convenience, and low if the
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majority of studies used community- or population-based samples. If blinded assessors were
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frequently used for the assessment of walking, the risk of bias was considered low, otherwise the
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risk was high. For the selection of a walking measure, whether or not the psychometric
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properties of a measure were considered by the study authors was used to gauge risk (i.e., if
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considered, then low risk of bias).
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RESULTS
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A total of 1,751 abstracts were screened, and 217 met the inclusion criteria for full text review.
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After full text review, 109 articles were included in the study (see Figure 1). A summary of the
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data extracted from each included article can be found in Appendix 2. Among the included
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articles, many did not specify the type of spina bifida studied. In those that did, the most
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common type studied was myelomeningocele followed by lipomyelomeningocele and
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meningocele. Twenty-one (19%) of the included studies involved other pediatric populations in
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addition to spina bifida, with cerebral palsy and spinal cord injury being the most frequent. The
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ages of the children studied spanned the entire childhood range (i.e., 1 – 17 years).
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Methodological quality of included studies
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The quality evaluation summary for each included article can be found in Appendix 3, with the
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exception of the question regarding whether or not the assessor was blinded. Only 3 of the 109
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studies stated that a blinded assessor was used.26-28 The remaining studies did not comment on
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this methodological aspect, making the risk of bias unclear.
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Samples of convenience were used in 82 studies (75.2%), placing the study findings at a greater
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risk of bias than the studies that used community- and population-based samples (5.5% and
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1.8%, respectively). The sampling method was not outlined in 19 studies (17.4%). Thus, overall
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the risk of sampling bias in the included articles was high.
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Participant characteristics (age, gender, number and type of neural tube defect) were adequately
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defined in 65 studies (59.6%). All details, except gender, were reported in 8 studies (7.3%, rated
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‘partial’, Table 1), whereas the characteristics were inadequately reported in 36 studies (33%).
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The inclusion and exclusion criteria of the study were outlined in 47 studies (43.1%), while 37
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studies (33.9%) specified 1-2 criteria (rated ‘Limited’ as per Table l). A notable number of
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studies (25 or 22.9%) did not list any inclusion or exclusion criteria for their study participants.
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Most studies were prospectively planned and executed (77.1%).
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Few studies considered the psychometric properties of the walking measures used, contributing
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to a high risk of bias. Fifteen studies (13.8%) reported on the reliability of the measure
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used9,13,26,27,29-39; however, the reported reliability was rarely established in children with spina
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bifida. Two exceptions were the 6-minute walk test and Timed Up and Go test; both having high
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test-retest reliability in children with spina bifida.30,38 Likewise, at least one type of validity was
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reported or tested for the walking measures used in 10 studies (9.2%).13,27,29,31,32,34,35,37,38,40
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Interpretability of a measure was reported at a slightly higher frequency (19 studies or
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17.4%).7,9,12,29-35,37,38,41-47 Most commonly, the scores on a walking measure were compared to
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control participants or normative data.7,9,29,31,32,34,35,41,43-47 The standard error of measurement and
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smallest detectable difference were calculated for the 6-minute walk test in children with spina
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bifida.30,33 In Williams et al.38 responsiveness was assessed for the Timed Up and Go Test in
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typically-developing children, but not in the study participants with spina bifida.
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Walking measures used in included articles
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Nineteen different walking measures were used across the 109 articles. The Hoffer Functional
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Ambulation Scale was most commonly used by researchers (36% of the articles). The Hoffer
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Scale is an categorical scale with four levels: community ambulator, household ambulator, non-
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functional ambulator, and non-ambulator.48 Various modifications to the Scale have been
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made13,26; however, it has remained an ordinal-level measure that outlines descriptive categories
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of walking. Another 21% of studies used custom-made, categorical scales, similar in nature to
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the Hoffer Scale. These scales could be divided into three groups based on the variable used to
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categorize the walking. Many scales were based on the type of assistive device used 29,41,49,50-59,60-
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walking distance as a means to define walking categories.72-74
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or the walking environment (e.g., outdoors/community, indoor/household)42, 63-71. Fewer used
Walking speed was also measured (18% of included articles), however, the method used varied.
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Some measured the speed over a pre-determined distance, such as 30m43,75-77, 6m78, 110m79 or a
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60.5m oval track44. Others used a pre-determined walking time to obtain a measure of speed
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(e.g., walking for 3 minutes45,77,80 or 6 minutes as part of the 6-minute walk test7,30,81). Only two
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studies used the 10-meter walk test.31,32 Self-selected walking speed was always measured,
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whereas fastest/maximum speed was also measured in four studies.43,44,76,82
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Measures less frequently used included the 6-minute walk test to measure walking
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distance7,9,10,26,30,33,83 (5% of articles), the Mobility Domain of the Pediatric Evaluation of
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Disability Inventory (PEDI)12,13,34-36,41,84,85 (5% of articles), and the Mobility Domain of the
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Pediatric Functional Independence Measure (WeeFIM)29,37,86-89 (4% of articles). The remaining
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11% of articles included outcomes that were used only 1-3 times: Timed Up and Go test38,
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Physiological Cost Index of Walking84,90,91, Gross Motor Function Measure32, Functional
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Independence Measure39, Movement Assessment Battery for Children34, Functional Ambulation
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Categories88, Activities Scale for Kids27, Functional Mobility Scale12,92, Personal NEM (Necker-
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Enfants Malades) motor scale93, and the Progressive Ambulation Scale89. Two studies rated the
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children’s ability to complete a number of functional tests, such as ascending and descending
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steps94,95 and a ramp95.
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Reasons for walking measurement
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Walking was evaluated in children with spina bifida for a variety of reasons. Most commonly,
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walking was measured so that the relationship between walking ability and another variable
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could be examined (Figure 2). For example, there was an interest in describing or quantifying the
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relationship between walking ability and quality of life35,87, wait time for rehabilitation37, spinal
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deformity27, extent of neurological impairment85 and peak VO27. Examining the effect of an
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orthotic device or surgical intervention on walking performance were also common reasons for
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measuring walking ability, whereas investigating the effect of a physical intervention, such as
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treadmill training12,26 or neuromuscular electrical stimulation89,94, was less common. A number
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of studies measured walking in order to describe the walking status of participants, either at one
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point in time6,34,48,50,56,96-101 or longitudinally54,73,102-105. There was also an interest in predicting
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the future walking status of children.40,51-53,65,103,106-108 Less commonly, walking was measured
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for methodological reasons, such as determining study eligibility30,33,83,90 or to assist with the
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prescription of exercise testing83. Only four studies examined one or more psychometric
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properties of a walking measure in the spina bifida population.31,33,36,38
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(Figure 2 near here)
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Results of machine learning analysis
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The machine learning analysis found a non-trivial pattern for the spatiotemporal output class, but
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not the categorical output class. For the spatiotemporal output class, the accuracy of the majority-
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class baseline was 70.6%. This means that if the majority class (i.e., did not use a spatiotemporal
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measure) was selected for every article, the resulting accuracy was 70.6%. In comparison, the
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accuracy from 10-fold-cross validation of the learned classifier was 77.1%. The difference in
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accuracy between the learned classifier and the majority-class baseline did not reach statistical
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significance (p=0.15).
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Figure 3 shows the decision tree algorithm for the spatiotemporal output class. The learned
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classifier could predict whether or not a spatiotemporal measure of walking was used by
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considering three of the four inputs: (1) the reason for the walking assessment (i.e., the study
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purpose), (2) the minimum age of the children tested, and (3) the rostral level of spinal lesion. If
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the reason for the walking assessment was to evaluate the effects of an orthosis, then a
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spatiotemporal measure was used. If the reason was to investigate the effects of surgery or to
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predict future walking status, then a spatiotemporal measure was not used (i.e., a categorical
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rating scale was used instead). When the purpose of the walking assessment was to describe
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walking ability (cross-sectional or longitudinal analyses), the choice was dependent on age. If the
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study sample included young children (<3 years of age), then spatiotemporal measures were not
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used. However, if all study participants were older than 3 years, the choice was further
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determined by the participants’ level of spinal lesion. If the sample included children with
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thoracic lesions, spatiotemporal measures were not used. If the sample included children with
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lumbar and sacral lesions only, then spatiotemporal measures were used. Likewise, if the reason
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for walking assessment was to investigate the relationship between walking and another variable,
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the same decision rule for the input of lesion level was seen.
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(Figure 3 near here)
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DISCUSSION
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We identified nineteen clinical measures that have been used to assess walking ability in children
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with spina bifida through a systematic review that followed PRISMA guidelines. The review
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resulted in 109 included articles, with publication dates spanning four decades. The nineteen
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walking measures identified were used for a variety of study purposes across multiple disciplines
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of medicine and rehabilitation. The Hoffer Functional Ambulation Scale48 and other categorical
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scales were most commonly used. Walking speed was also utilized as an outcome, although there
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was no standard approach to its measurement. By employing a machine learning analysis, we
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found a trend toward using spatiotemporal measures for children school-aged and older, for those
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with a lower level of spinal lesion, and when investigating the effects of an orthosis. Little work
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has been done to validate any of the identified walking measures, or to assess their
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responsiveness, in the spina bifida population. The reliability of the measures has also not been
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established the spina bifida population, with the exception of test-retest reliability of the 6-
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minute walk test in higher-functioning walkers30,33 and the Timed Up and Go test in a small
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sample38. Hence, while there is a clear interest and need to measure walking in children with
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spina bifida across multiple health care disciplines, clinicians and researchers lack valid, reliable
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and responsive measurement tools.
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The Hoffer Functional Ambulation Scale
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The Hoffer Scale, which was specifically designed for spina bifida48, was the most commonly
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used measure across the studies included in this review. This Scale is an appealing option for
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clinical use for several reasons. First, it is easy to use and does not require any training prior to
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use. Second, it can be administered quickly with little effort required from the child. Third, the
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Hoffer Scale is inclusive of all levels of walking ability, from fully independent to unable to
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walk. This means that all children with spina bifida, regardless of physical ability, can be rated
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on this one measure. Fourth, it can be used across the lifespan of someone with spina bifida.
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Consistent with these latter two points are our findings that there was a tendency to use
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categorical rating scales in studies involving young children and/or those with thoracic-level
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lesions (see Figure 3). And lastly, the Scale was shown to have convergent validity.40 Thus, there
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are numerous advantages to using the Hoffer Scale in clinical environments.
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Formal assessment of the clinical utility of the Hoffer Functional Ambulation Scale supports its
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use in clinical practice. Tyson and colleagues109 report a tool to assess the clinical utility of
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walking and mobility measures in neurological populations. The tool rates an outcome measure
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on four dimensions: time required, cost, need for equipment or training, and portability. A total
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score out 10 is assigned, with scores of 9 or 10 suggesting the measure is appropriate for clinical
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use.109 Application of their tool to the Hoffer Scale would result in a score of 10. Thus, the
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feasibility of using the Hoffer Scale in a clinical setting is high.
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Despite numerous advantages of the Hoffer Scale, there are two noteworthy limitations. With
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only four levels of ambulatory ability to choose from, the Hoffer Scale likely lacks
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discriminative validity and responsiveness. For example, two children can walk outdoors for
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short distances; however, one child walks with bilateral ankle foot orthoses at a speed of 0.8m/s,
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while the other walks with a walker at a speed of 0.4m/s. Despite considerable differences in
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their speeds and reliance on assistive devices, both children are classified as community
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ambulators. Thus, the Scale does not adequately discriminate between different ambulatory
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abilities. Similarly, the Hoffer Scale may only be able to detect very large changes in walking
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function (i.e., lacks responsiveness); however, this is speculative and needs to be confirmed
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through proper psychometric study. Overall, the Hoffer Functional Ambulation Scale provides a
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snapshot of a child’s walking ability, and is likely an effective way to describe a child’s walking
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status at a single point in time.
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Walking speed
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In addition to categorical rating scales, walking speed was measured in the included articles, but
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at a considerably lower frequency. Measuring walking speed has numerous advantages. Its
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clinical utility is high, as it is low-cost, portable, time-efficient, and does not require equipment.
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The 5-meter and 10-meter walk tests score 10/10 on the evaluation of clinical utility.109
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Furthermore, speed is a metric that is easily interpretable, and possibly more responsive than
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categorical scales.
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Despite these advantages, spatiotemporal measures, like speed, were not used to evaluate the
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effects of surgery or to predict future walking status. It is easier to make a prediction using a
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categorical rating scale than a ratio-level measure. Why spatiotemporal measures have not been
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used to assess the efficacy of surgery is not clear, but they are likely suitable for this study
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purpose.
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How to best measure walking speed in pediatric populations is unknown. We found that the
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walking distance over which speed was measured varied greatly, from as short as 6m78 to as long
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as the distance that could be covered in 10 minutes31. Self-selected speeds measured over
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different walking distances have been found to differ when directly compared in children with
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spina bifida and other neuromuscular diseases.31 Shorter distances are likely most feasible for
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children; compliance may be an issue with longer tests. Longer distances would also preclude
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lower-functioning walkers from participation. For example, De Groot and colleagues7 used the 6-
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minute walk test in those children rated as ‘Normal’ or ‘Community’ ambulators on the Hoffer
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Scale.
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In our review, we found little investigation of the validity, reliability and responsiveness of
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walking speed measurement in children with spina bifida.30 In adolescents with cerebral palsy,
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the 10-meter walk test has been shown to be a valid measure.110 However, the test-retest
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reliability of the 10-meter walk test (performed at a fast speed) was found to be inadequate in
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children with cerebral palsy aged 4-18 years.111 Yet, in the same study, the reliability of the 6-
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minute walk test was found to be excellent.111 Thus, while longer walking tests may not be as
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practical for pediatric populations, they may yield more reliable results. In addition, longer
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distances may be more representative of walking performance in the community.31 Investigation
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of the psychometric properties of short and long walking tests to measure speed in children with
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spina bifida is warranted. Adopting a standard approach to the assessment of walking speed for
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ambulatory children with spina bifida would facilitate accurate tracking of client progress and
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comparison of research findings across studies.
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Methodological quality of included articles
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We found that the majority of included articles involved samples of convenience and likely
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unblinded assessments. A notable proportion of the studies did not describe the participants’
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characteristics or the inclusion/exclusion criteria adequately. Furthermore, few articles explicitly
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considered the psychometric properties of the walking measures used. All together this
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contributes to a greater risk of bias for the measurement findings.
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Gaps in walking measurement for spina bifida
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Walking is a complex behavior that can be measured along different dimensions. Walking
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capacity refers to the ability to execute the task of walking, whereas walking performance refers
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to the ability to walk in one’s usual environment.112 Many of the measures identified in this
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review are indicators of walking capacity (e.g., 10-meter walk test), however, some of the
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ordinal scales probe walking performance. Our results suggest that psychometrically-sound
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measures of walking performance are needed for children with spina bifida, as has been
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suggested for neurological populations112.
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Furthermore, walking in one’s usual environment involves many different walking skills, such as
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negotiating obstacles and different surfaces.113 Most measures identified in this review assess a
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single walking task (i.e., walking over level ground). Quantitative scales consisting of multiple
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tasks relevant to daily walking are lacking for children with spina bifida. Such scales exist for
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adult populations (e.g., the Spinal Cord Injury Functional Ambulation Profile114), and could be
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adapted for pediatric populations.
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Strengths and limitations of review
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There are several noteworthy strengths of this systematic review. First, we described the current
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state of walking measurement in children with spina bifida, and identified areas in need of future
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research. Second, we incorporated a unique analysis (i.e., machine learning) into the review that
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provided richer insight into how the identified walking measures have been used in past research.
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Lastly, this review could serve as a model for future systematic reviews that aim to characterize
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the use of outcome measures in a patient population.
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There are also potential limitations to note. First the overall risk of bias of the included articles
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was deemed high. This means that although the review can accurately report what walking
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measures were used, appropriateness of these measures for the specific study purpose and
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participants may be questionable. The measures reported in this review do not necessarily
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represent the best measurement options. Second, the methods used to measure walking may
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change over time. We did not place a limit on the publication date of the articles in order to
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collect a sufficiently large amount of data. It is possible that the review results would be different
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had we limited our review to research conducted in the past decade. However, the machine
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learning analysis did not find a relationship between publication date and the choice of walking
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measure (i.e., spatiotemporal or categorical). Lastly, the focus of this review was walking, which
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is just one of the many gross motor skills important in childhood. Measures of general gross
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motor function are likely clinically useful for children with spina bifida. Future work could
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explore the state of gross motor function assessment in this population.
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Recommendations for walking measurement in children with spina bifida
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Currently, clinicians and researchers have few valid and reliable walking measures to choose
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from for children with spina bifida. A variety of custom-made, categorical scales were
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commonly used. For the time-being, we recommend using the Hoffer Functional Ambulation
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Scale as a means to classify a child’s walking at a single point in time. This will help to increase
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consistency across research and clinical practice. Further work is needed to evaluate the validity
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(e.g., discriminative) and reliability (e.g., test-retest, inter-rater) of the Hoffer Scale. If working
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with a child with good walking function (i.e., ‘Community’ or ‘Normal’ ambulators on the
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Hoffer Scale), the 6-minute walk test is appropriate and supported by the literature.30,33 Future
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research is needed to evaluate the use of the 6-minute walk test and other spatiotemporal
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measures in young children.
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We propose that both the research and clinical realms of spina bifida management would benefit
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from an organized approach to walking measurement. The measures should be chosen with a
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child’s age and lesion or functional level in mind, while at the same time reflect the multiple
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dimensions of walking function. The assessment could include both a categorical rating scale
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(i.e., Hoffer Scale) and also spatiotemporal measures. A small proportion (15%) of the articles
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reviewed included both categorical and spatiotemporal measures. Future work should build upon
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previous pediatric research to identify walking measures that are valid, reliable and responsive
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for children with spina bifida.
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Conclusions
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Through a systematic review we identified nineteen measures that have been used to examine
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walking in children with spina bifida. Spatiotemporal measures were more likely to be used for
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older children, and in children with lower spinal lesions (i.e., lumbar and sacral lesions). Little
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work has been done to establish the validity, reliability and/or responsiveness of walking
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measures in this population. Future research should focus on this knowledge gap so that clinical
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guidelines can be developed for walking measurement in spina bifida.
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2. Boulet SL, Yang Q, Mai C, Kirby RS, Collins JS, Robbins JM, Meyer R, Canfield MA, Mulinare J, National Birth Defects Prevention Network. Trends in the postfortification prevalence of spina bifida and anencephaly in the United States. Birth Defects Res A Clin Mol Teratol 2008; 82: 527-32.
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3. Díaz Llopis I, Bea Muñoz M, Martinez Agulló E, López Martinez A, García Aymerich V, Forner Valero JV. Ambulation in patients with myelomeningocele: a study of 1500 patients. Paraplegia 1993; 31: 28-32. 4. Iborra J, Pagès E, Cuxart A. Neurological abnormalities, major orthopaedic deformities and ambulation analysis in a myelomeningocele population in Catalonia (Spain). Spinal Cord 1999; 37: 351-7. 5. Williams EN, Broughton NS, Menelaus MB. Age-related walking in children with spina bifida. Dev Med Child Neurol 1999; 41: 446-9. 6. Asher M, Olson J. Factors affecting the ambulatory status of patients with spina bifida cystica. J Bone Joint Surg Am 1983; 65: 350-6.
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7. De Groot JF, Takken T, Schoenmakers MA, Vanhees L, Helders PJ. Limiting factors in peak oxygen uptake and the relationship with functional ambulation in ambulating children with spina bifida. Eur J Appl Physiol 2008; 104: 657-65. doi: 10.1007/s00421-008-0820-9. 8. Bare A, Vankoski SJ, Dias L, Danduran M, Boas S. Independent ambulators with high sacral myelomeningocele: the relation between walking kinematics and energy consumption. Dev Med Child Neurol 2001; 43: 16-21.
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71. Marreiros H, Monteiro L, Loff C, Calado E. Fractures in children and adolescents with spina bifida: the experience of a Portuguese tertiary-care hospital. Dev Med Child Neurol 2010; 52: 754-9. doi: 10.1111/j.1469-8749.2010.03658.x.
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72. Hunt G, Lewin W, Gleave J, Gairdner D. Predictors in open myelomeningocele with special reference to sensory level. Br Med J 1973; 4: 197-201. 73. Bartonek A, Saraste H, Samuelsson L, Margareta S. Ambulation in patients with myelomeningocele: a 12 year follow-up. J Pediatr Orthop 1999; 19: 202-6.
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74. Dudgeon BJ, Jaffe KM, Shurtleff DB. Variations in midlumbar myelomeningocele: implications for ambulation. Ped Phys Ther 1991; 3: 57-62.
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75. McCall RE, Schmidt WT. Clinical experience with the reciprocal gait orthosis in myelodysplasia. J Pediatr Orthop 1986; 6: 157-61. 76. Findley TW, Agre JC. Ambulation in the adolescent with myelomeningocele II: oxygen cost of mobility. Arch Phys Med Rehabil 1987; 69: 855-61. 77. Bartonek A, Saraste H, Knutson LM, Eriksson M. Orthotic treatment with Ferrari kneeankle-foot orthoses. Pediatr Phys Ther 1999; 11: 33-8.
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78. Rose GK, Stallard J, Sankarankutty M. Clinical evaluation of spina bifida patients using hip guidance orthosis. Dev Med Child Neurol 1981; 23: 30-40. 79. Bartonek A, Eriksson M, Saraste H. Heart rate and walking velocity during independent walking in children with low and midlumbar myelomeningocele. Pediatr Phys Ther 2002; 14: 185-90.
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80. Banta JV, Bell KJ, Muik EA, Fezio J. Parawalker: energy cost of walking. Eur J Pediatr Surg 1991; 1(Suppl 1): 7-10. 81. Alman BA, Bhandari M, Wright JG. Function of dislocated hips in children with lower level spina bifida. J Bone Joint Surg 1996; 78B: 294-8.
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82. Lough LK, Nielsen DH. Ambulation of children with myelomeningocele: parapodium versus parapodium with orlau swivel modification. Dev Med Child Neurol 1986; 28: 48997. 83. De Groot JF, Takken T, de Graaff S, Gooskens RHJM, Helders PJM, Vanhees L. Treadmill testing of children who have spina bifida and are ambulatory: does peak oxygen uptake reflect maximum oxygen uptake? Phys Ther 2009; 89: 679-87. doi: 10.2522/ptj.20080328. 84. Bartonek A, Saraste H. Factors influencing ambulation in myelomeningocele: a crosssectional study. Dev Med Child Neurol 2001; 43: 253-60. 30
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85. Norrlin S, Strinnholm M, Carlsoon M, Dahl M. Factors of significance for mobility in children with myelomeningocele. Acta Peediatr 2003; 92: 204-10.
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86. Adzick NS, Thom EA, Spong CY, Brock III JW, Burrows PK, Johnson MP, Howell LJ, Farrell JA, Dabrowiak ME, Sutton LN, Gupta N, Tulipan NB, D'Alton ME, Farmer DL. A randomized trial of prenatal versus postnatal repair of myelomeningocele. New Engl J Med 2011; 364: 993-1004. doi: 10.1056/NEJMoa1014379.
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87. Bier JB, Pierce A, Tremont M, Msall M. Medical, functional and social determinants of health-related quality of life in individuals with myelomeningocele. Dev Med Child Neurol 2005; 47: 609-12.
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88. Labruyère R, Gerbera CN Birrer-Brutscha K, Meyer-Heim A, van Hedel HJA. Requirements for and impact of a serious game for neuro-pediatric robot-assisted gait training. Res Dev Disabil 2013; 34: 3906-15. doi: 10.1016/j.ridd.2013.07.031. 89. Walker JL, Ryan SW, Coburn TR. Does threshold nighttime electrical stimulation benefit children with spina bifida? Clin Orthop Relat Res 2011; 469: 1297-1301. doi: 10.1007/s11999-010-1596-x.
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90. Franks CA, Palisano RJ, Darbee JC. The effect of walking with an assistive devices and using a wheelchair on school performance in students with myelomeningocele. Phys Ther 1991; 71: 570-7. 91. Katz DE, Haideri N, Song K, Wyrick P. Comparative study of conventional hip-knee-anklefoot orthoses versus reciprocating-gait orthoses for children with high-level paraparesis. J Pediatr Orthop 1997; 17: 377-86.
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92. Battibugli S, Gryfakis N, Dias L, Kelp-Lenane C, Figlioli S, Fitzgerald E, Hroma N, Seshadri R, Sullivan C. Functional gait comparison between children with myelomeningocele: shunt versus no shunt. Dev Med Child Neurol 2007; 49: 764-9. 93. Al-Holou WN, Muraszko KM, Garton HJ, Buchman SR, Maher CO. The outcome of tethered cord release in secondary and multiple repeat tethered cord syndrome. J Neurosurg Pediatr 2009; 4: 28-36. doi: 10.3171/2009.2.PEDS08339.
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94. Karmel-Ross K, Cooperman DR, Van Doren CL. The effect of electrical stimulation on quadriceps femoris muscle torque in children with spina bifida. Phys Ther 1992; 72: 723-30. 95. Krebs DE, Edelstein JE, Fishman S. Comparison of plastic/metal and leather/metal kneeankle-foot orthoses. Am J Phys Med Rehabil 1988; 67: 175-85. 96. De Souza LJ, Carroll N. Ambulation of the braced myelomeningocele patient. J Bone Joint Surg Am. 1976; 58: 1112-8. 31
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97. Findley TW, Agre JC, Habeck RV, Schmalz R, Birkebak RR, McNally MC. Ambulation in the adolescent with myelomeningocele I: early childhood predictors. Arch Phys Med Rehabil 1987; 68: 518-22.
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98. Okurowska-Zawada B, Kulak W, Otapowicz D, Sienkiewicz D, Paszko-Patej G, Wojtkowski J. Quality of life in children and adolescents with cerebral palsy and myelomeningocele. Pediatr Neurol 2011; 45: 163-8. doi: 10.1016/j.pediatrneurol.2011.04.006.
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99. Ross M, Brewer K, Wright V, Agur A. Closed neural tube defects: neurologic, orthopedic, and gait outcomes. Pediatr Phys Ther 2007; 19: 288-95.
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100. Samuelsson L, Skoog M. Ambulation in patients with myelomeningocele: a multivariate statistical analysis. J Pediatr Orthop 1988; 569-75. 101. Verhoef M, Barf HA, Post MWM, vanAsbeck FWA, Gooskens RHJM, Prevo AJH. Secondary impairments in young adults with spina bifida. Dev Med Child Neurol 2004; 46: 420-7. 102. Bartonek A, Gutierrez EM, Haglund-Åkerlind, Saraste H. The influence of spasticity in the lower limb muscles on gait pattern in children with sacral to midlumbar myelomeningocele: a gait analysis study. Gait Posture 2005; 22: 10-25.
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103. Guille JT, Benevides R, DeAlba CC, Siriram V, Kumar SJ. Lumbosacral agenesis: a new classification correlating spinal deformity and ambulatory potential. J Bone Joint Surg Am 2002; 84: 32-8.
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104. Lorente Molto FJ, Garrido IM. Retrospective review of L3 myelomeningocele in three age groups: should posterolateral iliopsoas transfer still be indicated to stabilize the hip? J Pediatr Orthop B 2005, 14: 177–84. 105. Müller EB, Nordwall A, Oden A. Progression of scoliosis in children with myelomeningocele. Spine 1994; 19: 147-50.
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106. Bartonek A. Motor development toward ambulation in preschool children with myelomeningocele-a prospective study. Pediatr Phys Ther 2010; 22: 52-60. doi: 10.1097/PEP.0b013e3181cc132b. 107. Swank M, Dias L. Myelomeningocele: a review of the orthopaedic aspects of 206 patients treated from birth with no selection criteria. Dev Med Child Neurol 1992; 34: 104752.
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108. Swank M, Dias LS. Walking ability in spina bifida patients: a model for predicting future ambulatory status based on sitting balance and motor level. J Pediatr Orthop 1994; 14: 7158.
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109. Tyson S, Connell L. The psychometric properties and clinical utility of measures of walking and mobility in neurological conditions: a systematic review. Clin Rehabil 2009; 23: 1018-33. doi: 10.1177/0269215509339004. 110. Chrysagis N, Skordilis EK, Koutsouki D. Validity and clinical utility of functional assessments in children with cerebral palsy. Arch Phys Med Rehabil 2014; 95: 36974. doi: 10.1016/j.apmr.2013.10.025.
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111. Thompson P, Beath T, Bell J, et al. Test-retest reliability of the 10-metre fast walk test and 6-minute walk test in ambulatory school-aged children with cerebral palsy. Dev Med Child Neurol 2008;50:370-6. doi: 10.1111/j.1469-8749.2008.02048.x. 112. Pearson OR, Busse ME, Van Deursen RWM, Wiles CM. Quantification of walking mobility in neurological disorders. Q J Med 2004; 97: 463-75. Doi:10.1093/qjmed/hch084. 113. Musselman KE, Yang JF. Walking tasks encountered by urban-dwelling adults and persons with incomplete spinal cord injuries. J Rehabil Med 2007; 39: 567-74.
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114. Musselman KE, Brunton K, Lam T, Yang JF. Spinal cord injury functional ambulation profile: a new measure of walking ability. Neurorehabil Neural Repair 2011; 25: 285-93. doi: 10.1177/1545968310381250.
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Figure Legends
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Figure 1: PRISMA Flow Diagram. Outline of the identification and screening of abstracts and
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articles.
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Figure 2: Reasons for Walking Assessment in Children with Spina Bifida. Number of included
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articles (bars) for each study purpose (left text). The purpose ‘Describe walking status’ was
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observed in cross-sectional and cohort studies. A study may have more than 1 stated purpose,
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thus the total number of articles reported here exceeds the total number included in the review
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Figure 3: Decision Tree for Choosing a Spatiotemporal Walking Measure in Children with
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Spina Bifida. For ‘Reason of walking measurement’: ‘If orthosis’ = if examining the effects of an
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orthosis on walking. ‘If surgery’ = if examining the effects of a surgical procedure on walking.
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‘If description’ = if describing walking status, at one point or over time. ‘If prediction’ = if
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predicting future walking status. ‘If relationship’ = if investigating the relationship between
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walking and another variable. ‘Rostral level’ refers to the highest level (sacral, lumbar or
938
thoracic) of spinal lesion among study participants.
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Table 1: Study Quality Evaluation Tool (adapted from Dobson et al.22)
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Decision Rules Adequate = all details provided; Partial = all details except gender; Inadequate = missing details. Convenience = participants recruited included patients from local hospital; Community-based = e.g., participants recruited from >1 local hospital or organization with aim of reaching all potential participants in the area; Population-based = as per community-based, but geographical area larger (e.g., country- or state/province- wide); Not stated = no details about sampling method provided. Stated = clear list of both provided; Limited = 1 or 2 points only; Not stated = no details about inclusion or exclusion provided. Prospective = walking data collected at time of study, e.g., objective exam; Retrospective = walking data collected prior to study initiation, e.g., chart review; Not stated = no details provided re: time of walking assessment. Yes; No; Not stated. Yes (list type(s), e.g., inter-rater, test-retest, internal consistency, etc.); No. Yes (list type(s), e.g., concurrent, criterion, content, etc.); No. Yes (list type(s), e.g., minimally important difference, responsiveness, cut-points, etc.); No.
3. Are inclusion and exclusion criteria stated?
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4. Was the walking assessment performed prospectively or retrospectively?
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Question 1. Are participant characteristics defined including age, gender, number, and type of NTD? 2. What sampling method was used?
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5. Was the assessor blinded to the study hypothesis and/or groups? 6. Was the reliability of the tool/classification stated or demonstrated? 7. Was the validity of the tool/classification stated or demonstrated? 8. Was the interpretability or responsiveness of the tool/classification reported or tested? NTD = neural tube defect.
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Appendix 1 PubMed Search
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("child"[MeSH Terms] OR "adolescent"[MeSH Terms] OR "child, preschool"[MeSH Terms] OR "adolescent" OR "adolescence" OR "teen" OR "teenager" OR "youth" OR "children" OR "child" OR "childhood" OR "preschool child" OR "school child" OR "toddler" OR "pediatric") AND ("spinal dysraphism"[MeSH Terms] OR "neural tube defects"[MeSH Terms] OR "meningomyelocele"[MeSH Terms] OR "spinal cord injuries"[MeSH Terms] OR "spina bifida"[All Fields] OR "myelomeningocele" OR "spinal cord injury" OR "neural tube defect" OR "lipomyelomeningocele" OR "lipomeningocele" OR "spinal cord malformation") AND ("walking"[MeSH Terms] OR "mobility limitation"[MeSH Terms] OR "gait"[MeSH Terms] OR "dependent ambulation"[MeSH Terms] OR "walking"[All Fields] OR "mobility" OR "six minute walk test" OR "ten meter walk test" OR "6 minute walk test" OR "10 meter walk test" OR "stepping" OR "ambulation" OR "motion analysis" OR "walking speed" OR "walking difficulty")
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Appendix 2: Description of Included Studies
n with SB Walking status (aged 117 yrs)
Author & Year Populations Walking Measure Reason for walking tested measurement
Type of study
Level of lesion: n
Age: mean + 1SD (range) yrs
Adzick et al. 1 2011
MMC
WeeFIM
Prospective randomized cohort
Thoracic: 7 L1-L2: 31 L3-L4: 75 L5-S1: 45
Follow-up at 1 108 & 2.5 yrs
Agre et al. 2 1987
MMC
Self-selected & To examine Prospective max speeds over relationship between cross-sectional 30m speed, muscle strength & aerobic capacity
Al-Holou et al. 3 2009
MMC & Modified Hoffer To study success of LMMC with scale & Personal untethering surgery tethered cord NEM motor scale
Alman et al. 19964
SB
Hoffer Scale & speed (calculated from 6min of walking on 50m track)
To compare metabolic Prospective energy used in follow-up walking between those who had operative relocation of the hip and those treated conservatively
Ammerman et al. 19985
SB
Categorized as: no assistance, assistive devices (e.g., braces, crutches), wheelchair only
To examine reProspective lationship between survey ambulatory status & psychiatric diagnoses
Apkon et al. 6 2009
MMC
Categorized as: ambulatory, partially ambulatory, nonambulatory
Asher et al. 19837
MMC
Ambulation categorized as: community, household, nonfunctional, nonambulatory
To study factors Prospective affecting ambulatory cross-sectional status in children with SB
Banta et al. 19918
MMC & SCI
Speed during steady state walking (>3min walking duration)
Bartnicki et al. 9 2012
MMC
Hoffer scale
To compare walking while wearing a RGO to walking while wearing the Parawalker To determine if ambulatory function correlated with sitting stability
L3: 6 L3-L4 mixed: 12 L4: 34
Age of untethering 12.3 + 5.9 (1025.9)
Operated 14.5 + 5.8; Conservative 16.4 + 5.4
Not stated; 89 met inclusion
Modified Hoffer: n Normal: 15 Community: 8 Household: 11 Exercise: 9 Nonambulator: 40
Not Speed: Conservative = stated; 52 0.643 + 0.102m/s; total Operated = 0.775 + 0.126m/s
Category: n No assistance: 10 Assistive devices: 36 Wheelchair only: 6
9.75 (4-18)
24
Category: n Ambulatory: 10 Partially ambulatory: 2 Nonambulatory: 12
Thoracic: 28 L1-L2: 12 L3: 20 L4: 17 L5: 9 Sacral: 12
14.33 (5.75 31.83)
T12: 3
Ages of 3 children with SB: 10.83, 10.83, 6.58
Not Category: n stated; 98 Community: 20 (all total with L5 & all the sacral except 1) Nonfunctional: 40 (all those in thoracic-L2) *** All results not indicated 3 Not indicated
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Mean self-selected speed (km/hr): L2 & above: 1.9 L3-4: 3.5 L5-sacral: 3.9 No motor deficit: 4.8 Mean max speed (km/hr): L2 & above: 2.4 L3-4: 6.4 L5-sacral: 9.3 No motor deficit: 16.0
12.94 + 3.59 (6- 54 18)
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Sacral: 5 Lumbar: 23 Thoracolumbar: 9 Thoracic: 3 Other (occipital encephalocele, LMMC): 11 To examine Prospective Thoracic or high relationship between cross-sectional lumbar: 6 ambulatory status & Mid-lumbar: 9 bone mineral density Sacral: 9
Prospective case series
Prenatal surgery group had higher WeeFIM scores & were more likely to walk without orthotics
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Thoracic: 17 Lumbar: 61 Sacral: 6
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L2 & above: 6 12.6 + 1.2 (10- 33 L3-4: 7 15) L5-sacral: 17 No motor deficit: 3
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Compare motor development between prenatal & postnatal surgical repair of MMC
Prospective T12: 7 cross-sectional L1: 4 L3: 2 L4: 4 L5: 2
21.4 (13-35)
Not stated Hoffer: n Community: 6 Househould: 1 Non-ambulators: 12
Hoffer scale
Bartonek et al. MMC 1999b11
Hoffer scale, speed over 30 metres & speed over 3 minutes
Bartonek et al. MMC 12 1999c
Hoffer scale & Outcome looked at walking distance over time categorized as: >1,000 m, 100 1,000 m, 10 - 100 m, <10 m
MMC
Modified Hoffer scale, mobility domain of PEDI, PCI
Prospective cohort
L3: 3 L4: 3 L5: 5 Sacral: 21
To examine how Prospective Not stated motor paresis of cross-sectional lower limbs relates to ambulatory status
17.2 (5-40)
Not stated Hoffer: n Community: 41 Household: 14 Nonfunctional: 11 Nonambulators: 7
6-7 yrs
2
Hoffer: n Household: 1 2nd child could not be clearly classified into community or household 12-54 yrs Not stated Hoffer (baseline): n Community: 41 Household: 12 Nonfunctional: 7 Distance (baseline): n >1,000m: 35 100-1,000m: 10 10-100m: 4 <10m: 8 7.6 (3.2-11.4) 53 Results for PCI & PEDI broken down by muscle function score in Tables V & VI
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Comparison between Prospective Thoracic to sacral different cross-sectional classification systems depending on neurological level of impairment To compare Ferrari Case report Mid to low level KAFO's with lumbar AFO's
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Bartonek et al. MMC 1999a10
low & midlumbar Hoffer scale & self To compare outcomes Prospective selected walking with AFO & KAFO cross-sectional speed over 110m corridor
10.7 + 2.8 (513)
Bartonek et al. MMC 200515
Modified Hoffer scale
Sacral to midTo classify ambulatory Prospective status of participants cross-sectional lumbar
10.3 (6.8-17.6) 38
Hoffer: n Community: 38
Bartonek 201016
MMC
Modified Hoffer scale
Prospective Not stated Used to established an expected cross-sectional ambulation outcome based on muscle function class to guide orthotic program guidelines
Children 43 followed from birth until they reached 6 yrs
Walking achieved at 1year follow-up in 2/38 children, at 1.5-year follow-up in 7/39, at 2year follow-up in 14/36, at 3-year followup in 21/28, at 4-year follow-up in 28/36, at 6year follow-up in 30/38
Basobas et al. 200317
MMC, SCI, Ambulation myopathies, categorized as: CP community, household, nonambulatory SB FIM
To assess outcomes of Retrospective anterior spinal fusion cohort with anterior instrumentation surgery As an outcome Case report measure for a membrane based biofeedback machine
Not stated
10.42 (1.5818.75)
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Category: n Community: 9 Household: 6 Non-ambulators: 6
Not stated
12.5
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Mobility FIM=1 at baseline, after training score improved to 6
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Battibugli et al. MMC 200719
FMS (rates walking on 6point ordinal scale at 3 distances: 5, 50 & 500m)
To compare outcomes Retrospective in children with SB cohort who do & who do not have a shunt
Sacral: 118 Low lumbar: 31 Thoracic/high lumbar: 12
Participants 161 with no shunt: 9.92 + 3.92 (4.75-17.75) Participants with a shunt: 10.17 + 3.92 (4.83-18.08)
Benzer et al. 201220
Ambulation categorized as: Independent, assisted, nonambulatory
To evaluate influence Retrospective of ambulatory status cohort on renal functions, clinical & radiological findings
Thoracic: 3 Lumbar: 80
7.1 + 0.61
MMC
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Hoffer: n Community: 4 Household: 4
FMS: 5 meters: means of 5.05 (no shunt), 4.88 (with shunt) 50 meters: means of 4.97 (no shunt), 4.60 (with shunt) 500 meters: means of 4.84 (no shunt), 4.15 (with shunt) Category: n Independent: 8 Assisted: 14 Nonambulatory: 61
MMC
Buffart et al. 22 2008
MMC
Modified Hoffer scale
To examine Prospective relationship between cross-sectional ambulatory status & physical activity, aerobic fitness, obsesity
Thoracic: 2% 21.1 + 4.5 Thoracic-lumbar: 14% Lumbar: 29% Lumbosacral: 41% Sacral: 14% *** exact numbers not stated
Not Modified Hoffer: n stated; 51 Community: 15 total Household: 8 nonfunctional: 28
Carbonari de Faria et al. 23 2013
MMC
Hoffer scale
To compare Prospective neuromotor cohort development (including gait) between children who did & did not undergo intrauterine MMC repair
Thoracic: 3 Upper lumbar: 1 Lower lumbar: 5 Sacral: 4
13
Chang et al. 24 2008
MMC & LMMC
Modified Hoffer scale
3.84 (3.75 to 4.41)
Not WeeFIM's motor stated; 34 component not a total significant contribution to quality of Life ***specific WeeFIM results not reported
Hoffer: n Community: 6 Nonambulator: 1 Not all ambulation data were reported
Prospective Thoracic: 1 cross-sectional High lumbar: 3 Midlumbar: 9 Lumbosacral: 28 Sacral: 14 No neurological deficit: 23
15.24 + 6.74
Prospective T12 to L3-4 cross-sectional
RGOs: 26 n=15, 8.1 (3.715.0) HKAFO: n=11, 9.0 (6.111.8)
Speed (m/s): RGO: 0.27 + 0.11 HKAFO: 0.68 + 0.20
Follow-up walking assessment done at 2 years; exact mean age of group not reported
Category: n Community: 9 Household: 4 Nonfunctional: 16
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To describe clinical features & their correlates within children in Taiwan with spinal dysraphism as a clinical guide for future management Speed measured To compare energy during each efficiency in children minute of with MMC ambulating assessment of with either RGO or HKAFO energy consumption during walking Ambulation To determine the categorized as: prognostic value of community, neurophysiological household, investigations nonfunctional compared to clinical neurological exams in children with SB
13 + 6 (4- 27)
L1-L2: 3 L3-L4: 15 L5-Sacral: 10
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To identify factors related to quality of life
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WeeFIM
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Bier et al. 200521
Cuppen et al. 26 2013
SB
Danzer et al. 27 2009
MMC
Categorized as: independent walkers, assisted walkers (walk with appliances), nonambulatory (wheelchair bound)
To examine lower Retrospective High (T-L2): 10 extremity neuromotor cross-sectional Mid (L3-4): 24 function & Low (L5-S1): 20 ambulatory potential in infancy & early childhood of children who had midgestation MMC closure
5.58 + 1.52 (3- 54 9.42)
Category at follow-up: n Independent: 37 Assisted: 13 Nonambulatory: 4
Danzer et al. 201128
MMC
WeeFIM & Ambulation categorized as: independent, assisted, wheelchair dependent
To describe potential Prospective functional limitations cohort & to evaluate risk factors associated with impaired functional status in children that underwent fetal MMC closure
5 years of age, 26 but exact mean, SD & range not reported
WeeFIM (mobility domain): Functional independence in 62% Ambulation status: n Independent: 21 Assisted: 5
Anatomical level of spine: Thoracic: 7 Lumbar: 27 Sacral: 2 Motor impairment level: Thoracic: 12 Lumbar: 16 Sacral: 8
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Prospective cohort
Not Modified Hoffer: n stated; 78 Community: 56 total Household: 8 Nonambulators: 14 *** Results further broken down by MMC & LMMC in Table 1
Not stated
29
6MWT, speed (calculated during 6MWT), Hoffer scale
De Groot et al. SB 30 2009
De Groot et al. SB 31 2010
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To explore the Retrospective relationship between cross-sectional VO2peak & functional ambulation
10.4 + 3.1 (617)
23
6MWT, Modified 6MWT performed to Prospective Hoffer scale determine cohort individualized protocol for VO2max test
10.3 + 4.9
20
Speed (calculated To determine the during 6MWT) & reproducibility of Hoffer scale gross & net energy expenditure during gait in ambulatory children & adolescents with SB 6MWT & To evaluate the Prospective Not stated Modified Hoffer effects of treadmill Randomized scale training program controlled trial compared with usual care in ambulatory children & adolescents with SB
10.8 + 3.4
14
L5-S1: 10 S1-S4: 6 No motor loss: 7
AIS level: L3-L4: 2 L4-L5: 7 L5-S1: 6 S2 and below: 1 No motor loss: 4 Prospective AIS level: cross-sectional L3-L4: 1 L4-L5: 10 S1-S4: 1 No motor loss: 2
Control group: 32 11.1 + 2.6 Intervention group: 10.3 + 2.9
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MMC
6MWT & Modified Hoffer scale
To assess the Prospective reliability & psychometric agreement of study maximal & submaximal exercise measures in normal ambulatory & community ambulatory children & adolescents with SB
De Souza & 34 Caroll 1976
MMC
Hoffer scale
To identify factors that determine ambulatory status
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Categorized as: To determine long-distance influence of muscle ambulators, short- strength on distance ambulatory status ambulators, nonfunctional ambulators Speed (measured To compare energy during cost of walking assessment of between children with oxygen CP & children with SB consumption)
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Duffy et al. 199636
CP & SB
Evaggelinou & Drowatzky 199137
SB
Categorized as: ambulatory, household-only, nonambulatory
L3-L4: 2 L4-L5: 10 L5-S1: 6 S2 and below: 5
Prospective Thoracic: 4 cross-sectional Upper lumbar: 19 Lower lumbar: 13 Sacral: 32 Retrospective chart review
Not stated
Prospective L3-L4: 11 cross-sectional L5-S1: 10
To compare Prospective informationcross-sectional processing capabilities among children with differing walking abilities
T11: 1 T12-L1: 1 L1: 2 L1-L2: 2 L3: 5 L3-L4: 2 L4: 3 L4-L5: 1 L5: 2 Saccral: 3 Occulta: 1
Hoffer: n Normal: 17 Community: 6 6MWT: mean distance + 1SD for group = 391.4 + 61 m 6MWT: mean + 1SD distance walked for group = 418 + 95 m
Hoffer: n Normal: 8 Community: 6 Speed: Group mean + 1SD = 70.0 + 13.8 m/min
RI PT
De Groot et al. SB 200829
10.7 + 3.5
23
Hoffer: n Normal: 9 Community: 23 Baseline 6MWT distance: Control group: 372.1 + 116.5 m Intervention group: 344.8 + 125.3 m Hoffer: n Normal: 10 Community: 13 6MWT: Mean + 1SD at baseline for group = 406.8 + 90.7 m
Not reported, Not Hoffer: n recruited ages stated; 68 Community: 23 12 & up total Household: 15 Nonfunctional: 23 Nonambulators: 7 14.33 (6Not Category: n 27.75) stated; 83 long-distance: 25 total short-distance: 26 nonfunctional: 32
L3/L4: 8.5 (5- 21 12) L5/S1: 8.9 (512)
Speed: group means L3/L4: 40.7 m/min L5/S1: 51.7 m/min
8-18
Category: n Ambulatory: 8 Household-only: 6 Nonambulatory: 7
23
ACCEPTED MANUSCRIPT Not stated
WeeFIM
To determine whether Prospective longer waiting times cohort for rehabilitation were associated with change in child functional status
Findley et al. 198739
MMC
Hoffer scale
To identify walking ability in adolescents with SB
Findley & Agre MMC 40 1988
Self selected & maximal speed (measured over 30m distance)
To determine whether Prospective energy cost of cohort mobility, relative level of effort during mobility, & speed at which one exerted 70% of maximal aerobic power could be estimated from measures easily obtained in the clinic
Franks et al. 199141
MMC
PCI
GerritsmaBleeker et al. 199742
MMC & paraplegia
Thoracic: 14 Upper lumbar: 4 Lower lumbar: 16 Sacral: 19 No weakness: 10
13 (11-16)
77
WeeFIM Severity Category: Mild to none (total score >75): 64 Moderate (total score 50-75): 52 Severe (total score <50): 8 Hoffer: n Community (never use wheelchair): 30 Community (occasionally use wheelchair): 26 Household: 0 Therapeutic: 1 Wheelchair only: 20 Self selected speed reported by impairment level (mean+1SD m/min): L2 & above: 32 (n=1) L3-L4: 58 + 10 L5-sacral: 65 + 5 No motor deficit: 80 + 12
Neurological level 10-15 yrs defined by lowest root level with grade 3 strength on MMT L2 & above: 1 L3-L4: 5 L5-sacral: 16 No motor deficit: 3
25
M AN U
Inclusion criteria for study was measured PCI was greater than 1.00 Hoffer scale while To review walking wearing a RGO status in children who have used a RGO
Prospective case series
L4: 2 L5: 1
15, 10, 9
3
PCI values = 1.46, 2.15, 2.06
Retrospective chart review
L1: 6 L2: 1 L3: 6
4.8 + 2.0
13
Categorized as: Community, independent indoors, nonfunctional
Retrospective cohort
Not stated
11 (4-22) for 6 entire group (age of children with MMC not reported) 4.3 (1-10.2) at 5 first visit; 20 (16.1-24.7) at follow-up
Hoffer: n Community: 3 Household: 8 Nonfunctional: 2 Category: n Independ. indoors: 3 Nonfunctional: 3
Lumbosacral Hoffer's scale agenesis &/or MMC
To assess type & Retrospective extent of spinal review of charts malformation, degree & radiographs of lower extremity involvement & ambulatory capacity over time, in order to predict ambulatory potential & develop guidelines for orthopedic management
motor level: "flail lower extremity": 2 L2: 2 L3: 1 L4: 1
Hassan et al. 201045
hemophilia, juvenile idiopathic arthritis, MMC & LMMC
6MWT
To compare 6MWT Prospective values of pediatric cohort patient populations to reference values, & assess differences in predicted distances
L5 and below: 22
10.3 + 3.1 (618)
22
6MWT for group: 391 + 61 m
Hisaba et al. 201246
MMC
Hoffer's scale
To evaluate outcomes Retrospective of intrauterine cohort surgery
Anatomical levels: T12-L1: 1 L1-L2: 1 L2-L3: 2 L4-L5: 2 Functioal levels: L1-L2: 1 L4-L5: 3 S1-S2: 2
Followed prenatally to 3.5 yrs
6
Hoffer: n Community: 3 Household: 2 Nonambulator: 1
AC C
EP
Guille et al. 44 2002
To assess effects of surgery to correct knee & hip flexion deformity
TE D
Grujic & Aparisi MMC & CP 43 1982
Prospective cohort
3.77 + 1.11 (2- 124 9)
RI PT
SB
SC
Feldman et al. 200838
Hoffer: n (lumbosacral agenesis & MMC): Nonambulator: 5
To study factors associated with walking ability
MMC
Walking categorized as: independently, with crutches & orthoses, with crutches without orthoses, wheelchair only
To evaluate long-term Retrospective clinical effects after cohort having performed a Grice arthrodesis of a valgus unstable foot
MMC
Categorized as: good (can walk >20 yards with or without aids), poor (walking only a few steps or having some function in legs), no mobility (no useful walking)
To analyze severity of Prospective disability relative to cohort numerour factors (e.g., sensory level, presence or absence of neural plaque, motor activity of the legs)
Ilharreborde et DMD, CP, SB, polio-myelitic al. 200950 or traumatic paraplegia, SMD, scoliosis
Categorized as: walkers, walk with crutches, nonwalkers
To examine the effect Retrospective Not stated of long spinal fusion review of charts on functional status & radiographs
Jaworek et al. 201351
MMC
To evaluate the mobility & quality of life after surgical treatment
Kaiser & Rudeberg 52 1986
MMC
Categorized as: community ambulation, ambulation with crutches, ambulation with wheelchair, nonambulation Ambulation categorized as: Without aids/with short irons, With calipers, Wheelchair
12.8 + 3.9 (517)
34
SCI & MMC
Hoffer scale & functional tests (free-walking 24.4 m on a level surface, descending 20 steps, ascending 20 steps) Speed & PCI
Hoffer: n Community: 10 Household: 2 Nonfunctional: 2 Non-ambulators: 20
Category: n Independently: 16 (7 of whom used orthoses) With crutches & orthoses: 3 With crutches without orthoses: 1 Wheelchair only: 3
RI PT
At surgery: 6.6 23 ± 1.8 Follow-up: 19.4 ± 3.8
High sensory level 1.25-7.67 (T5-T10): 33 Intermediate (T11L3): 28 Low (L4 and below): 18
Age at operation: 15.3 ± 2.6
Prospective High hernia (T6-L1): 9.8 (2-16) cross-sectional 11 Low hernia (L2-L5): 8
To compare outcomes Prospective between children who cohort did & did not meet Lorber's criteria for surgery
AC C
Karmel-Ross et SB al. 199253
Katz et al. 199754
of lesion based on preserved sensorimotor function: Thoracic: 8 Upper lumbar: 11 Lower lumbar: 11 Sacral: 4 Lumbosacral
80
Category: n Good: 30 Poor: 33 No mobility: 16
SC
Hunt et al. 49 1973
chart review, longitudinal cohort
M AN U
Høiness & Kirkhus 200948
ACCEPTED MANUSCRIPT Retrospective Neurological level
Hoffer scale (original article)
TE D
MMC
Not stated
Not stated
Not Preop category: n stated; 56 Walkers: 11 total Walk with crutches: 6 Nonwalkers: 39
19
Category: n Community ambulation: 1 Ambulation with crutches & ambulation with wheelchair: 17 Nonambulation: 1
34
Category: n Without aids/with short irons: 13 With calipers: 12 Wheelchair: 4 Too young for assessment: 5 Hoffer: n Community: 4 Results for functional tests not reported
EP
Hoffer et al. 197347
To describe the sample & to examine the functional carryover of electical stimulation.
Prospective, L2-3 cohort, single group pretest posttest design
ages in yrs (n): 4 5(2), 12(2), 21(1)
To evaluate & compare metabolic cost of walking with RGO and HKAFO
Prospective cohort
6.83 (2.1711.75)
Functional level: Thoracaic or thoracolumbar: 4 High lumbar: 4
8
Mean speed (m/min): RGO: 14.6 HKAFO: 11.9
SB, UMN pathology, nemaline myopathy
ACCEPTED MANUSCRIPT Prospective, L3-4
Functional tests To compare 2 types of (timed): KAFOs (PM vs LM) ascending & descending 5 stairs, ascending & descending a ramp, rising from the floor & ambulating 20m
cohort, longitudingal quasiexperimental cross-over design
FAC, WeeFIM, Speed
To describe the sample with respect to their virtual rehabilitation game performance
Prospective not stated cross-sectional
Lemelle et al. 57 2006
MC, MMC
Categorized as: wheelchair-bound (inside & outside), wheelchair-bound (outside), stand & walk with walking brace, walk with minor aid
To examine Prospective Not stated relationship between cross-sectional walking ability, incontinence, & HRQOL
Lewis et al. 58 2004
MMC
Ambulation categorized as: independent, with assistance, wheelchair-bound
To compare longterm Retrospective motor outcomes & cohort ambutatory status at ages 2 & 10 yrs between children born by elective cesarean section or vaginal delivery (labour)
Lorente Molto & Garrido 59 2005
MMC
Hoffer scale
To evaluate the Retrospective relationship between cohort hip stability & walking ability during early childhood, adolescence & adulthood
Not Stairs (s): stated; 12 PM: 30.0 + 13.4; total LM: 34: + 14.5 Ramp (s): PM: 36.2 + 40.5; LM: 30.9: + 34.7 From floor (s): PM: 17.8 + 25.5; LM: 21.5 + 36.4
13.4 + 3.6 (5.5- 1 19.0)
FAC level: n 5: 11 4: 6 3: 1 1: 1 WeeFIM: mean 89 + 8 Speed: mean 1.6 + 0.3 km/hour 124/160 were able to walk; specific numbers not included
14.4 + 1.84
160
EP
TE D
M AN U
SC
Labruyere et al. MMC, CP, CVA,TBI, 201356 spastic ataxia, CNS demyelination
Whole group at baseline: 9.7 + 2.7 (521)
RI PT
Krebs et al. 198855
Anatomical level: Cesarean group: L2 ± 2.2 Labour group: L3 ± 1.4 Motor level: Cesarean group: L2 ± 2.0 Labour group: L3 ± 1.2 L3: 29
data collected 87 from participants' files at ages 2 & 10
Category at age 2: n Independent: 15 With assistance: 56 Wheelchair-bound: 16 Category at age 10: n Independent: 11 With assistance: 36 Wheelchair-bound: 40
Age at time of 29 surgery: 2.21 (0.92-6) Records reviewed at 25 yrs & 13-18 yrs; interviewed as adults
Hoffer: n At 2-5 yrs: Community: 21 Household: 4 Non-functional: 4 Non-ambulation: 0 At 13-18 yrs: Community: 19 Household: 3 Non-functional: 3 Non-ambulation: 4 Self-selected speed (m/min): Swivel mean: 7.23 + 0.73 Parapodium mean: 9.18 + 1.88 Maximal speed: Swivel mean: 14.78 + 2.06 Parapodium mean: 21.63 + 3.70 Hoffer at baseline: nonfunctional; Hoffer with orthosis: community
Self selected & maximal speed
To compare Prospective ambulation between cross-sectional Parapodium & ORLAU swivel modification
T10: 2 T11: 1 T12: 2 L1: 2 L2: 2
6.1 + 1.31 (49)
9
Magee & Kenny Myelodysplasia 200861
Hoffer scale
To describe efect of Strutter Orthosis on mobility
L3-L4: 1
12 years
1
AC C
MMC & Lough & Nielsen 198660 traumatic paraplegia
Prospective case report
ACCEPTED MANUSCRIPT Motor level
113
Categorgy: n Full walkers: 61 Partial walkers: 13 Nonambulatory: 36 Below age of walking: 3
McCall & MMC, CP, Hoffer & Maximal Follow-up measure of Prospective Thoracic: 20 63 cross-sectional Upper lumbar: 13 Schmidt 1986 osteo-genesis spped (over 30m) children who were imperfecta, prescribed an RGO Lower lumbar: 7 arthrogryposis, paraplegia, polio, SMA, microcephaly
1.25-16
41
Hoffer: n Community: 24 Household: 8 Therapeutic: 5 Nonambulators: 4 Max speed with RGO: group mean=0.52 + 0.22m/s
McDonald et al. MMC 64 1991
Categorized as: nonabmulatory, partial, community (according to Shurtleff et al. 1989)
Not stated
Moerchen et al. SB 65 2011
FMS & PEDI
Moore et al. 200166
MMC
Speed
Müller et al. 1992a67
MMC
Modified Hoffer scale
Müller et al. 68 1992b
MMC
Modified Hoffer To assess the Prospective scale (see Table 1 outcomes of scoliosis cohort in the paper for surgery customized scoring)
Müller et al. 199469
MMC
SC
Prospective Not stated cross-sectional
M AN U
To determine the degree to which specific muscles influence mobility, & to document the natual history of mobility amongst those with different patterns of strength To evaluate a homebased treadmill intervention To compare the efficiency of reciproccal gait or swing-through gait
(International Myelodysplasia Study Group criteria): Thoracic: 6 Upper lumbar: 22 Lower lumbar: 42 Sacral: 43
Prospective case report
L4-L5: 1
Prospective L3-4: 13 cross-sectional
To investigate the use Prospective of Boston bracing to cohort treat scoliosis in MMC
AC C
MüllerMMC Godeffroy et al. 200870
RI PT
10.67 + 4.83 (0.5-18)
To examine the Retrospective relationship between cross-sectional ambulatory status & incidence of fractures
TE D
Modified version of International Myelodysplasia Study Criteria Manual ambulation categorized as: nonambulatory, partial, full, below age of walking
Not Category: n stated; Community: 139 190 total Partial: 30 Nonambulators: 61
1.5
1
MMC group: 17.1 (12-24) Control: 14.4 (12-23)
14
Thoracic: 1 10.2 + 3.1 (5- 20 Upper lumbar: 8 14) Lower lumbar (L4 & below): 11
Not stated
EP
Marreiros et al. SB 201062
Hoffer scale
To understand the Retrospective Not stated natural history of cross-sectional scoliosis in individuals with MMC
Categorized as: Community, household/ near environment, wheelchair user
To identify factors associated with HRQOL
Surgery was 14 performed at a mean age of 12.4 (9- 16) yrs, at time of follow-up mean age of the children was 15.6 (1 020) yrs 15.7 (6-27)
Prospective Thoracic (T10- L2): 9 12.08 + 2.33 cross-sectional Lumbar (L3-4): 11 (8-16) Sacral (L5-S2): 25
Mobility component of PEDI: 4/25 FMS: 0 Speed(m/min): Reciprocal gait: 40.49 + 14.21 Swing-through gait: 53.95 + 12.81 Modified Hoffer at baseline: n Community: 0 Household: 4 Nonfunctional: 14 Nonambulators: 2 Mean Modified Hoffer score: preop = 2 (nonfunctional), postop = 1 (nonambulator)
Not Incomplete data stated; 64 presented (Hoffer data total reported for n=16 in Table 4) 45
Community: 21 Household/near environment: 17 Wheelchair user: 7
ACCEPTED MANUSCRIPT Prospective Thoracic: 6
Categorized as: level A (fully independent & walking with no support); level B (independent but requiring support); level C (dependent but upright & standing in parallel bars or walking with a "drag through" gait); level D (confined to wheelchair) PEDI & Hoffer scale
To utilize muscle power at birth to predict ambulatory status later in childhood
3-8
95
Thoracolumbar: 13 Lumbar: 36 Lumbo-sacral: 29 Sacral: 11
OkurowskaZawada et al. 201173
MMC & CP
Hoffer scale
To describe the motor Prospective Level was lowest 10.85 + 3.75 (5- 34 function of cross-sectional level on better side 16) participants at which child could perform antigravity movement through available range: Thoracic: 13 Lumbar:17 Sacral: 4
Ozaras et al. 200574
SB aperta, MMC
Categorized as: non-ambulatory, therapeutic ambulation, household ambulation, functional ambulator
Ambulation was one Prospective component of cohort assessment to identify children with SB who showed signs of CP
Upper thoracic: 11 4.99 + 3.44 Lower thoracic: 6 Range not included Upper lumbar: 5 Lower lumbar: 6
28
Hoffer scale
To identify factors that could predict future mobility
Thoracic: 5 Thoracolumbar: 9 Lumbar: 43 Lumbosacral: 23 Sacral: 10
10.6 + 6.25 (1.4 to 26.7)
Not Hoffer: % sample stated; 90 Community: 42% total Household: 16% Exercise: 16% Wheelchair dependent (nonwalker): 27%
To assess Prospective predictability of post- cohort natal function using an in utero reflex technique in fetuses
Low sacral lesions
Ambulation 8 assessed at 35 yrs
Walk with no or min assistance: 3 Walk with assistance: 1 Unable to walk: 4
To describe ambulation in children who underwent triple or double muscle transfers
Level based on voluntary control of knee/ankle & sensation: L3: 7 L4: 28 L5: 12 T10: 3 T12: 8 L1: 3 L3: 1 L4: 6
Age at follow- Not up: stated; 47 9.75 (4.67total 20.25)
Follow-up data reported for 41/47 (baseline data not reported): Community: 37 Household: 4
7 (1.67-14.5)
Hoffer while using orthosis: n Community: 13 Household: 8
Phillips & MMC 77 Lindseth 1992
Phillips et al. 78 1995
M AN U
TE D
EP
Retrospective cohort
AC C
SB
MMC
Categorized as: walk with no or min assistance, walk with assistance (walker), unable to walk Hoffer scale
Hoffer scale
8.7 (6-11)
32
SC
MMC
Petrikovsky et al. 200176
Prospective Not stated cross-sectional
Level A: 22 Level B: 48 Level C: 20 Level D: 5
Norrlin et al. 72 2003
Pauly & Cremer MMC 75 2013
As an outcome of neurological impairment
cohort
RI PT
Murdoch et al. MMC & myelocele 197971
Retrospective cohort
To describe Retrospective ambulatory status of cohort children with RGO or hip guidance orthosis
21
Hoffer: n Community: 11 Household: 6 Nonfunctional: 9 Nonambulator: 6 PEDI (caregiver assistance, group mean): 76.7 + 18.3 Hoffer: n Community: 3 Household/near environment: 10 Wheelchair user: 21
Therapeutic: 8 Nonambulatory: 20
CP, MMC, HSP
Self-selected To compare measure speed (10MWT) & of speed obtained 10-min WT with 10MWT & 10min WT (Part 1), & to assess repeatability of 10-min WT (Part 2)
Rose et al. 80 1981
SB
Modified Hoffer scale, speed measured over 20 feet distance
Ross et al. 81 2007
LMMC, MC, Hoffer scale intradural lipoma, filum terminale lipoma, split cord malformation, dermal sinus, caudal regression syndrome
To describe Retrospective ambulatory status in a cross-sectional large group of children with closed NTD
Samuelsson & 82 Skoog 1988
MMC
To classifiy participants by ambulatory ability
SC
0.9-22.9; Not Hoffer: n mean & 1SD stated; Community: 98 age reported 104 total Household: 1 for each type Nonambulators: 5 of NTD in Table 4
Prospective cohort
Hoffer scale, step To evaluate effects of Prospective distance/ length, orthotic treatment cohort maximum nonstop walking performance & speed as measured on a scale of 1 to 5
thoracic: 35 L1-L2: 5 L3: 26 L4: 32 L5: 20 Sacral: 45 Not stated
14.83 (2-40)
Not Hoffer: n stated; Community: 63 163 total Household: 21 Nonambulators: 51
5.17 (1.1732.92)
Not Results are reported stated; 67 only as % of total participants in each bracing group. E.g., 60% of patients with Ferrari type orthosis walked for >50 minutes & with stepping distances >40cm were seen 25 with Hoffer: n MMC or Normal: 13 LMMC (44 Community: 1 total) Household: 1 Wheelchair dependent: 4 Too young: 5 44 Modified Hoffer: n MMC: Normal: 14 Community: 4 Household: 2 Nonambulant: 1 Too young: 9 LMMC: Normal: 9 Community: 4 Household: 1
AC C
EP
Schiltenwolf et SB 83 al. 1991
T9: 1 T10: 3 T11: 5 T11/12: 1 T12: 6 T12/L1: 1 L1: 1 L1/L2: 2 L2: 3 L2/L3: 1 L3: 1 L5: 2 Not stated
Part 1: 8 Whole group results for Part 2: 11 speed (m/s): Part 1: 10MWT: 1.00 + 0.3 (0.15-1.61) 10-min WT: 0.87 + 0.24 (0.30-1.34) Part 2: 10-min WT #1: 0.97 + 0.26 (0.16-1.31) 10-min WT #2: 0.98 + 0.25 (0.17-1.33) 9.7 + 2.7 (5.67- 27 Baseline data 15.58) Hoffer: n Community: 2 Household: 6 Therapeutic: 18 Chairbound: 3 Speed (ft/min): 26.0 + 15.2
Part 1: 12.4 + 3.9 (4-28) Part 2: 11.5 + 3.5 (6-16)
M AN U
To determine whether Prospective the hip guidance cohort orthosis improved ambulatory status
TE D
Hoffer scale
cohort & prospective cohort
RI PT
ACCEPTED MANUSCRIPT Retrospective Not stated
Pirpiris et al. 200379
Schoenmakers et al. 200384
Tethered Cord Syndrome (MMC & LMMC)
Modified Hoffer scale
As an outcome Prospective measure for the Cohort Study effectiveness of tethered cord release surgery
High level (above L4): 5 Low lumbar: 6 Sacral: 2
Age at operation: 6.17 + 5 Long term followup: 13.58 + 5.5
Schoenmakers et al. 200485
MMC & LMMC
Modified Hoffer scale, MABC & Dutch Version of PEDI
To investigate Prospective functional outcome in cross-sectional children with sacral level MMC and LMMC
S1 or Below: all participants MMC: 30 LMMC:14
MMC: 6.0 + 4.9 (0-18) LMMC: 8.4 + 4.9 (0-18)
Modified Hoffer scale, Dutch Version of PEDI
Schoenmakers et al. 200988
Lumbosacral 6MWT & MMC & Modified Hoffer LMMC scale
Seitzberg et al. MMC 200889
MMC
Stallard et al. 91 1991
MMC
Stillwell et al. 92 1984
SB
Hoffer scale
Suson et al. 201093
SB occulta, MMC, LMMC & sacral agenesis
Swank & Dias 199294
MMC
Categorized as: ambulates fully, ambulates with devices, ambulates minimally with devices, wheelchair bound Hoffer scale
Swank & Dias 199495
SB
To predict adult Retrospective ambulation status Cohort Study based on ambulation status between ages of 5-8 yrs
To compare the effect Prospective of surgical versus non cross-sectional surgical treatment of hip dislocation
10
23
Modified Hoffer (in children >2.5yrs): n Nonambulators: 53 Ambulators: 50 Modified Hoffer: n Presurgery: Nonambulatory: 6 Exercise: 3 Household: 1
Modified Hoffer: n Normal: 10 (MMC), 7 (LMMC) Community: 6 (MMC) 6MWT means (m): 353 + 108 (MMC), 424 + 65 (LMMC)
At age 5-8 yrs: At or above L2: 1 L3: 7 L4: 8 L5: 8 At or below S1: 8
5.0-8.0
52
L3: 22 L4: 6 L5:1 S1:1
Non41 operative: 10 (4-19) Operative: 6 (216) SD not included
Category: n Wheelchair user: 14 Community ambulator with walking aid: 11 Community ambulator without walking aid: 27
Category: n Household: 1 Community (unable to use stairs): 6 Community (able to use stairs): 23
Hoffer: n Community: 18 Household: 22 Therapeutic: 12 Not Hoffer: n stated; 47 Community: 32 total Household: 3 Nonambulatory: 12
To identify ambulatory abiliy of patients with omphaloceleexstrophyimperforate anusspinal defects
Not Category: n stated; 68 Ambulated fully: 37 total Ambulated with devices: 15 Ambulated minimally with devices: 2 Wheelchair bound: 8
AC C Modified Hoffer scale
122
To measure Prospective Complete lesions L1- Up to 15 yrs performance with the cross-sectional T4 (specific numbers not ParaWalker orthosis reported) To determine the Prospective L2: 3 (10->25) number of individuals cohort L3: 16 who were walking 10 L4: 15 yers after iliopsoas L5: 13 transplantation
EP
Sherk et al. 90 1991
Categorized as: wheelchair user, community ambulator with walking aid, community ambulator without walking aid Ambulation categorized as: exercise, household, community (unable to use stairs), community (able to use stairs) Hoffer scale
7.9 + 5.2 (1cross-sectional L1-3: 26 18) L4-5: 45 Sacral: 26 To evaluate the Prospective T11: 3 9.3 + 2.4 effectiveness of spinal cross-sectional T12: 2 fusion surgery L2: 1 L3: 1 L3-4: 1 L5: 1 S1: 1 To identify Prospective AIS level: 6-18 associations with cross-sectional MMC: muscle strength, L5-S1: 7 aerobic capacity & S1-S4: 5 physical activity in No Motor Deficit: 4 independent, LMMC: ambulatory children L5-S1: 3 with lumbosacral SB S1-S4: 1 No Motor Deficit: 3
RI PT
Shoenmakers et MMC 87 al. 2005b
ACCEPTED MANUSCRIPT Prospective Thoracic: 26
To identify determinants of HRQOL
SC
Modified Hoffer scale, Dutch version of PEDI
M AN U
MMC
TE D
Schoenmakers et al. 2005a86
Retrospective Not stated cross-sectional
17 (0.5-34)
52
To identify prognostic Retrospective indicators of future review walking ability
Thoracic: 71 Lumbar: 73 Sacral: 62
5.11 (0.08-13) 206
To develop a model that would predict independence in walking
Thoracic (L3 or above): 60 Lumbar (L3-5): 81 Sacral: 63
Followed from 206 birth to a mean age of 9.58
Prospective cohort
Hoffer: n Community: 118 Household: 41 Nonambulators: 57 Hoffer: n Community: 118 Household: 36 Nonambulatory: 25
ACCEPTED MANUSCRIPT Thoracic: 1
Tezcan & Simsek 201396
CP & SB
Ambulatory classification according to Flanagan 2011
To investigate the Prospective relationship between cross-sectional HRQL & numerous factors including level of ambulation
9.25 + 3.82 (5- 70 Thoraco-lumbar: 16 18) Lumbar: 47 Lumbo-sacral: 5 Sacral: 1
Thomas et al. 97 2001
MMC
Classification by Schopler & Menelaus 1987, speed
To identify differences Prospective in walking speed & Cohort oxygen cost between walking with RGOs vs HKAFOs
T12: 4 L1: 4 L2: 3 L3: 10 L3-4: 2
Video analysis & questionnaire to assign score on ordinal scale: 1=primarily nonambulatory; 2=primarily ambulatory, uses wheelchair for distances >100 feet; 3=primarily ambulatory, does not use wheelchair in community; 4=always ambulatory, no diagnosis
To examine the Prospective Not stated relationship between cross-sectional lower extremity bone mineral density & level of ambulation
Categorized as: independent, partial (walking with crutches or walkers), nonambulatory PEDI & ambulation categorized as: independent, With walking aid, Nonambulant
To correlate ambulatory status with EMG findings
Tsai et al. 100 2002
MMC & LMMC
van den BergEmons et al. 2001101
MMC
Hoffer scale
Verhoef et al. 102 2004
SB
Modified Hoffer scale
RI PT
SC
6-13
M AN U
MMC & LMMC
Prospective Thoracic: 1 cross-sectional L1-2: 1 L3: 1 L4-S1: 19 S2-4: 12 No deficit: 13 To investigate Prospective No deficit: 15 functional cross-sectional Thoracic: 1 performance, & to L1-2: 3 correlate ambulatory L3: 2 status with PEDI score L4-S1: 24 S2-4: 18
4.4 (0.5-12)
47
Category: n Independent: 36 Partial: 2 Nonambulatory: 9
MMC = 4.60 + Not 3.18 (.58-12) stated; 63 LMMC= 3.81 + total 2.97 (.5- 11)
Mobility domain of PEDI: Norm standard scores MMC: 25.8 + 19.5 LMMC: 49.1 + 14.1 Scaled scores MMC: 57.6 + 28.3 LMMC: 72.7 + 29.5 MMC ambulatory status: n Independent: 6 With aid: 1 Non-ambulatory: 3 LMMC ambulatory status: n Independent: 30 Non-ambulatory: 2 To measure extent of Prospective Thoracic/lumbar: 2 18 + 4 (14-26) Not Hoffer: n hypoactivity in cross-sectional Lumbar: 3 stated; 14 Community: 3 adolescents & young Lumbar/sacral: 8 total Household: 6 adults with MMC with Sacral: 1 Nonambulators: 5 an activity monitor
AC C
EP
Tsai et al. 99 2001
TE D
Thompson et al. SB, CP, muscular 200098 dystrophy, spinal cancer, TD children
RGOs: 8.0 (4-15) HKAFO: 8.17 (5-13)
Independent ambulation with no assistive devices: 3 Independent ambulation with assistive device: 27 Ambulating with assistive device at home & wheelchair in community: 23 Wheelchair bound: 17 23 Category: n RGO: Community: 7 Household: 7 HKAFO: Community: 9 Speed: data over time reported in Table 2 9 with SB Category: n Level 1: 5 Level 2: 4 Level 3: 7 Level 4: all TD children
L2 & above: 73 To describe secondary Prospective health conditions in a cross-sectional L3-5: 68 Dutch adolescent S1 & below: 38 group with SB aperta & occulta
20. 75 + 2.92 (16-26)
Not Hoffer: n stated; Normal: 64 179 total Community: 28 Household: 17 Nonfunctional: 9 Nonambulatory: 61
PAS (ordinal scale), WeeFIM
As an outcome measure for treatment with nighttime electrical stimulation
Dutch version of SB, neuroWassenbergthe PEDI Severijnen et al. metabolic 105 disorders, 2003 osteo-genesis imperfecta, encephalopathy Williams et al. 106 1983
Myelodisplasia
Self selected & fast speeds over a level, 60.5m oval track
Williams et al. 107 2005
CP, SB
TUG
Winchester et al. 2002108
CP, ABI, SB, GMFM & speed DS & autism (10MWT)
Yngve et al. 109 1984
MMC, muscular dystrophy, SCI, CP, MS, spinal cord embolism or arterovenous malformation, LMMC
To establish the Prospective reliability of the Dutch psychometric version of the PEDI study
To document energy requirements for ambulation & wheelchair propulsion, & to compare these values with those of TD children To investigate the reliability & validity of the TUG in children with and without disabilities To evaluate a hippotherapy program
12.5 + 2.7 (716)
Thoracolumbar: 2 7 (4-12) Low lumbar/lumbosacral: 5
Not stated
Prospective Thoracic: 1 cross-sectional Lumbar: 11 Sacral: 3
80
7
Hoffer: n Community: 21 Household: 5 Therapeutic: 8 Nonambulators: 46 ASK (mean + 1SD): 62.4 + 25.6 All participants walked; No specific results reported for PAS or WeeFIM, only p-values
3.5 + 1.8 (0.58- 7 (53 7.33) total)
Mean scores on mobility domain of PEDI ranged 33.1 - 36.6
(5.1-12.7)
Speed (m/min): Self selected: 40.8 + 12.5 Fast: 69.4 + 16.6
15
Prospective Low level lesions, cross-sectional specific levels not stated
10.10 + 4.10 (5- 8 19)
Prospective Not stated cross-sectional
6.36
To evaluate the Prospective Thoracic: 3 effectiveness of a RGO cross-sectional L1: 1 L3: 3 L4: 6 L5: 1 L5-S1: 1 Sacral: 1
EP
Speed Ambulation categorized as: community, household, exercise, nonambulator
Prospective cohort
L1-3: 13 L4-5: 15 Sacral: 13
RI PT
MMC
To determine the Prospective relationship between cross-sectional spinal deformity & overall physical function & self perception
SC
Walker et al. 104 2011
ACCEPTED MANUSCRIPT T12 or higher: 39
Hoffer scale & ASK
M AN U
SB
TE D
Wai et al. 2005103
1.5-15
TUG (group with SB): 8 + 1.5 s
1 with SB GMFM: 42/42 (7 total) Speed (m/min): Pre-test: 16.6 Post-test 1: 17.9 Post-test 2: 28.8 16 Community: 4 Household: 4 Exercise: 5 Nonambulator: 2
AC C
Abbreviations: SD=standard deviation; SB=spina bifida; MMC=myelomeningocele; FIM=Functional Independence Measure; LMMC=lipomyelomeningocele; NEM=Necker-Enfants Malades; SCI=spinal cord injury; RGO=reciprocating gait orthosis; KAFO=knee-ankle-foot orthosis; AFO=ankle-foot orthosis; PEDI=Pediatric Evaluation of Disability Inventory; PCI=Physiological Cost Index; CP=cerebral palsy; FMS=Functional Mobility Scale; HKAFO=hip-knee-ankle-foot orthosis; 6MWT=6-minute walk test; AIS=American Spinal Injury Association Impairment Scale; MMT=manual muscle test; DMD=Duchenne Muscular Dystrophy; SMD=spinal muscular dystrophy; UMN=upper motor neuron; PM=plastic/metal; LM=leather/metal; CVA=cerebral vascular accident; TBI=traumatic brain injury; CNS=central nervous system; FAC=Functional Abmulation Categories; MC=meningocele; HRQOL=health-related quality of life; SMA=spinal muscular atrophy; HSP=hereditary spastic paresis; 10MWT=10-meter walk test; 10-minWT=10-minute walk test; NTD=neural tube defects; MABC=Movement Assessment Battery for Children; TD=typically-developing; EMG=electromyographic; ASK=Activities Scale for Kids; PAS=Progressive Ambulation Scale; TUG=Timed Up & Go test; ABI=acquired brain injury; DS=Downs syndrome; GMFM=Gross Motor Function Measure; MS=Multiple Sclerosis.
Appendix 3: Quality Assessment of Included Studies
ACCEPTED MANUSCRIPT
Interpretation: Participant characteristics : Adequate = All details provided; Partial = All details except gender; Inadequate = missing details. Sampling method : Convenience; Community-based; Population-based; Not stated. Inclusion/exclusion criteria : Stated = clear list of both provided; Limited = 1 or 2 points only; Not stated. Data Collection: Prospective = objective exam; Retrospective = chart review; Not stated.
Participant characteristics defined?
Sampling method Inclusion/ exclusion criteria stated?
Prospective or retrospective data collection?
Reliability of Validity of Interpretability or measure stated or measure stated or responsiveness of tested? tested? measure stated/tested?
Adequate
Convenience
Stated
Prospective
No
Agre et al. 1987
Adequate
Not stated
Not stated
Prospective
No
Al-Holou et al. 3 2009
Adequate
Convenience
Stated
Prospective
No
Alman et al. 1996
Inadequate
Convenience
Limited
Prospective
No
Ammerman et al. 5 1998
Adequate
Convenience
Not stated
Prospective
No
Apkon et al. 2009
Adequate
Convenience
Stated
Prospective
7
Adequate
Convenience
Limited
Prospective
8
Banta et al. 1991
Adequate
Convenience
Not stated
Bartnicki et al. 20129
Adequate
Not stated
Stated
Bartonek et al. 10 1999a
Adequate
Convenience
Stated
Bartonek et al. 11 1999b
Adequate
Not stated
Not stated
Bartonek et al. 12 1999c
Adequate
Convenience
Limited
Bartonek & Saraste Inadequate 13 2001
Convenience
Bartonek et al. 14 2002
Inadequate
Convenience
Bartonek et al. 200515
Inadequate
Convenience
Bartonek 201016
Inadequate
Convenience
Basobas et al. 200317
Inadequate
Convenience
Batavia et al. 200118
Inadequate
Convenience
6
Asher et al. 1983
No
Yes - speed expressed as % of normal
No
No
No
No
No
No
No
No
No
No
No
No
RI PT
No
SC
4
No
M AN U
2
Prospective
No
No
No
Prospective
No
No
No
Prospective
No
No
No
Prospective
No
No
No
Prospective
No
No
No
Limited
Prospective
No
No
No
Not stated
Prospective
No
No
No
Limited
Prospective
No
No
No
Limited
Prospective
No
No
No
Limited
Retrospective
No
No
No
Not stated
Prospective
No
No
No
No
TE D
1
Adzick et al. 2011
EP
Author & Year
Convenience
Stated
Retrospective
Benzer et al. 201220 Adequate
Convenience
Not stated
Retrospective
No
No
No
Bier et al. 200521
Adequate
Convenience
Not stated
Prospective
No
No
No
Adequate
Convenience
Stated
Prospective
No
No
No
Carbonari de Faria Partial et al. 201323
Convenience
Stated
Not stated
No
No
No
Chang et al. 200824 Adequate
Convenience
Limited
Prospective
No
No
No
Battibugli et al. 200719
Buffart et al. 200822
AC C Adequate
Yes - FIM: MRC= 0.92-0.95 (interrater, test-retest, equivalence) No
Cuddeford et al. 25 1997
Partial
Not stated
Not stated
Prospective
No
No
Yes - speed compared to normal value
Cuppen et al. 26 2013
Inadequate
Convenience
Stated
Prospective
No
No
No
ACCEPTED MANUSCRIPT Stated Retrospective No
Partial
Convenience
Danzer et al. 201128
Inadequate
Convenience
Not stated
Prospective
Yes - WeeFIM stated to be reliable
Yes - WeeFIM Yes - WeeFIM score stated to be valid compared to normal value
De Groot et al. 200829
Adequate
Convenience
Stated
Retrospective
No
No
Yes - 6MWT (expressed as % of predicted)
De Groot et al. 30 2009
Inadequate
Convenience
Stated
Prospective
No
No
No
De Groot et al. 201031
Adequate
Convenience
Stated
Prospective
Yes - speed: ICC=0.97 for testretest
No
Yes - speed: SEM=2.5 m/min; SDD=6.8 m/min
De Groot et al. 2011a32
Inadequate
Community-based Stated
Prospective
Yes - 6MWT: testretest stated excellent
No
No
De Groot et al. 33 2011b
Adequate
Convenience
Stated
Prospective
Yes - 6MWT: ICC=0.98 for testretest
No
Yes - 6MWT: SEM=13.1m; SDD=36.3m
De Souza & Carroll Partial 34 1976
Convenience
Limited
Prospective
No
No
No
Dudgeon et al. 35 1991
Adequate
Convenience
Stated
Retrospective
Duffy et al. 1996
36
Inadequate
Convenience
Limited
Prospective
Evaggelinou & 37 Drowatzky 1991
Inadequate
Community-based Not stated
Feldman et al. 200838
Inadequate
Community-based Stated
Findley et al. 198739
Adequate
Population-based
Limited
Findley & Agre 40 1988
Adequate
Not stated
Limited
Convenience
Gerritsma-Bleeker Adequate 42 et al. 1997
Convenience
No
SC No
No
No
No
Yes - speed compared to normal value No
M AN U
No
Prospective
No
No
Prospective
Yes - WeeFIM: interrater (Kappa 0.44-0.82) stated
Yes - WeeFIM Yes - WeeFIM: stated to be valid severity rating for total score given
Prospective
No
No
No
Prospective
No
No
No
TE D
Franks et al. 199141 Adequate
No
RI PT
Danzer et al. 200927
Stated
Prospective
No
No
No
Limited
Retrospective
No
No
No
Limited
Restrospective
No
No
No
No
No
Convenience
Guille et al. 2002
Adequate
Convenience
Limited
Retrospective
No
Hassan et al. 201045
Adequate
Not stated
Stated
Prospective
Yes - 6MWT: stated No to be reliable
Yes - 6MWT: score compared to normal values
AC C
44
EP
Inadequate
Grujic & Aparisi 198243
Hisaba et al. 201246 Partial
Convenience
Stated
Retrospective
No
No
No
Hoffer et al. 197347 Adequate
Convenience
Stated
Retrospective
No
No
No
Høiness & Kirkhus Inadequate 200948
Convenience
Limited
Retrospective
No
No
No
Hunt et al. 197349
Inadequate
Convenience
Limited
Prospective
No
No
No
Ilharreborde et al. Inadequate 200950
Convenience
Limited
Retrospective
No
No
No
Jaworek et al. 201351
Not stated
Not stated
Prospective
No
No
No
Convenience
Not stated
Not stated
No
No
No
Adequate
Kaiser & Rudeberg Inadequate 52 1986 Kramel-Ross et al. 53 1992
Partial
Not stated
Limited
Prospective
No
No
No
Katz et al. 199754
Adequate
Convenience
Limited
Prospective
No
No
No
55
ACCEPTED MANUSCRIPT Limited Prospective No
No
No
Labruyere et al. 56 2013
Adequate
Not stated
Stated
Prospective
No
No
No
Lemelle et al. 57 2006
Inadequate
Community-based Stated
Prospective
No
No
No
Lewis et al. 2004
Partial
Convenience
Limited
Retrospective
No
No
No
Lorente Molto & 59 Garrido 2005
Adequate
Convenience
Limited
Retrospective & prospective
No
No
No
Lough & Nielsen 60 1986
Adequate
Convenience
Not stated
Prospective
No
No
No
Magee & Kenny 200861
Adequate
Not stated
Not stated
Not stated
No
No
No
Marreiros et al. 62 2010
Inadequate
Convenience
Stated
Retrospective
No
McCall & Schmidt 63 1986
Adequate
Not stated
Limited
Prospective
No
McDonald et al. 64 1991
Inadequate
Not stated
Limited
Prospective
No
Moerchen et al. 65 2011
Adequate
Convenience
Stated
Prospective
Moore et al. 2001
Adequate
Not stated
Not stated
Prospective
Müller et al. 67 1992a
Adequate
Convenience
Limited
Müller et al. 1992b68
Inadequate
Convenience
Limited
Not stated
Stated Stated
66
Müller et al. 199469 Adequate Adequate
Convenience
Murdoch et al. 197971
Inadequate
Convenience
Norrlin et al. 72 2003
Adequate
Not stated
OkurowskaZawada et al. 201173
Adequate
Convenience
Ozaras et al. 2005
Adequate
Convenience
Pauly & Cremer 201375
Partial
Convenience
Petrikovsky et al. 200176
No
No
No
No
No
No
Yes - FMS: 50m functional for home & daycare
No
No
Yes - speed compared to control values
No
No
No
Prospective
No
No
No
Retrospective & prospective
No
No
No
Prospective
No
No
No
Limited
Prospective
No
No
No
Stated
Prospective
No
No
No
Stated
Prospective
No
No
No
Not stated
Prospective
No
No
No
Not stated
Retrospective
No
Yes - Hoffer: convergent validity shown
No
AC C
74
No
EP
Müller-Godeffroy 70 et al. 2008
No
Prospective
TE D
58
RI PT
Not stated
SC
Inadequate
M AN U
Krebs et al. 1988
Inadequate
Convenience
Not stated
Prospective
No
No
No
Phillips & Lindseth Inadequate 199277
Convenience
Limited
Retrospective
No
No
No
Phillips et al. 199578
Adequate
Convenience
Limited
Retrospective
No
No
No
Pirpiris et al. 200379 Adequate
Convenience
Stated
Retrospective & Prospective
Yes - 10 min WT: ICC=0.91 for testretest
Yes - speed compared with normal values
Rose et al. 198180
Inadequate
Convenience
Limited
Prospective
No
Yes - 10MWT & 10 min WT: convergent validity No
No
Ross et al. 2007
Adequate
Convenience
Limited
Retrospective
No
No
No
Samuelsson & 82 Skoog 1988
Adequate
Convenience
Stated
Prospective
No
No
No
Schiltenwolf et al. 83 1991
Inadequate
Not stated
Not stated
Prospective
No
No
No
81
ACCEPTED MANUSCRIPT Not stated Prospective No
Shoenmakers et al. Adequate 200384
Convenience
Shoenmakers et al. Adequate 200485
Convenience
Stated
Prospective
Schoenmakers et 86 al. 2005a
Adequate
Convenience
stated
Prospective
Shoenmakers et al. Adequate 87 2005b
Convenience
Limited
Prospective
Yes - PEDI: stated to be reliable
Yes - PEDI: stated No to be valid
Schoenmakers et 88 al. 2009
Adequate
Convenience
Stated
Prospective
No
No
Seitzberg et al. 200889
Adequate
Convenience
Stated
Retrospective
No
Sherk et al. 1991
Adequate
Convenience
Not stated
Prospective
No
Stallard et al. 91 1991
Inadequate
Convenience
Limited
Prospective
No
Stillwell et al. 198492
Adequate
Convenience
Limited
Prospective
No
Suson et al. 2010
Inadequate
Convenience
Not stated
Retrospective
Swank & Dias 94 1992
Adequate
Convenience
Stated
Retrospective
Swank & Dias 95 1994 Tezcan & Simsek 96 2013
Inadequate
Convenience
Limited
Adequate
Convenience
Stated
Thomas et al. 200197
Adequate
Convenience
Stated
Thompson et al. 98 2000
Inadequate
Community-based Limited
Tsai et al. 200199
Adequate
Convenience
Not stated
Tsai et al. 2002
Adequate
Convenience
van denBergEmons et al. 101 2001 Verhoef et al. 2004102
Adequate
Convenience
Adequate
Population-based
Wai et al. 2005103
Adequate
Convenience
Walker et al. 2011104
Adequate
Convenience
AC C
100
WassenbergSeverijnen et al. 2003105
RI PT
No No
No
No
No
No
No
No
SC
No
No
No
No
No
No
M AN U
No
No
Prospective
No
No
No
Prospective
No
No
No
Prospective
No
No
No
Prospective
No
No
No
Stated
Prospective
No
No
Yes - PEDI scores compared with normative data
Stated
Prospective
No
No
No
Stated
Prospective
No
No
No
Stated
Prospective
Yes - ASK: stated to Yes - ASK: stated be reliable to be valid
No
Stated
Prospective
No
No
No
Yes - PEDI Mobility No scale: ICC>0.94 for test-retest, interrater No No
No
Stated
Prospective
Adequate
Not stated
Not stated
Prospective
Williams et al. 2005107
Inadequate
Community-based Stated
Prospective
Winchester et al. 2002108
Inadequate
Not stated
Stated
Prospective
Adequate
Convenience
Stated
Prospective
109
Yes - PEDI: scores compared to normative values
No
Convenience
Yngve et al. 1984
Yes - MBAC & PEDI (normative data)
No
Inadequate
Williams et al. 1983106
Yes - MABC & PEDI: Yes - MABC: conreliability stated current validity (not specific to SB) stated (not specific to SB) Yes - PEDI: intra- & Yes - PEDI: content inter-interviewer & construct stated stated
No
Prospective
TE D
93
EP
90
No
Yes - speed compared to normal values
Yes - TUG: ICC=0.99 Yes - TUG: for within session convergent test-retest validity shown
Yes - TUG: responsiveness shown in TD children Yes - speed Yes - GMFM: stated Yes - GMFM: high intra- & stated valid in CP, compared to interrater in CP BI & DS normal values No
Abbreviations: FIM=Functional Independence Measure; MRC=median reliability coefficient; 6MWT=6-minute walk test;
No
Yes - speeds compared to community walking requirements
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ICC=intraclass correlations coefficient; SEM=standard error of measurement; SDD=smallest detectable difference; FMS=Functional Mobility Scale; 10 min WT=10-minute walk test; 10MWT=10-meter walk test; MABC=Movement Assessment Battery for Children; PEDI=Pediatric Evaluation of Disability Inventory; SB=spina bifida; ASK=Activities Scale for Kids; TUG=Timed Up & Go test; TD=typically-developing; GMFM=Gross Motor Function Measure; CP=cerebral palsy; BI=brain injury; DS=Down syndrome.
ACCEPTED MANUSCRIPT
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ACCEPTED MANUSCRIPT
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66. Moore CA, Nejad B, Novak RA, Dias LS. Energy cost of walking in low lumbar myelomeningocele. J Pediatr Orthop 2001; 21: 388-91. 67. Müller EB, Nordwall A, von Wendt L. The influence of scoliosis brace treatment on function in children with myelomeningocele. Acta Paediatr 1992a; 81: 925-8. 68. Müller EB, Nordwall A, von Wendt L. Influence of surgical treatment of scoliosis in children with spina bifida on ambulation and motoric skills. Acta Paediatr 1992b; 81: 173-6. 69. Müller EB, Nordwall A, Oden A. Progression of scoliosis in children with myelomeningocele. Spine 1994; 19: 147-50.
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70. Müller-Godeffroy E, Michael T, Poster M, Seidel U, Schwarke D, Thyen U. Self-reported health-related quality of life in children and adolescents with myelomeningocele. Dev Med Child Neurol 2008; 50: 456-61. doi: 10.1111/j.1469-8749.2008.02054.x.
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71. Murdoch A, Young DG. A study of the relation between neonatal assessment of muscle power and later mobility in children with spina bifida defects. Z Kinderchir Grenzgeb 1979; 28: 387-92. 72. Norrlin S, Strinnholm M, Carlsoon M, Dahl M. Factors of significance for mobility in children with myelomeningocele. Acta Paediatr 2003; 92: 20410.
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73. Okurowska-Zawada B, Kulak W, Otapowicz D, Sienkiewicz D, Paszko-Patej G, Wojtkowski J. Quality of life in children and adolescents with cerebral palsy and myelomeningocele. Pediatr Neurol 2011; 45: 163-8. doi: 10.1016/j.pediatrneurol.2011.04.006.
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74. Ozaras N, Yalcin S, Ofluoglu D, Gureri B, Cabukoglu C, Erol B. Are some cases of spina bifida combined with cerebral palsy? A study of 28 cases. Eura Medicophys 2005; 41: 239-42. 75. Pauly M, Cremer R. Levels of mobility in children and adolescents with spina bifida - clinical parameters predicting mobility and maintenance of these skills. Eur J Pediatr Surg 2013; 23: 110-4. doi: 10.1055/s-0032-1324689. 76. Petrikovsky B, Pavlakis SG, Sichinava L. In utero testing of leg-withdrawal; prediction of ambulation in cases of meningomyelocele? Neonatal Intensive Care 2001; 14: 13-4.
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77. Phillips DP, Lindseth RE. Ambulation after transfer of adductors, external oblique and tensor fascia lata in myelomeningocele. J Pediatr Orthop 1992; 12: 712-7. 78. Phillips DL, Field RE, Broughton NS, Menelaus MB. Reciprocating orthoses for children with myelomeningocele. J Bone Joint Surg [Br] 1995; 77B: 110-3.
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79. Pirpiris M, Wilkinson AJ, Rodda J, Nguyen TC, Baker RJ, Nattrass GR, Graham HK. Walking speed in children and young adults with neuromuscular disease: comparison between two assessment methods. J Pediatr Orthop 2003; 23: 302-307.
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80. Rose GK, Stallard J, Sankarankutty M. Clinical evaluation of spina bifida patients using hip guidance orthosis. Dev Med Child Neurol 1981; 23: 30-40. 81. Ross M, Brewer K, Wright V, Agur A. Closed neural tube defects: neurologic, orthopedic, and gait outcomes. Pediatr Phys Ther 2007; 19: 288-95. 82. Samuelsson L, Skoog M. Ambulation in patients with myelomeningocele: a multivariate statistical analysis. J Pediatr Orthop 1988; 569-75. 83. Schiltenwolf M, Carstens C, Rohwedder J, Gründel E. Results of orthotic treatment in children with myelomeningocele. Eur J Pediatr Surg 1991; 1 Suppl 1: 50-2.
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84. Schoenmakers MAGC, Gooskens RHJM, Gulmans VAM, Hanlo PW, Vandertop WP, Uiterwaal CSPM, Helders PJM. Long-term outcome of neurosurgical untethering on neurosegmental motor and ambulation levels. Dev Med Child Neurol 2003; 45: 551-5.
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85. Schoenmakers MAGC, Gulmans VAM, Gooskens RHJM, Helders PJM. Spina bigida at the sacral level: more than minor gait disturbances. Clin Rehabil 2004; 18: 178-85. 86. Schoenmakers MAGC, Uiterwaal CSPM, Gulmans VAM, Gooskens RHJM, Helders PJM. Determinants of functional independence and quality of life in children with spina bifida. Clin Rehabil 2005a; 19: 677-85.
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87. Schoenmakers MA, Gulmans VA, Gooskens RH, Pruijs JE, Helders PJ. Spinal fusion in children with spina bifida: influence on ambulation level and functional abilities. Eur Spine J 2005b; 14: 415-22.
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88. Schoenmakers MAGC, De Groot JF, Gorter JW, Hillaert JL, Helders PJ, Takken T. Muscle strength, aerobic capacity and physical activity in independent ambulating children with lumbosacral spina bifida. Disabil Rehabil 2009; 31: 259-66. doi: 10.1080/09638280801923235. 89. Seitzberg A, Lind M, Biering-Sorensen F. Ambulation in adults with mylomeningocele. Is it possible to predict the level of ambulation in early life? Childs Nerv Syst 2008; 24: 231-7. 90. Sherk HH, Uppal GS, Lane G, Melchionni J. Treatment versus non-treatment of hip dislocations in ambulatory patients with myelomeningocele. Dev Med Child Neurol 1991; 33: 491-4.
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91. Stallard J, Major RE, Butler PB. The orthotic ambulation performance of paraplegic myelomeningocele children using the ORLAU parawalker treatment system. Clin Rehabil 1991; 5: 111-4. 92. Stillwell A, Menelaus MB. Walking ability after transplantation of the iliopsoas. J Bone Joint Surg 1984; 66B: 656-9.
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94. Swank M, Dias L. Myelomeningocele: a review of the orthopaedic aspects of 206 patients treated from birth with no selection criteria. Dev Med Child Neurol 1992; 34: 1047-52. 95. Swank M, Dias LS. Walking ability in spina bifida patients: a model for predicting future ambulatory status based on sitting balance and motor level. J Pediatr Orthop 1994; 14: 715-8. 96. Tezcan S, Simsek TT. Comparison of health-related quality of life between children with cerebral palsy and spina bifida. Res Dev Disabil 2013; 34: 2725-33. doi: 10.1016/j.ridd.2013.05.017. 97. Thomas SS, Buckon CE, Melchionni J, Magnusson M, Aiona MD. Longitudinal assessment of oxygen cost and velocity in children with myelomeningocele: comparison of the hip-knne-ankle-foot orthosis and the reciprocating gait orthosis. J Pediatr Orthop 2001; 21: 798-803.
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S0003-9993(15)00384-6
DOI:
10.1016/j.apmr.2015.04.014
Reference:
YAPMR 56185
To appear in:
ARCHIVES OF PHYSICAL MEDICINE AND REHABILITATION
Received Date: 12 February 2015 Revised Date:
16 April 2015
Accepted Date: 21 April 2015
Please cite this article as: Bisaro DL, Bidonde J, Kane KJ, Bergsma SA, Musselman KE, Past and current use of walking measures for children with spina bifida: a systematic review, ARCHIVES OF PHYSICAL MEDICINE AND REHABILITATION (2015), doi: 10.1016/j.apmr.2015.04.014. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Running Head: Walking measurement in spina bifida
review
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Past and current use of walking measures for children with spina bifida: a systematic
Derek L Bisaro1 MPT, Julia Bidonde1 PhD, Kyra J Kane1 MSc, Shane A Bergsma2 PhD, Kristin
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E Musselman1,3,4 PhD
School of Physical Therapy, College of Medicine, University of Saskatchewan, Saskatoon, SK;
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Department of Computer Science, College of Arts and Science, University of Saskatchewan,
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Saskatoon, SK; 3Toronto Rehabilitation Institute – University Health Network, Toronto, ON; Department of Physical Therapy, Faculty of Medicine, University of Toronto, Toronto, ON.
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This work has been accepted for presentation at the 2015 meeting of the Canadian Physiotherapy Association.
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This work was supported by grants from the Spina Bifida and Hydrocephalus Foundation of Canada and the College of Medicine, University of Saskatchewan, to KEM.
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Corresponding Author:
Kristin Musselman PT, PhD SCI Mobility Lab, Lyndhurst Centre – TRI 520 Sutherland Drive, Toronto, ON, M4G 3V9 [email protected] 416-597-3422 ext.6190
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Running Head: Walking measurement in spina bifida
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Past and current use of walking measures for children with spina bifida: a systematic
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review
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ABSTRACT
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Objective: To describe walking measurement in children with spina bifida, and to identify
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patterns in the use of walking measures in this population.
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Data Sources: Seven medical databases were searched from inception until March 2014. Search
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terms encompassed three themes: 1) children, 2) spina bifida, and 3) walking.
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Study Selection: Articles were included if participants were children aged 1-17 years with spina
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bifida, and if walking was measured. Articles were excluded if the assessment was restricted to
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kinematic, kinetic or electromyographic analyses of walking. A total of 1,751 abstracts were
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screened by two authors independently, and 109 articles were included in this review.
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Data Extraction: Data were extracted using standardized forms. Extracted data included study
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and participant characteristics, and details about the walking measures used, including
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psychometric properties. Two authors evaluated the methodological quality of articles using a
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previously published framework that considers sampling method, study design, and
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psychometric properties of the measures used.
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Data Synthesis: Nineteen walking measures were identified. Ordinal-level rating scales (e.g.,
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Hoffer Functional Ambulation Scale) were most commonly used (57% of articles), followed by
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ratio-level, spatiotemporal measures, such walking speed (18% of articles). Walking was
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measured for a variety of reasons relevant to multiple health care disciplines. A machine learning
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analysis was used to identify patterns in the use of walking measures. The learned classifier
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predicted whether or not a spatiotemporal measure was used with 77.1% accuracy. A trend to use
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spatiotemporal measures in older children and those with lumbar and sacral spinal lesions was
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identified. Most articles were prospective studies that used samples of convenience and
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unblinded assessors. Few articles evaluated or considered the psychometric properties of the
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walking measures.
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Conclusions: Despite a demonstrated need to measure walking in children with spina bifida, few
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valid, reliable and responsive measures have been established for this population.
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Keywords: walking, spinal dysraphism, review
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Spina bifida is one of the most common congenital birth defects, with a current prevalence of
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about 0.3-0.4/1,000 births in Canada1 and the United States2. With this condition, a neural tube
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defect results in damage to the spinal cord, brain and/or meninges. Walking is a challenge for
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many children with spina bifida. More than half of those with neurological lesions at or below
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the thoracic level achieve walking at some point in their childhood, with the walking rate
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increasing as the lesion level decreases.3-6 As a result of their sensorimotor impairments, many
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children with spina bifida walk with abnormal gait patterns. This significantly increases the
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energy cost of walking and reduces their walking endurance.7-11 A notable proportion of children
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with spina bifida lose the ability to walk as they age.4,5
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Achieving and/or improving the ability to walk is an important goal for many children with spina
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bifida and their families, as walking enables greater participation in daily activities, recreation,
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and contributes positively to quality of life. As a result, walking is often a focus of the medical
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management and rehabilitation of those children who have the potential to walk.3 Considerable
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effort is spent investigating therapeutic approaches that may lead to improved walking outcomes
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for children with spina bifida, such as walking training11,12, surgical procedures13,14, neural
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prostheses15 and orthoses16-18.
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In order for researchers and clinicians to accurately evaluate the effects of an intervention on
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walking, and to follow a child’s walking ability over time, valid, reliable and responsive
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measures of walking must be used. Laboratory-based assessments involving sophisticated
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motion analysis systems can measure kinematic gait characteristics in children with spina
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bifida.16,19 Since most clinicians do not have access to such systems, walking measures that can
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be administered in a clinical setting are required. Furthermore, the measures must have good
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psychometric properties (i.e., validity, reliability, responsiveness) to be useful. To date, however,
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there is little guidance for clinicians and researchers as to what walking measures are useful for
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children with spina bifida.
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As a first step toward providing guidance on how to assess walking in children with spina bifida,
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we performed a systematic review and critical evaluation of the literature. Our objectives were
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to: 1) identify the walking measures used in clinical settings for children with spina bifida, 2)
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describe the circumstances under which these measures were used, and 3) evaluate these
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measures with respect to their psychometric properties reported in the literature.
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METHODS
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A systematic review was performed following the PRISMA (Preferred Reporting Items for
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Systematic Reviews and Meta-Analysis) guidelines.20 There is no protocol for this review. To
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form the review question, a modified PICOS (population, interventions or indicators,
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comparators, outcomes and study design) framework was used. The population was children
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(i.e., aged 1-17 years, inclusive) with spina bifida. The indicator was a measure of walking that
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could be used in a clinical setting. There were no comparators. The outcome of interest was
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walking ability, and there were no restrictions on study design.
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Search Strategy
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A literature search was completed in consultation with an Information Specialist at the
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University of Saskatchewan. Seven databases were searched (Medline, PubMed, Embase,
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Scopus, Web of Science, Cinahl, and Amed) from the earliest known record until March 11,
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2014. The search terms included keywords and controlled vocabulary (where applicable) and
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focused on the following themes: 1) children, 2) spina bifida, and 3) walking (see Appendix 1 for
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search example). No restrictions were placed on the language, date, or type of publication.
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Duplicate records were removed using a research management tool (REFworks, Bethesda, MD).
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All abstracts were then independently reviewed by two authors (___ and ___) to select the
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articles for full text review. The inclusion criteria were as follows:
1) Study participants were children with spina bifida (diagnoses included: spinal
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dysraphism, neural tube defects, meningomyelocele, congenital or nontraumatic spinal
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cord injuries, spina bifida, myelomeningocele, lipomyelomeningocele, lipomeningocele,
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spinal cord malformation).
2) Functional walking capacity, defined as “the ability to ambulate daily using reciprocal
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steps overground for short distances” with or without assistive devices 21, was assessed or
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reported in some way. This definition of was chosen since it reflects walking that is
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clinically relevant.
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Exclusion criteria included:
1) Studies where the participants were exclusively infants (i.e. <1 year old) or exclusively
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adults (i.e., >18 years old).
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2) Studies that did not include any individuals with spina bifida.
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3) Studies that did not examine walking function (e.g., examined gross motor function or
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wheeled mobility).
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123 124 125 126 127
4) Studies in which walking was evaluated only through kinematic, kinetic or electromyographic analyses (i.e., measures not routinely used in clinical settings). 5) Studies in which walking ability was given a dichotomous classification of ambulatory or non-ambulatory.
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6) Animal studies, modelling studies, narrative review articles, conference proceedings, editorials or letters to the editor.
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Data extraction
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The following data were extracted from included full text articles using a standard data
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extraction form: patient populations tested, type of neural tube defect, level of lesion, age of
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participants, number of child participants with spina bifida, method of participant recruitment,
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methods of walking assessment, purpose of walking assessment, psychometric properties of the
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walking measures used, results of walking measurement, and type of study. For articles that were
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not in English, individuals proficient in the language and/or Google Translate assisted with
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translation, and in one case the author of the article was contacted for the information in English.
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Methodological quality of the included articles was assessed by adapting the methods of Dobson
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et al.22 Nine methodological characteristics were assessed, such as the sampling method and
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psychometric properties of the walking measures used (see Table 1). These categories were rated
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as adequate /inadequate or stated/not stated depending on the question. Methodological quality
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was evaluated by a pair of reviewers independently and the final decisions reached through
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consensus.
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(Table 1 near here)
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Data Synthesis
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Descriptive summaries
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Extracted data from all included articles were summarized to describe the use of walking
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measures among children with spina bifida. As this is a descriptive review, there is no principal
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summary measure. The following analyses were completed:
1) The number of times a given measure was used was counted and expressed as a
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percentage of the total number of times a walking measure was used in the included
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articles.
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2) The number of times a given study purpose was reported was counted. For example,
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possible study purposes included investigating the relationship between walking ability
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and another variable, or examining the effects of a surgical or orthotic intervention. 3) For each walking measure identified, information concerning its validity, reliability,
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responsiveness and/or interpretability (e.g., interpretation of scores relative to normative
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data, cut-off values) was aggregated.
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Statistical machine learning analysis
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To further describe how walking measures are used in children with spina bifida, we determined
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whether there was any pattern to the selection of a measure. For example, are categorical rating
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scales chosen because of participant age, and/or because of the range of lesion levels? If such
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patterns exist, they would inform future measurement guidelines. The included studies were
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classified along two separate dimensions. First, each study was classified as to whether it
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included a spatiotemporal, ratio-level measure (e.g., walking speed or distance) or not. Second,
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each study was classified as to whether it included a categorical, ordinal-level measure (e.g.,
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Hoffer Functional Ambulation Scale) or not. While not all measures identified in this review fell
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into one of these two categories (e.g., the Walking, Running and Jumping dimension of the
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Gross Motor Function Measure), all studies included at least one measure that did. To detect
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whether there were any patterns to the selection of spatiotemporal or categorical measures, we
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performed a statistical analysis using techniques from the discipline of machine learning.
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Specifically, we trained two decision tree classifiers23 to predict whether (A) a spatiotemporal
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measure would be used or not, and (B) whether a categorical measure would be used or not. For
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both the (A) and (B) analyses, the classifiers made their predictions on the basis of the following
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inputs from each study: (1) the reason for the walking assessment, (2) the minimum and
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maximum ages of the children tested, (3) the rostral and caudal levels of spinal lesion (labeled as
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thoracic, lumbar or sacral), and (4) the publication date.
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The C4.5 decision tree algorithm was chosen as the classifier because it performs well on a
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variety of problems, and generates a learned model that is easily interpretable.23 Moreover, as
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opposed to logistic regression, C4.5 is able to consider nonlinear combinations of the inputs, and
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is therefore able to learn a more expressive predictive model. To train the C4.5 classifier, the
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Weka data mining tool was used (with default parameter settings).24
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After training the classifier, we tested its ability to predict the output class on new, previously-
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unseen data. Specifically, we determined whether the classifier discovered a non-trivial
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relationship between the inputs and the output class. A trivial relationship is one where the most
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common output is always selected, regardless of the inputs. In machine learning terminology,
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such a classifier is referred to as the majority-class baseline. For example, in our analysis, the
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majority-class baseline would always predict ‘categorical’ as the output class, since categorical
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measures were more commonly used. If the C4.5 classifier was able to predict the correct output
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at a level above the majority-class baseline, it would suggest that there was some non-trivial
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pattern to the choice of walking measure. To evaluate the classifier’s prediction accuracy on new
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data, 10-fold-cross-validation was used. McNemar’s Test was used to determine whether the
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learned classifier was more accurate than the majority-class baseline. Alpha was set at 0.05.
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Analysis of risk of bias
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Lastly, the overall risk of bias (i.e., bias of the walking measurements performed) of the included
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articles was evaluated by synthesizing the data extracted regarding methodological quality. Risk
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of bias may be high, low, or unclear (i.e., insufficient information).25 Information concerning
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sampling, blinding, and the selection of a walking measurement were considered. Sampling bias
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was considered high if the majority of studies used samples of convenience, and low if the
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majority of studies used community- or population-based samples. If blinded assessors were
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frequently used for the assessment of walking, the risk of bias was considered low, otherwise the
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risk was high. For the selection of a walking measure, whether or not the psychometric
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properties of a measure were considered by the study authors was used to gauge risk (i.e., if
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considered, then low risk of bias).
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RESULTS
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A total of 1,751 abstracts were screened, and 217 met the inclusion criteria for full text review.
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After full text review, 109 articles were included in the study (see Figure 1). A summary of the
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data extracted from each included article can be found in Appendix 2. Among the included
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articles, many did not specify the type of spina bifida studied. In those that did, the most
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common type studied was myelomeningocele followed by lipomyelomeningocele and
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meningocele. Twenty-one (19%) of the included studies involved other pediatric populations in
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addition to spina bifida, with cerebral palsy and spinal cord injury being the most frequent. The
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ages of the children studied spanned the entire childhood range (i.e., 1 – 17 years).
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(Figure 1 near here)
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Methodological quality of included studies
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The quality evaluation summary for each included article can be found in Appendix 3, with the
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exception of the question regarding whether or not the assessor was blinded. Only 3 of the 109
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studies stated that a blinded assessor was used.26-28 The remaining studies did not comment on
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this methodological aspect, making the risk of bias unclear.
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Samples of convenience were used in 82 studies (75.2%), placing the study findings at a greater
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risk of bias than the studies that used community- and population-based samples (5.5% and
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1.8%, respectively). The sampling method was not outlined in 19 studies (17.4%). Thus, overall
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the risk of sampling bias in the included articles was high.
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Participant characteristics (age, gender, number and type of neural tube defect) were adequately
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defined in 65 studies (59.6%). All details, except gender, were reported in 8 studies (7.3%, rated
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‘partial’, Table 1), whereas the characteristics were inadequately reported in 36 studies (33%).
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The inclusion and exclusion criteria of the study were outlined in 47 studies (43.1%), while 37
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studies (33.9%) specified 1-2 criteria (rated ‘Limited’ as per Table l). A notable number of
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studies (25 or 22.9%) did not list any inclusion or exclusion criteria for their study participants.
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Most studies were prospectively planned and executed (77.1%).
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Few studies considered the psychometric properties of the walking measures used, contributing
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to a high risk of bias. Fifteen studies (13.8%) reported on the reliability of the measure
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used9,13,26,27,29-39; however, the reported reliability was rarely established in children with spina
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bifida. Two exceptions were the 6-minute walk test and Timed Up and Go test; both having high
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test-retest reliability in children with spina bifida.30,38 Likewise, at least one type of validity was
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reported or tested for the walking measures used in 10 studies (9.2%).13,27,29,31,32,34,35,37,38,40
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Interpretability of a measure was reported at a slightly higher frequency (19 studies or
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17.4%).7,9,12,29-35,37,38,41-47 Most commonly, the scores on a walking measure were compared to
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control participants or normative data.7,9,29,31,32,34,35,41,43-47 The standard error of measurement and
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smallest detectable difference were calculated for the 6-minute walk test in children with spina
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bifida.30,33 In Williams et al.38 responsiveness was assessed for the Timed Up and Go Test in
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typically-developing children, but not in the study participants with spina bifida.
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Walking measures used in included articles
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Nineteen different walking measures were used across the 109 articles. The Hoffer Functional
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Ambulation Scale was most commonly used by researchers (36% of the articles). The Hoffer
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Scale is an categorical scale with four levels: community ambulator, household ambulator, non-
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functional ambulator, and non-ambulator.48 Various modifications to the Scale have been
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made13,26; however, it has remained an ordinal-level measure that outlines descriptive categories
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of walking. Another 21% of studies used custom-made, categorical scales, similar in nature to
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the Hoffer Scale. These scales could be divided into three groups based on the variable used to
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categorize the walking. Many scales were based on the type of assistive device used 29,41,49,50-59,60-
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62
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walking distance as a means to define walking categories.72-74
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or the walking environment (e.g., outdoors/community, indoor/household)42, 63-71. Fewer used
Walking speed was also measured (18% of included articles), however, the method used varied.
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Some measured the speed over a pre-determined distance, such as 30m43,75-77, 6m78, 110m79 or a
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60.5m oval track44. Others used a pre-determined walking time to obtain a measure of speed
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(e.g., walking for 3 minutes45,77,80 or 6 minutes as part of the 6-minute walk test7,30,81). Only two
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studies used the 10-meter walk test.31,32 Self-selected walking speed was always measured,
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whereas fastest/maximum speed was also measured in four studies.43,44,76,82
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Measures less frequently used included the 6-minute walk test to measure walking
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distance7,9,10,26,30,33,83 (5% of articles), the Mobility Domain of the Pediatric Evaluation of
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Disability Inventory (PEDI)12,13,34-36,41,84,85 (5% of articles), and the Mobility Domain of the
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Pediatric Functional Independence Measure (WeeFIM)29,37,86-89 (4% of articles). The remaining
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11% of articles included outcomes that were used only 1-3 times: Timed Up and Go test38,
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Physiological Cost Index of Walking84,90,91, Gross Motor Function Measure32, Functional
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Independence Measure39, Movement Assessment Battery for Children34, Functional Ambulation
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Categories88, Activities Scale for Kids27, Functional Mobility Scale12,92, Personal NEM (Necker-
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Enfants Malades) motor scale93, and the Progressive Ambulation Scale89. Two studies rated the
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children’s ability to complete a number of functional tests, such as ascending and descending
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steps94,95 and a ramp95.
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Reasons for walking measurement
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Walking was evaluated in children with spina bifida for a variety of reasons. Most commonly,
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walking was measured so that the relationship between walking ability and another variable
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could be examined (Figure 2). For example, there was an interest in describing or quantifying the
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relationship between walking ability and quality of life35,87, wait time for rehabilitation37, spinal
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deformity27, extent of neurological impairment85 and peak VO27. Examining the effect of an
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orthotic device or surgical intervention on walking performance were also common reasons for
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measuring walking ability, whereas investigating the effect of a physical intervention, such as
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treadmill training12,26 or neuromuscular electrical stimulation89,94, was less common. A number
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of studies measured walking in order to describe the walking status of participants, either at one
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point in time6,34,48,50,56,96-101 or longitudinally54,73,102-105. There was also an interest in predicting
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the future walking status of children.40,51-53,65,103,106-108 Less commonly, walking was measured
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for methodological reasons, such as determining study eligibility30,33,83,90 or to assist with the
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prescription of exercise testing83. Only four studies examined one or more psychometric
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properties of a walking measure in the spina bifida population.31,33,36,38
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(Figure 2 near here)
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Results of machine learning analysis
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The machine learning analysis found a non-trivial pattern for the spatiotemporal output class, but
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not the categorical output class. For the spatiotemporal output class, the accuracy of the majority-
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class baseline was 70.6%. This means that if the majority class (i.e., did not use a spatiotemporal
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measure) was selected for every article, the resulting accuracy was 70.6%. In comparison, the
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accuracy from 10-fold-cross validation of the learned classifier was 77.1%. The difference in
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accuracy between the learned classifier and the majority-class baseline did not reach statistical
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significance (p=0.15).
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Figure 3 shows the decision tree algorithm for the spatiotemporal output class. The learned
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classifier could predict whether or not a spatiotemporal measure of walking was used by
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considering three of the four inputs: (1) the reason for the walking assessment (i.e., the study
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purpose), (2) the minimum age of the children tested, and (3) the rostral level of spinal lesion. If
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the reason for the walking assessment was to evaluate the effects of an orthosis, then a
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spatiotemporal measure was used. If the reason was to investigate the effects of surgery or to
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predict future walking status, then a spatiotemporal measure was not used (i.e., a categorical
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rating scale was used instead). When the purpose of the walking assessment was to describe
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walking ability (cross-sectional or longitudinal analyses), the choice was dependent on age. If the
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study sample included young children (<3 years of age), then spatiotemporal measures were not
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used. However, if all study participants were older than 3 years, the choice was further
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determined by the participants’ level of spinal lesion. If the sample included children with
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thoracic lesions, spatiotemporal measures were not used. If the sample included children with
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lumbar and sacral lesions only, then spatiotemporal measures were used. Likewise, if the reason
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for walking assessment was to investigate the relationship between walking and another variable,
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the same decision rule for the input of lesion level was seen.
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(Figure 3 near here)
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DISCUSSION
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We identified nineteen clinical measures that have been used to assess walking ability in children
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with spina bifida through a systematic review that followed PRISMA guidelines. The review
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resulted in 109 included articles, with publication dates spanning four decades. The nineteen
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walking measures identified were used for a variety of study purposes across multiple disciplines
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of medicine and rehabilitation. The Hoffer Functional Ambulation Scale48 and other categorical
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scales were most commonly used. Walking speed was also utilized as an outcome, although there
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was no standard approach to its measurement. By employing a machine learning analysis, we
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found a trend toward using spatiotemporal measures for children school-aged and older, for those
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with a lower level of spinal lesion, and when investigating the effects of an orthosis. Little work
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has been done to validate any of the identified walking measures, or to assess their
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responsiveness, in the spina bifida population. The reliability of the measures has also not been
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established the spina bifida population, with the exception of test-retest reliability of the 6-
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minute walk test in higher-functioning walkers30,33 and the Timed Up and Go test in a small
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sample38. Hence, while there is a clear interest and need to measure walking in children with
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spina bifida across multiple health care disciplines, clinicians and researchers lack valid, reliable
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and responsive measurement tools.
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The Hoffer Functional Ambulation Scale
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The Hoffer Scale, which was specifically designed for spina bifida48, was the most commonly
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used measure across the studies included in this review. This Scale is an appealing option for
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clinical use for several reasons. First, it is easy to use and does not require any training prior to
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use. Second, it can be administered quickly with little effort required from the child. Third, the
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Hoffer Scale is inclusive of all levels of walking ability, from fully independent to unable to
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walk. This means that all children with spina bifida, regardless of physical ability, can be rated
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on this one measure. Fourth, it can be used across the lifespan of someone with spina bifida.
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Consistent with these latter two points are our findings that there was a tendency to use
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categorical rating scales in studies involving young children and/or those with thoracic-level
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lesions (see Figure 3). And lastly, the Scale was shown to have convergent validity.40 Thus, there
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are numerous advantages to using the Hoffer Scale in clinical environments.
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Formal assessment of the clinical utility of the Hoffer Functional Ambulation Scale supports its
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use in clinical practice. Tyson and colleagues109 report a tool to assess the clinical utility of
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walking and mobility measures in neurological populations. The tool rates an outcome measure
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on four dimensions: time required, cost, need for equipment or training, and portability. A total
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score out 10 is assigned, with scores of 9 or 10 suggesting the measure is appropriate for clinical
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use.109 Application of their tool to the Hoffer Scale would result in a score of 10. Thus, the
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feasibility of using the Hoffer Scale in a clinical setting is high.
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Despite numerous advantages of the Hoffer Scale, there are two noteworthy limitations. With
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only four levels of ambulatory ability to choose from, the Hoffer Scale likely lacks
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discriminative validity and responsiveness. For example, two children can walk outdoors for
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short distances; however, one child walks with bilateral ankle foot orthoses at a speed of 0.8m/s,
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while the other walks with a walker at a speed of 0.4m/s. Despite considerable differences in
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their speeds and reliance on assistive devices, both children are classified as community
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ambulators. Thus, the Scale does not adequately discriminate between different ambulatory
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abilities. Similarly, the Hoffer Scale may only be able to detect very large changes in walking
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function (i.e., lacks responsiveness); however, this is speculative and needs to be confirmed
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through proper psychometric study. Overall, the Hoffer Functional Ambulation Scale provides a
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snapshot of a child’s walking ability, and is likely an effective way to describe a child’s walking
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status at a single point in time.
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Walking speed
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In addition to categorical rating scales, walking speed was measured in the included articles, but
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at a considerably lower frequency. Measuring walking speed has numerous advantages. Its
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clinical utility is high, as it is low-cost, portable, time-efficient, and does not require equipment.
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The 5-meter and 10-meter walk tests score 10/10 on the evaluation of clinical utility.109
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Furthermore, speed is a metric that is easily interpretable, and possibly more responsive than
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categorical scales.
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Despite these advantages, spatiotemporal measures, like speed, were not used to evaluate the
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effects of surgery or to predict future walking status. It is easier to make a prediction using a
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categorical rating scale than a ratio-level measure. Why spatiotemporal measures have not been
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used to assess the efficacy of surgery is not clear, but they are likely suitable for this study
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purpose.
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How to best measure walking speed in pediatric populations is unknown. We found that the
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walking distance over which speed was measured varied greatly, from as short as 6m78 to as long
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as the distance that could be covered in 10 minutes31. Self-selected speeds measured over
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different walking distances have been found to differ when directly compared in children with
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spina bifida and other neuromuscular diseases.31 Shorter distances are likely most feasible for
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children; compliance may be an issue with longer tests. Longer distances would also preclude
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lower-functioning walkers from participation. For example, De Groot and colleagues7 used the 6-
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minute walk test in those children rated as ‘Normal’ or ‘Community’ ambulators on the Hoffer
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Scale.
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In our review, we found little investigation of the validity, reliability and responsiveness of
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walking speed measurement in children with spina bifida.30 In adolescents with cerebral palsy,
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the 10-meter walk test has been shown to be a valid measure.110 However, the test-retest
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reliability of the 10-meter walk test (performed at a fast speed) was found to be inadequate in
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children with cerebral palsy aged 4-18 years.111 Yet, in the same study, the reliability of the 6-
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minute walk test was found to be excellent.111 Thus, while longer walking tests may not be as
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practical for pediatric populations, they may yield more reliable results. In addition, longer
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distances may be more representative of walking performance in the community.31 Investigation
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of the psychometric properties of short and long walking tests to measure speed in children with
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spina bifida is warranted. Adopting a standard approach to the assessment of walking speed for
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ambulatory children with spina bifida would facilitate accurate tracking of client progress and
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comparison of research findings across studies.
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Methodological quality of included articles
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We found that the majority of included articles involved samples of convenience and likely
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unblinded assessments. A notable proportion of the studies did not describe the participants’
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characteristics or the inclusion/exclusion criteria adequately. Furthermore, few articles explicitly
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considered the psychometric properties of the walking measures used. All together this
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contributes to a greater risk of bias for the measurement findings.
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Gaps in walking measurement for spina bifida
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Walking is a complex behavior that can be measured along different dimensions. Walking
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capacity refers to the ability to execute the task of walking, whereas walking performance refers
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to the ability to walk in one’s usual environment.112 Many of the measures identified in this
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review are indicators of walking capacity (e.g., 10-meter walk test), however, some of the
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ordinal scales probe walking performance. Our results suggest that psychometrically-sound
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measures of walking performance are needed for children with spina bifida, as has been
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suggested for neurological populations112.
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Furthermore, walking in one’s usual environment involves many different walking skills, such as
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negotiating obstacles and different surfaces.113 Most measures identified in this review assess a
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single walking task (i.e., walking over level ground). Quantitative scales consisting of multiple
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tasks relevant to daily walking are lacking for children with spina bifida. Such scales exist for
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adult populations (e.g., the Spinal Cord Injury Functional Ambulation Profile114), and could be
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adapted for pediatric populations.
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Strengths and limitations of review
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There are several noteworthy strengths of this systematic review. First, we described the current
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state of walking measurement in children with spina bifida, and identified areas in need of future
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research. Second, we incorporated a unique analysis (i.e., machine learning) into the review that
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provided richer insight into how the identified walking measures have been used in past research.
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Lastly, this review could serve as a model for future systematic reviews that aim to characterize
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the use of outcome measures in a patient population.
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There are also potential limitations to note. First the overall risk of bias of the included articles
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was deemed high. This means that although the review can accurately report what walking
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measures were used, appropriateness of these measures for the specific study purpose and
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participants may be questionable. The measures reported in this review do not necessarily
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represent the best measurement options. Second, the methods used to measure walking may
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change over time. We did not place a limit on the publication date of the articles in order to
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collect a sufficiently large amount of data. It is possible that the review results would be different
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had we limited our review to research conducted in the past decade. However, the machine
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learning analysis did not find a relationship between publication date and the choice of walking
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measure (i.e., spatiotemporal or categorical). Lastly, the focus of this review was walking, which
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is just one of the many gross motor skills important in childhood. Measures of general gross
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motor function are likely clinically useful for children with spina bifida. Future work could
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explore the state of gross motor function assessment in this population.
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Recommendations for walking measurement in children with spina bifida
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Currently, clinicians and researchers have few valid and reliable walking measures to choose
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from for children with spina bifida. A variety of custom-made, categorical scales were
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commonly used. For the time-being, we recommend using the Hoffer Functional Ambulation
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Scale as a means to classify a child’s walking at a single point in time. This will help to increase
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consistency across research and clinical practice. Further work is needed to evaluate the validity
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(e.g., discriminative) and reliability (e.g., test-retest, inter-rater) of the Hoffer Scale. If working
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with a child with good walking function (i.e., ‘Community’ or ‘Normal’ ambulators on the
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Hoffer Scale), the 6-minute walk test is appropriate and supported by the literature.30,33 Future
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research is needed to evaluate the use of the 6-minute walk test and other spatiotemporal
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measures in young children.
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We propose that both the research and clinical realms of spina bifida management would benefit
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from an organized approach to walking measurement. The measures should be chosen with a
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child’s age and lesion or functional level in mind, while at the same time reflect the multiple
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dimensions of walking function. The assessment could include both a categorical rating scale
479
(i.e., Hoffer Scale) and also spatiotemporal measures. A small proportion (15%) of the articles
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reviewed included both categorical and spatiotemporal measures. Future work should build upon
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previous pediatric research to identify walking measures that are valid, reliable and responsive
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for children with spina bifida.
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Conclusions
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Through a systematic review we identified nineteen measures that have been used to examine
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walking in children with spina bifida. Spatiotemporal measures were more likely to be used for
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older children, and in children with lower spinal lesions (i.e., lumbar and sacral lesions). Little
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work has been done to establish the validity, reliability and/or responsiveness of walking
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measures in this population. Future research should focus on this knowledge gap so that clinical
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guidelines can be developed for walking measurement in spina bifida.
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111. Thompson P, Beath T, Bell J, et al. Test-retest reliability of the 10-metre fast walk test and 6-minute walk test in ambulatory school-aged children with cerebral palsy. Dev Med Child Neurol 2008;50:370-6. doi: 10.1111/j.1469-8749.2008.02048.x. 112. Pearson OR, Busse ME, Van Deursen RWM, Wiles CM. Quantification of walking mobility in neurological disorders. Q J Med 2004; 97: 463-75. Doi:10.1093/qjmed/hch084. 113. Musselman KE, Yang JF. Walking tasks encountered by urban-dwelling adults and persons with incomplete spinal cord injuries. J Rehabil Med 2007; 39: 567-74.
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114. Musselman KE, Brunton K, Lam T, Yang JF. Spinal cord injury functional ambulation profile: a new measure of walking ability. Neurorehabil Neural Repair 2011; 25: 285-93. doi: 10.1177/1545968310381250.
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893 894 895 896 897 898 899 900 901 902 903 904 905 906 907 908 909 910 911 912 913 914 915 916 917 918 919 920 921
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Figure Legends
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Figure 1: PRISMA Flow Diagram. Outline of the identification and screening of abstracts and
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Figure 2: Reasons for Walking Assessment in Children with Spina Bifida. Number of included
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articles (bars) for each study purpose (left text). The purpose ‘Describe walking status’ was
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observed in cross-sectional and cohort studies. A study may have more than 1 stated purpose,
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Figure 3: Decision Tree for Choosing a Spatiotemporal Walking Measure in Children with
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Spina Bifida. For ‘Reason of walking measurement’: ‘If orthosis’ = if examining the effects of an
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orthosis on walking. ‘If surgery’ = if examining the effects of a surgical procedure on walking.
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‘If description’ = if describing walking status, at one point or over time. ‘If prediction’ = if
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predicting future walking status. ‘If relationship’ = if investigating the relationship between
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walking and another variable. ‘Rostral level’ refers to the highest level (sacral, lumbar or
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thoracic) of spinal lesion among study participants.
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Table 1: Study Quality Evaluation Tool (adapted from Dobson et al.22)
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Decision Rules Adequate = all details provided; Partial = all details except gender; Inadequate = missing details. Convenience = participants recruited included patients from local hospital; Community-based = e.g., participants recruited from >1 local hospital or organization with aim of reaching all potential participants in the area; Population-based = as per community-based, but geographical area larger (e.g., country- or state/province- wide); Not stated = no details about sampling method provided. Stated = clear list of both provided; Limited = 1 or 2 points only; Not stated = no details about inclusion or exclusion provided. Prospective = walking data collected at time of study, e.g., objective exam; Retrospective = walking data collected prior to study initiation, e.g., chart review; Not stated = no details provided re: time of walking assessment. Yes; No; Not stated. Yes (list type(s), e.g., inter-rater, test-retest, internal consistency, etc.); No. Yes (list type(s), e.g., concurrent, criterion, content, etc.); No. Yes (list type(s), e.g., minimally important difference, responsiveness, cut-points, etc.); No.
3. Are inclusion and exclusion criteria stated?
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4. Was the walking assessment performed prospectively or retrospectively?
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Question 1. Are participant characteristics defined including age, gender, number, and type of NTD? 2. What sampling method was used?
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5. Was the assessor blinded to the study hypothesis and/or groups? 6. Was the reliability of the tool/classification stated or demonstrated? 7. Was the validity of the tool/classification stated or demonstrated? 8. Was the interpretability or responsiveness of the tool/classification reported or tested? NTD = neural tube defect.
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Appendix 1 PubMed Search
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("child"[MeSH Terms] OR "adolescent"[MeSH Terms] OR "child, preschool"[MeSH Terms] OR "adolescent" OR "adolescence" OR "teen" OR "teenager" OR "youth" OR "children" OR "child" OR "childhood" OR "preschool child" OR "school child" OR "toddler" OR "pediatric") AND ("spinal dysraphism"[MeSH Terms] OR "neural tube defects"[MeSH Terms] OR "meningomyelocele"[MeSH Terms] OR "spinal cord injuries"[MeSH Terms] OR "spina bifida"[All Fields] OR "myelomeningocele" OR "spinal cord injury" OR "neural tube defect" OR "lipomyelomeningocele" OR "lipomeningocele" OR "spinal cord malformation") AND ("walking"[MeSH Terms] OR "mobility limitation"[MeSH Terms] OR "gait"[MeSH Terms] OR "dependent ambulation"[MeSH Terms] OR "walking"[All Fields] OR "mobility" OR "six minute walk test" OR "ten meter walk test" OR "6 minute walk test" OR "10 meter walk test" OR "stepping" OR "ambulation" OR "motion analysis" OR "walking speed" OR "walking difficulty")
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Appendix 2: Description of Included Studies
n with SB Walking status (aged 117 yrs)
Author & Year Populations Walking Measure Reason for walking tested measurement
Type of study
Level of lesion: n
Age: mean + 1SD (range) yrs
Adzick et al. 1 2011
MMC
WeeFIM
Prospective randomized cohort
Thoracic: 7 L1-L2: 31 L3-L4: 75 L5-S1: 45
Follow-up at 1 108 & 2.5 yrs
Agre et al. 2 1987
MMC
Self-selected & To examine Prospective max speeds over relationship between cross-sectional 30m speed, muscle strength & aerobic capacity
Al-Holou et al. 3 2009
MMC & Modified Hoffer To study success of LMMC with scale & Personal untethering surgery tethered cord NEM motor scale
Alman et al. 19964
SB
Hoffer Scale & speed (calculated from 6min of walking on 50m track)
To compare metabolic Prospective energy used in follow-up walking between those who had operative relocation of the hip and those treated conservatively
Ammerman et al. 19985
SB
Categorized as: no assistance, assistive devices (e.g., braces, crutches), wheelchair only
To examine reProspective lationship between survey ambulatory status & psychiatric diagnoses
Apkon et al. 6 2009
MMC
Categorized as: ambulatory, partially ambulatory, nonambulatory
Asher et al. 19837
MMC
Ambulation categorized as: community, household, nonfunctional, nonambulatory
To study factors Prospective affecting ambulatory cross-sectional status in children with SB
Banta et al. 19918
MMC & SCI
Speed during steady state walking (>3min walking duration)
Bartnicki et al. 9 2012
MMC
Hoffer scale
To compare walking while wearing a RGO to walking while wearing the Parawalker To determine if ambulatory function correlated with sitting stability
L3: 6 L3-L4 mixed: 12 L4: 34
Age of untethering 12.3 + 5.9 (1025.9)
Operated 14.5 + 5.8; Conservative 16.4 + 5.4
Not stated; 89 met inclusion
Modified Hoffer: n Normal: 15 Community: 8 Household: 11 Exercise: 9 Nonambulator: 40
Not Speed: Conservative = stated; 52 0.643 + 0.102m/s; total Operated = 0.775 + 0.126m/s
Category: n No assistance: 10 Assistive devices: 36 Wheelchair only: 6
9.75 (4-18)
24
Category: n Ambulatory: 10 Partially ambulatory: 2 Nonambulatory: 12
Thoracic: 28 L1-L2: 12 L3: 20 L4: 17 L5: 9 Sacral: 12
14.33 (5.75 31.83)
T12: 3
Ages of 3 children with SB: 10.83, 10.83, 6.58
Not Category: n stated; 98 Community: 20 (all total with L5 & all the sacral except 1) Nonfunctional: 40 (all those in thoracic-L2) *** All results not indicated 3 Not indicated
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Mean self-selected speed (km/hr): L2 & above: 1.9 L3-4: 3.5 L5-sacral: 3.9 No motor deficit: 4.8 Mean max speed (km/hr): L2 & above: 2.4 L3-4: 6.4 L5-sacral: 9.3 No motor deficit: 16.0
12.94 + 3.59 (6- 54 18)
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Sacral: 5 Lumbar: 23 Thoracolumbar: 9 Thoracic: 3 Other (occipital encephalocele, LMMC): 11 To examine Prospective Thoracic or high relationship between cross-sectional lumbar: 6 ambulatory status & Mid-lumbar: 9 bone mineral density Sacral: 9
Prospective case series
Prenatal surgery group had higher WeeFIM scores & were more likely to walk without orthotics
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Thoracic: 17 Lumbar: 61 Sacral: 6
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L2 & above: 6 12.6 + 1.2 (10- 33 L3-4: 7 15) L5-sacral: 17 No motor deficit: 3
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Compare motor development between prenatal & postnatal surgical repair of MMC
Prospective T12: 7 cross-sectional L1: 4 L3: 2 L4: 4 L5: 2
21.4 (13-35)
Not stated Hoffer: n Community: 6 Househould: 1 Non-ambulators: 12
Hoffer scale
Bartonek et al. MMC 1999b11
Hoffer scale, speed over 30 metres & speed over 3 minutes
Bartonek et al. MMC 12 1999c
Hoffer scale & Outcome looked at walking distance over time categorized as: >1,000 m, 100 1,000 m, 10 - 100 m, <10 m
MMC
Modified Hoffer scale, mobility domain of PEDI, PCI
Prospective cohort
L3: 3 L4: 3 L5: 5 Sacral: 21
To examine how Prospective Not stated motor paresis of cross-sectional lower limbs relates to ambulatory status
17.2 (5-40)
Not stated Hoffer: n Community: 41 Household: 14 Nonfunctional: 11 Nonambulators: 7
6-7 yrs
2
Hoffer: n Household: 1 2nd child could not be clearly classified into community or household 12-54 yrs Not stated Hoffer (baseline): n Community: 41 Household: 12 Nonfunctional: 7 Distance (baseline): n >1,000m: 35 100-1,000m: 10 10-100m: 4 <10m: 8 7.6 (3.2-11.4) 53 Results for PCI & PEDI broken down by muscle function score in Tables V & VI
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Comparison between Prospective Thoracic to sacral different cross-sectional classification systems depending on neurological level of impairment To compare Ferrari Case report Mid to low level KAFO's with lumbar AFO's
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Bartonek et al. MMC 1999a10
low & midlumbar Hoffer scale & self To compare outcomes Prospective selected walking with AFO & KAFO cross-sectional speed over 110m corridor
10.7 + 2.8 (513)
Bartonek et al. MMC 200515
Modified Hoffer scale
Sacral to midTo classify ambulatory Prospective status of participants cross-sectional lumbar
10.3 (6.8-17.6) 38
Hoffer: n Community: 38
Bartonek 201016
MMC
Modified Hoffer scale
Prospective Not stated Used to established an expected cross-sectional ambulation outcome based on muscle function class to guide orthotic program guidelines
Children 43 followed from birth until they reached 6 yrs
Walking achieved at 1year follow-up in 2/38 children, at 1.5-year follow-up in 7/39, at 2year follow-up in 14/36, at 3-year followup in 21/28, at 4-year follow-up in 28/36, at 6year follow-up in 30/38
Basobas et al. 200317
MMC, SCI, Ambulation myopathies, categorized as: CP community, household, nonambulatory SB FIM
To assess outcomes of Retrospective anterior spinal fusion cohort with anterior instrumentation surgery As an outcome Case report measure for a membrane based biofeedback machine
Not stated
10.42 (1.5818.75)
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Category: n Community: 9 Household: 6 Non-ambulators: 6
Not stated
12.5
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Mobility FIM=1 at baseline, after training score improved to 6
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Battibugli et al. MMC 200719
FMS (rates walking on 6point ordinal scale at 3 distances: 5, 50 & 500m)
To compare outcomes Retrospective in children with SB cohort who do & who do not have a shunt
Sacral: 118 Low lumbar: 31 Thoracic/high lumbar: 12
Participants 161 with no shunt: 9.92 + 3.92 (4.75-17.75) Participants with a shunt: 10.17 + 3.92 (4.83-18.08)
Benzer et al. 201220
Ambulation categorized as: Independent, assisted, nonambulatory
To evaluate influence Retrospective of ambulatory status cohort on renal functions, clinical & radiological findings
Thoracic: 3 Lumbar: 80
7.1 + 0.61
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Hoffer: n Community: 4 Household: 4
FMS: 5 meters: means of 5.05 (no shunt), 4.88 (with shunt) 50 meters: means of 4.97 (no shunt), 4.60 (with shunt) 500 meters: means of 4.84 (no shunt), 4.15 (with shunt) Category: n Independent: 8 Assisted: 14 Nonambulatory: 61
MMC
Buffart et al. 22 2008
MMC
Modified Hoffer scale
To examine Prospective relationship between cross-sectional ambulatory status & physical activity, aerobic fitness, obsesity
Thoracic: 2% 21.1 + 4.5 Thoracic-lumbar: 14% Lumbar: 29% Lumbosacral: 41% Sacral: 14% *** exact numbers not stated
Not Modified Hoffer: n stated; 51 Community: 15 total Household: 8 nonfunctional: 28
Carbonari de Faria et al. 23 2013
MMC
Hoffer scale
To compare Prospective neuromotor cohort development (including gait) between children who did & did not undergo intrauterine MMC repair
Thoracic: 3 Upper lumbar: 1 Lower lumbar: 5 Sacral: 4
13
Chang et al. 24 2008
MMC & LMMC
Modified Hoffer scale
3.84 (3.75 to 4.41)
Not WeeFIM's motor stated; 34 component not a total significant contribution to quality of Life ***specific WeeFIM results not reported
Hoffer: n Community: 6 Nonambulator: 1 Not all ambulation data were reported
Prospective Thoracic: 1 cross-sectional High lumbar: 3 Midlumbar: 9 Lumbosacral: 28 Sacral: 14 No neurological deficit: 23
15.24 + 6.74
Prospective T12 to L3-4 cross-sectional
RGOs: 26 n=15, 8.1 (3.715.0) HKAFO: n=11, 9.0 (6.111.8)
Speed (m/s): RGO: 0.27 + 0.11 HKAFO: 0.68 + 0.20
Follow-up walking assessment done at 2 years; exact mean age of group not reported
Category: n Community: 9 Household: 4 Nonfunctional: 16
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To describe clinical features & their correlates within children in Taiwan with spinal dysraphism as a clinical guide for future management Speed measured To compare energy during each efficiency in children minute of with MMC ambulating assessment of with either RGO or HKAFO energy consumption during walking Ambulation To determine the categorized as: prognostic value of community, neurophysiological household, investigations nonfunctional compared to clinical neurological exams in children with SB
13 + 6 (4- 27)
L1-L2: 3 L3-L4: 15 L5-Sacral: 10
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To identify factors related to quality of life
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Cuddeford et al. MMC 25 1997.
WeeFIM
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Bier et al. 200521
Cuppen et al. 26 2013
SB
Danzer et al. 27 2009
MMC
Categorized as: independent walkers, assisted walkers (walk with appliances), nonambulatory (wheelchair bound)
To examine lower Retrospective High (T-L2): 10 extremity neuromotor cross-sectional Mid (L3-4): 24 function & Low (L5-S1): 20 ambulatory potential in infancy & early childhood of children who had midgestation MMC closure
5.58 + 1.52 (3- 54 9.42)
Category at follow-up: n Independent: 37 Assisted: 13 Nonambulatory: 4
Danzer et al. 201128
MMC
WeeFIM & Ambulation categorized as: independent, assisted, wheelchair dependent
To describe potential Prospective functional limitations cohort & to evaluate risk factors associated with impaired functional status in children that underwent fetal MMC closure
5 years of age, 26 but exact mean, SD & range not reported
WeeFIM (mobility domain): Functional independence in 62% Ambulation status: n Independent: 21 Assisted: 5
Anatomical level of spine: Thoracic: 7 Lumbar: 27 Sacral: 2 Motor impairment level: Thoracic: 12 Lumbar: 16 Sacral: 8
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Prospective cohort
Not Modified Hoffer: n stated; 78 Community: 56 total Household: 8 Nonambulators: 14 *** Results further broken down by MMC & LMMC in Table 1
Not stated
29
6MWT, speed (calculated during 6MWT), Hoffer scale
De Groot et al. SB 30 2009
De Groot et al. SB 31 2010
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To explore the Retrospective relationship between cross-sectional VO2peak & functional ambulation
10.4 + 3.1 (617)
23
6MWT, Modified 6MWT performed to Prospective Hoffer scale determine cohort individualized protocol for VO2max test
10.3 + 4.9
20
Speed (calculated To determine the during 6MWT) & reproducibility of Hoffer scale gross & net energy expenditure during gait in ambulatory children & adolescents with SB 6MWT & To evaluate the Prospective Not stated Modified Hoffer effects of treadmill Randomized scale training program controlled trial compared with usual care in ambulatory children & adolescents with SB
10.8 + 3.4
14
L5-S1: 10 S1-S4: 6 No motor loss: 7
AIS level: L3-L4: 2 L4-L5: 7 L5-S1: 6 S2 and below: 1 No motor loss: 4 Prospective AIS level: cross-sectional L3-L4: 1 L4-L5: 10 S1-S4: 1 No motor loss: 2
Control group: 32 11.1 + 2.6 Intervention group: 10.3 + 2.9
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MMC
6MWT & Modified Hoffer scale
To assess the Prospective reliability & psychometric agreement of study maximal & submaximal exercise measures in normal ambulatory & community ambulatory children & adolescents with SB
De Souza & 34 Caroll 1976
MMC
Hoffer scale
To identify factors that determine ambulatory status
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Categorized as: To determine long-distance influence of muscle ambulators, short- strength on distance ambulatory status ambulators, nonfunctional ambulators Speed (measured To compare energy during cost of walking assessment of between children with oxygen CP & children with SB consumption)
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Dudgeon et al. MMC 199135
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Duffy et al. 199636
CP & SB
Evaggelinou & Drowatzky 199137
SB
Categorized as: ambulatory, household-only, nonambulatory
L3-L4: 2 L4-L5: 10 L5-S1: 6 S2 and below: 5
Prospective Thoracic: 4 cross-sectional Upper lumbar: 19 Lower lumbar: 13 Sacral: 32 Retrospective chart review
Not stated
Prospective L3-L4: 11 cross-sectional L5-S1: 10
To compare Prospective informationcross-sectional processing capabilities among children with differing walking abilities
T11: 1 T12-L1: 1 L1: 2 L1-L2: 2 L3: 5 L3-L4: 2 L4: 3 L4-L5: 1 L5: 2 Saccral: 3 Occulta: 1
Hoffer: n Normal: 17 Community: 6 6MWT: mean distance + 1SD for group = 391.4 + 61 m 6MWT: mean + 1SD distance walked for group = 418 + 95 m
Hoffer: n Normal: 8 Community: 6 Speed: Group mean + 1SD = 70.0 + 13.8 m/min
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10.7 + 3.5
23
Hoffer: n Normal: 9 Community: 23 Baseline 6MWT distance: Control group: 372.1 + 116.5 m Intervention group: 344.8 + 125.3 m Hoffer: n Normal: 10 Community: 13 6MWT: Mean + 1SD at baseline for group = 406.8 + 90.7 m
Not reported, Not Hoffer: n recruited ages stated; 68 Community: 23 12 & up total Household: 15 Nonfunctional: 23 Nonambulators: 7 14.33 (6Not Category: n 27.75) stated; 83 long-distance: 25 total short-distance: 26 nonfunctional: 32
L3/L4: 8.5 (5- 21 12) L5/S1: 8.9 (512)
Speed: group means L3/L4: 40.7 m/min L5/S1: 51.7 m/min
8-18
Category: n Ambulatory: 8 Household-only: 6 Nonambulatory: 7
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WeeFIM
To determine whether Prospective longer waiting times cohort for rehabilitation were associated with change in child functional status
Findley et al. 198739
MMC
Hoffer scale
To identify walking ability in adolescents with SB
Findley & Agre MMC 40 1988
Self selected & maximal speed (measured over 30m distance)
To determine whether Prospective energy cost of cohort mobility, relative level of effort during mobility, & speed at which one exerted 70% of maximal aerobic power could be estimated from measures easily obtained in the clinic
Franks et al. 199141
MMC
PCI
GerritsmaBleeker et al. 199742
MMC & paraplegia
Thoracic: 14 Upper lumbar: 4 Lower lumbar: 16 Sacral: 19 No weakness: 10
13 (11-16)
77
WeeFIM Severity Category: Mild to none (total score >75): 64 Moderate (total score 50-75): 52 Severe (total score <50): 8 Hoffer: n Community (never use wheelchair): 30 Community (occasionally use wheelchair): 26 Household: 0 Therapeutic: 1 Wheelchair only: 20 Self selected speed reported by impairment level (mean+1SD m/min): L2 & above: 32 (n=1) L3-L4: 58 + 10 L5-sacral: 65 + 5 No motor deficit: 80 + 12
Neurological level 10-15 yrs defined by lowest root level with grade 3 strength on MMT L2 & above: 1 L3-L4: 5 L5-sacral: 16 No motor deficit: 3
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Prospective case series
L4: 2 L5: 1
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PCI values = 1.46, 2.15, 2.06
Retrospective chart review
L1: 6 L2: 1 L3: 6
4.8 + 2.0
13
Categorized as: Community, independent indoors, nonfunctional
Retrospective cohort
Not stated
11 (4-22) for 6 entire group (age of children with MMC not reported) 4.3 (1-10.2) at 5 first visit; 20 (16.1-24.7) at follow-up
Hoffer: n Community: 3 Household: 8 Nonfunctional: 2 Category: n Independ. indoors: 3 Nonfunctional: 3
Lumbosacral Hoffer's scale agenesis &/or MMC
To assess type & Retrospective extent of spinal review of charts malformation, degree & radiographs of lower extremity involvement & ambulatory capacity over time, in order to predict ambulatory potential & develop guidelines for orthopedic management
motor level: "flail lower extremity": 2 L2: 2 L3: 1 L4: 1
Hassan et al. 201045
hemophilia, juvenile idiopathic arthritis, MMC & LMMC
6MWT
To compare 6MWT Prospective values of pediatric cohort patient populations to reference values, & assess differences in predicted distances
L5 and below: 22
10.3 + 3.1 (618)
22
6MWT for group: 391 + 61 m
Hisaba et al. 201246
MMC
Hoffer's scale
To evaluate outcomes Retrospective of intrauterine cohort surgery
Anatomical levels: T12-L1: 1 L1-L2: 1 L2-L3: 2 L4-L5: 2 Functioal levels: L1-L2: 1 L4-L5: 3 S1-S2: 2
Followed prenatally to 3.5 yrs
6
Hoffer: n Community: 3 Household: 2 Nonambulator: 1
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Guille et al. 44 2002
To assess effects of surgery to correct knee & hip flexion deformity
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Prospective cohort
3.77 + 1.11 (2- 124 9)
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Hoffer: n (lumbosacral agenesis & MMC): Nonambulator: 5
To study factors associated with walking ability
MMC
Walking categorized as: independently, with crutches & orthoses, with crutches without orthoses, wheelchair only
To evaluate long-term Retrospective clinical effects after cohort having performed a Grice arthrodesis of a valgus unstable foot
MMC
Categorized as: good (can walk >20 yards with or without aids), poor (walking only a few steps or having some function in legs), no mobility (no useful walking)
To analyze severity of Prospective disability relative to cohort numerour factors (e.g., sensory level, presence or absence of neural plaque, motor activity of the legs)
Ilharreborde et DMD, CP, SB, polio-myelitic al. 200950 or traumatic paraplegia, SMD, scoliosis
Categorized as: walkers, walk with crutches, nonwalkers
To examine the effect Retrospective Not stated of long spinal fusion review of charts on functional status & radiographs
Jaworek et al. 201351
MMC
To evaluate the mobility & quality of life after surgical treatment
Kaiser & Rudeberg 52 1986
MMC
Categorized as: community ambulation, ambulation with crutches, ambulation with wheelchair, nonambulation Ambulation categorized as: Without aids/with short irons, With calipers, Wheelchair
12.8 + 3.9 (517)
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Hoffer scale & functional tests (free-walking 24.4 m on a level surface, descending 20 steps, ascending 20 steps) Speed & PCI
Hoffer: n Community: 10 Household: 2 Nonfunctional: 2 Non-ambulators: 20
Category: n Independently: 16 (7 of whom used orthoses) With crutches & orthoses: 3 With crutches without orthoses: 1 Wheelchair only: 3
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High sensory level 1.25-7.67 (T5-T10): 33 Intermediate (T11L3): 28 Low (L4 and below): 18
Age at operation: 15.3 ± 2.6
Prospective High hernia (T6-L1): 9.8 (2-16) cross-sectional 11 Low hernia (L2-L5): 8
To compare outcomes Prospective between children who cohort did & did not meet Lorber's criteria for surgery
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Katz et al. 199754
of lesion based on preserved sensorimotor function: Thoracic: 8 Upper lumbar: 11 Lower lumbar: 11 Sacral: 4 Lumbosacral
80
Category: n Good: 30 Poor: 33 No mobility: 16
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chart review, longitudinal cohort
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Hoffer scale (original article)
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MMC
Not stated
Not stated
Not Preop category: n stated; 56 Walkers: 11 total Walk with crutches: 6 Nonwalkers: 39
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Category: n Community ambulation: 1 Ambulation with crutches & ambulation with wheelchair: 17 Nonambulation: 1
34
Category: n Without aids/with short irons: 13 With calipers: 12 Wheelchair: 4 Too young for assessment: 5 Hoffer: n Community: 4 Results for functional tests not reported
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Hoffer et al. 197347
To describe the sample & to examine the functional carryover of electical stimulation.
Prospective, L2-3 cohort, single group pretest posttest design
ages in yrs (n): 4 5(2), 12(2), 21(1)
To evaluate & compare metabolic cost of walking with RGO and HKAFO
Prospective cohort
6.83 (2.1711.75)
Functional level: Thoracaic or thoracolumbar: 4 High lumbar: 4
8
Mean speed (m/min): RGO: 14.6 HKAFO: 11.9
SB, UMN pathology, nemaline myopathy
ACCEPTED MANUSCRIPT Prospective, L3-4
Functional tests To compare 2 types of (timed): KAFOs (PM vs LM) ascending & descending 5 stairs, ascending & descending a ramp, rising from the floor & ambulating 20m
cohort, longitudingal quasiexperimental cross-over design
FAC, WeeFIM, Speed
To describe the sample with respect to their virtual rehabilitation game performance
Prospective not stated cross-sectional
Lemelle et al. 57 2006
MC, MMC
Categorized as: wheelchair-bound (inside & outside), wheelchair-bound (outside), stand & walk with walking brace, walk with minor aid
To examine Prospective Not stated relationship between cross-sectional walking ability, incontinence, & HRQOL
Lewis et al. 58 2004
MMC
Ambulation categorized as: independent, with assistance, wheelchair-bound
To compare longterm Retrospective motor outcomes & cohort ambutatory status at ages 2 & 10 yrs between children born by elective cesarean section or vaginal delivery (labour)
Lorente Molto & Garrido 59 2005
MMC
Hoffer scale
To evaluate the Retrospective relationship between cohort hip stability & walking ability during early childhood, adolescence & adulthood
Not Stairs (s): stated; 12 PM: 30.0 + 13.4; total LM: 34: + 14.5 Ramp (s): PM: 36.2 + 40.5; LM: 30.9: + 34.7 From floor (s): PM: 17.8 + 25.5; LM: 21.5 + 36.4
13.4 + 3.6 (5.5- 1 19.0)
FAC level: n 5: 11 4: 6 3: 1 1: 1 WeeFIM: mean 89 + 8 Speed: mean 1.6 + 0.3 km/hour 124/160 were able to walk; specific numbers not included
14.4 + 1.84
160
EP
TE D
M AN U
SC
Labruyere et al. MMC, CP, CVA,TBI, 201356 spastic ataxia, CNS demyelination
Whole group at baseline: 9.7 + 2.7 (521)
RI PT
Krebs et al. 198855
Anatomical level: Cesarean group: L2 ± 2.2 Labour group: L3 ± 1.4 Motor level: Cesarean group: L2 ± 2.0 Labour group: L3 ± 1.2 L3: 29
data collected 87 from participants' files at ages 2 & 10
Category at age 2: n Independent: 15 With assistance: 56 Wheelchair-bound: 16 Category at age 10: n Independent: 11 With assistance: 36 Wheelchair-bound: 40
Age at time of 29 surgery: 2.21 (0.92-6) Records reviewed at 25 yrs & 13-18 yrs; interviewed as adults
Hoffer: n At 2-5 yrs: Community: 21 Household: 4 Non-functional: 4 Non-ambulation: 0 At 13-18 yrs: Community: 19 Household: 3 Non-functional: 3 Non-ambulation: 4 Self-selected speed (m/min): Swivel mean: 7.23 + 0.73 Parapodium mean: 9.18 + 1.88 Maximal speed: Swivel mean: 14.78 + 2.06 Parapodium mean: 21.63 + 3.70 Hoffer at baseline: nonfunctional; Hoffer with orthosis: community
Self selected & maximal speed
To compare Prospective ambulation between cross-sectional Parapodium & ORLAU swivel modification
T10: 2 T11: 1 T12: 2 L1: 2 L2: 2
6.1 + 1.31 (49)
9
Magee & Kenny Myelodysplasia 200861
Hoffer scale
To describe efect of Strutter Orthosis on mobility
L3-L4: 1
12 years
1
AC C
MMC & Lough & Nielsen 198660 traumatic paraplegia
Prospective case report
ACCEPTED MANUSCRIPT Motor level
113
Categorgy: n Full walkers: 61 Partial walkers: 13 Nonambulatory: 36 Below age of walking: 3
McCall & MMC, CP, Hoffer & Maximal Follow-up measure of Prospective Thoracic: 20 63 cross-sectional Upper lumbar: 13 Schmidt 1986 osteo-genesis spped (over 30m) children who were imperfecta, prescribed an RGO Lower lumbar: 7 arthrogryposis, paraplegia, polio, SMA, microcephaly
1.25-16
41
Hoffer: n Community: 24 Household: 8 Therapeutic: 5 Nonambulators: 4 Max speed with RGO: group mean=0.52 + 0.22m/s
McDonald et al. MMC 64 1991
Categorized as: nonabmulatory, partial, community (according to Shurtleff et al. 1989)
Not stated
Moerchen et al. SB 65 2011
FMS & PEDI
Moore et al. 200166
MMC
Speed
Müller et al. 1992a67
MMC
Modified Hoffer scale
Müller et al. 68 1992b
MMC
Modified Hoffer To assess the Prospective scale (see Table 1 outcomes of scoliosis cohort in the paper for surgery customized scoring)
Müller et al. 199469
MMC
SC
Prospective Not stated cross-sectional
M AN U
To determine the degree to which specific muscles influence mobility, & to document the natual history of mobility amongst those with different patterns of strength To evaluate a homebased treadmill intervention To compare the efficiency of reciproccal gait or swing-through gait
(International Myelodysplasia Study Group criteria): Thoracic: 6 Upper lumbar: 22 Lower lumbar: 42 Sacral: 43
Prospective case report
L4-L5: 1
Prospective L3-4: 13 cross-sectional
To investigate the use Prospective of Boston bracing to cohort treat scoliosis in MMC
AC C
MüllerMMC Godeffroy et al. 200870
RI PT
10.67 + 4.83 (0.5-18)
To examine the Retrospective relationship between cross-sectional ambulatory status & incidence of fractures
TE D
Modified version of International Myelodysplasia Study Criteria Manual ambulation categorized as: nonambulatory, partial, full, below age of walking
Not Category: n stated; Community: 139 190 total Partial: 30 Nonambulators: 61
1.5
1
MMC group: 17.1 (12-24) Control: 14.4 (12-23)
14
Thoracic: 1 10.2 + 3.1 (5- 20 Upper lumbar: 8 14) Lower lumbar (L4 & below): 11
Not stated
EP
Marreiros et al. SB 201062
Hoffer scale
To understand the Retrospective Not stated natural history of cross-sectional scoliosis in individuals with MMC
Categorized as: Community, household/ near environment, wheelchair user
To identify factors associated with HRQOL
Surgery was 14 performed at a mean age of 12.4 (9- 16) yrs, at time of follow-up mean age of the children was 15.6 (1 020) yrs 15.7 (6-27)
Prospective Thoracic (T10- L2): 9 12.08 + 2.33 cross-sectional Lumbar (L3-4): 11 (8-16) Sacral (L5-S2): 25
Mobility component of PEDI: 4/25 FMS: 0 Speed(m/min): Reciprocal gait: 40.49 + 14.21 Swing-through gait: 53.95 + 12.81 Modified Hoffer at baseline: n Community: 0 Household: 4 Nonfunctional: 14 Nonambulators: 2 Mean Modified Hoffer score: preop = 2 (nonfunctional), postop = 1 (nonambulator)
Not Incomplete data stated; 64 presented (Hoffer data total reported for n=16 in Table 4) 45
Community: 21 Household/near environment: 17 Wheelchair user: 7
ACCEPTED MANUSCRIPT Prospective Thoracic: 6
Categorized as: level A (fully independent & walking with no support); level B (independent but requiring support); level C (dependent but upright & standing in parallel bars or walking with a "drag through" gait); level D (confined to wheelchair) PEDI & Hoffer scale
To utilize muscle power at birth to predict ambulatory status later in childhood
3-8
95
Thoracolumbar: 13 Lumbar: 36 Lumbo-sacral: 29 Sacral: 11
OkurowskaZawada et al. 201173
MMC & CP
Hoffer scale
To describe the motor Prospective Level was lowest 10.85 + 3.75 (5- 34 function of cross-sectional level on better side 16) participants at which child could perform antigravity movement through available range: Thoracic: 13 Lumbar:17 Sacral: 4
Ozaras et al. 200574
SB aperta, MMC
Categorized as: non-ambulatory, therapeutic ambulation, household ambulation, functional ambulator
Ambulation was one Prospective component of cohort assessment to identify children with SB who showed signs of CP
Upper thoracic: 11 4.99 + 3.44 Lower thoracic: 6 Range not included Upper lumbar: 5 Lower lumbar: 6
28
Hoffer scale
To identify factors that could predict future mobility
Thoracic: 5 Thoracolumbar: 9 Lumbar: 43 Lumbosacral: 23 Sacral: 10
10.6 + 6.25 (1.4 to 26.7)
Not Hoffer: % sample stated; 90 Community: 42% total Household: 16% Exercise: 16% Wheelchair dependent (nonwalker): 27%
To assess Prospective predictability of post- cohort natal function using an in utero reflex technique in fetuses
Low sacral lesions
Ambulation 8 assessed at 35 yrs
Walk with no or min assistance: 3 Walk with assistance: 1 Unable to walk: 4
To describe ambulation in children who underwent triple or double muscle transfers
Level based on voluntary control of knee/ankle & sensation: L3: 7 L4: 28 L5: 12 T10: 3 T12: 8 L1: 3 L3: 1 L4: 6
Age at follow- Not up: stated; 47 9.75 (4.67total 20.25)
Follow-up data reported for 41/47 (baseline data not reported): Community: 37 Household: 4
7 (1.67-14.5)
Hoffer while using orthosis: n Community: 13 Household: 8
Phillips & MMC 77 Lindseth 1992
Phillips et al. 78 1995
M AN U
TE D
EP
Retrospective cohort
AC C
SB
MMC
Categorized as: walk with no or min assistance, walk with assistance (walker), unable to walk Hoffer scale
Hoffer scale
8.7 (6-11)
32
SC
MMC
Petrikovsky et al. 200176
Prospective Not stated cross-sectional
Level A: 22 Level B: 48 Level C: 20 Level D: 5
Norrlin et al. 72 2003
Pauly & Cremer MMC 75 2013
As an outcome of neurological impairment
cohort
RI PT
Murdoch et al. MMC & myelocele 197971
Retrospective cohort
To describe Retrospective ambulatory status of cohort children with RGO or hip guidance orthosis
21
Hoffer: n Community: 11 Household: 6 Nonfunctional: 9 Nonambulator: 6 PEDI (caregiver assistance, group mean): 76.7 + 18.3 Hoffer: n Community: 3 Household/near environment: 10 Wheelchair user: 21
Therapeutic: 8 Nonambulatory: 20
CP, MMC, HSP
Self-selected To compare measure speed (10MWT) & of speed obtained 10-min WT with 10MWT & 10min WT (Part 1), & to assess repeatability of 10-min WT (Part 2)
Rose et al. 80 1981
SB
Modified Hoffer scale, speed measured over 20 feet distance
Ross et al. 81 2007
LMMC, MC, Hoffer scale intradural lipoma, filum terminale lipoma, split cord malformation, dermal sinus, caudal regression syndrome
To describe Retrospective ambulatory status in a cross-sectional large group of children with closed NTD
Samuelsson & 82 Skoog 1988
MMC
To classifiy participants by ambulatory ability
SC
0.9-22.9; Not Hoffer: n mean & 1SD stated; Community: 98 age reported 104 total Household: 1 for each type Nonambulators: 5 of NTD in Table 4
Prospective cohort
Hoffer scale, step To evaluate effects of Prospective distance/ length, orthotic treatment cohort maximum nonstop walking performance & speed as measured on a scale of 1 to 5
thoracic: 35 L1-L2: 5 L3: 26 L4: 32 L5: 20 Sacral: 45 Not stated
14.83 (2-40)
Not Hoffer: n stated; Community: 63 163 total Household: 21 Nonambulators: 51
5.17 (1.1732.92)
Not Results are reported stated; 67 only as % of total participants in each bracing group. E.g., 60% of patients with Ferrari type orthosis walked for >50 minutes & with stepping distances >40cm were seen 25 with Hoffer: n MMC or Normal: 13 LMMC (44 Community: 1 total) Household: 1 Wheelchair dependent: 4 Too young: 5 44 Modified Hoffer: n MMC: Normal: 14 Community: 4 Household: 2 Nonambulant: 1 Too young: 9 LMMC: Normal: 9 Community: 4 Household: 1
AC C
EP
Schiltenwolf et SB 83 al. 1991
T9: 1 T10: 3 T11: 5 T11/12: 1 T12: 6 T12/L1: 1 L1: 1 L1/L2: 2 L2: 3 L2/L3: 1 L3: 1 L5: 2 Not stated
Part 1: 8 Whole group results for Part 2: 11 speed (m/s): Part 1: 10MWT: 1.00 + 0.3 (0.15-1.61) 10-min WT: 0.87 + 0.24 (0.30-1.34) Part 2: 10-min WT #1: 0.97 + 0.26 (0.16-1.31) 10-min WT #2: 0.98 + 0.25 (0.17-1.33) 9.7 + 2.7 (5.67- 27 Baseline data 15.58) Hoffer: n Community: 2 Household: 6 Therapeutic: 18 Chairbound: 3 Speed (ft/min): 26.0 + 15.2
Part 1: 12.4 + 3.9 (4-28) Part 2: 11.5 + 3.5 (6-16)
M AN U
To determine whether Prospective the hip guidance cohort orthosis improved ambulatory status
TE D
Hoffer scale
cohort & prospective cohort
RI PT
ACCEPTED MANUSCRIPT Retrospective Not stated
Pirpiris et al. 200379
Schoenmakers et al. 200384
Tethered Cord Syndrome (MMC & LMMC)
Modified Hoffer scale
As an outcome Prospective measure for the Cohort Study effectiveness of tethered cord release surgery
High level (above L4): 5 Low lumbar: 6 Sacral: 2
Age at operation: 6.17 + 5 Long term followup: 13.58 + 5.5
Schoenmakers et al. 200485
MMC & LMMC
Modified Hoffer scale, MABC & Dutch Version of PEDI
To investigate Prospective functional outcome in cross-sectional children with sacral level MMC and LMMC
S1 or Below: all participants MMC: 30 LMMC:14
MMC: 6.0 + 4.9 (0-18) LMMC: 8.4 + 4.9 (0-18)
Modified Hoffer scale, Dutch Version of PEDI
Schoenmakers et al. 200988
Lumbosacral 6MWT & MMC & Modified Hoffer LMMC scale
Seitzberg et al. MMC 200889
MMC
Stallard et al. 91 1991
MMC
Stillwell et al. 92 1984
SB
Hoffer scale
Suson et al. 201093
SB occulta, MMC, LMMC & sacral agenesis
Swank & Dias 199294
MMC
Categorized as: ambulates fully, ambulates with devices, ambulates minimally with devices, wheelchair bound Hoffer scale
Swank & Dias 199495
SB
To predict adult Retrospective ambulation status Cohort Study based on ambulation status between ages of 5-8 yrs
To compare the effect Prospective of surgical versus non cross-sectional surgical treatment of hip dislocation
10
23
Modified Hoffer (in children >2.5yrs): n Nonambulators: 53 Ambulators: 50 Modified Hoffer: n Presurgery: Nonambulatory: 6 Exercise: 3 Household: 1
Modified Hoffer: n Normal: 10 (MMC), 7 (LMMC) Community: 6 (MMC) 6MWT means (m): 353 + 108 (MMC), 424 + 65 (LMMC)
At age 5-8 yrs: At or above L2: 1 L3: 7 L4: 8 L5: 8 At or below S1: 8
5.0-8.0
52
L3: 22 L4: 6 L5:1 S1:1
Non41 operative: 10 (4-19) Operative: 6 (216) SD not included
Category: n Wheelchair user: 14 Community ambulator with walking aid: 11 Community ambulator without walking aid: 27
Category: n Household: 1 Community (unable to use stairs): 6 Community (able to use stairs): 23
Hoffer: n Community: 18 Household: 22 Therapeutic: 12 Not Hoffer: n stated; 47 Community: 32 total Household: 3 Nonambulatory: 12
To identify ambulatory abiliy of patients with omphaloceleexstrophyimperforate anusspinal defects
Not Category: n stated; 68 Ambulated fully: 37 total Ambulated with devices: 15 Ambulated minimally with devices: 2 Wheelchair bound: 8
AC C Modified Hoffer scale
122
To measure Prospective Complete lesions L1- Up to 15 yrs performance with the cross-sectional T4 (specific numbers not ParaWalker orthosis reported) To determine the Prospective L2: 3 (10->25) number of individuals cohort L3: 16 who were walking 10 L4: 15 yers after iliopsoas L5: 13 transplantation
EP
Sherk et al. 90 1991
Categorized as: wheelchair user, community ambulator with walking aid, community ambulator without walking aid Ambulation categorized as: exercise, household, community (unable to use stairs), community (able to use stairs) Hoffer scale
7.9 + 5.2 (1cross-sectional L1-3: 26 18) L4-5: 45 Sacral: 26 To evaluate the Prospective T11: 3 9.3 + 2.4 effectiveness of spinal cross-sectional T12: 2 fusion surgery L2: 1 L3: 1 L3-4: 1 L5: 1 S1: 1 To identify Prospective AIS level: 6-18 associations with cross-sectional MMC: muscle strength, L5-S1: 7 aerobic capacity & S1-S4: 5 physical activity in No Motor Deficit: 4 independent, LMMC: ambulatory children L5-S1: 3 with lumbosacral SB S1-S4: 1 No Motor Deficit: 3
RI PT
Shoenmakers et MMC 87 al. 2005b
ACCEPTED MANUSCRIPT Prospective Thoracic: 26
To identify determinants of HRQOL
SC
Modified Hoffer scale, Dutch version of PEDI
M AN U
MMC
TE D
Schoenmakers et al. 2005a86
Retrospective Not stated cross-sectional
17 (0.5-34)
52
To identify prognostic Retrospective indicators of future review walking ability
Thoracic: 71 Lumbar: 73 Sacral: 62
5.11 (0.08-13) 206
To develop a model that would predict independence in walking
Thoracic (L3 or above): 60 Lumbar (L3-5): 81 Sacral: 63
Followed from 206 birth to a mean age of 9.58
Prospective cohort
Hoffer: n Community: 118 Household: 41 Nonambulators: 57 Hoffer: n Community: 118 Household: 36 Nonambulatory: 25
ACCEPTED MANUSCRIPT Thoracic: 1
Tezcan & Simsek 201396
CP & SB
Ambulatory classification according to Flanagan 2011
To investigate the Prospective relationship between cross-sectional HRQL & numerous factors including level of ambulation
9.25 + 3.82 (5- 70 Thoraco-lumbar: 16 18) Lumbar: 47 Lumbo-sacral: 5 Sacral: 1
Thomas et al. 97 2001
MMC
Classification by Schopler & Menelaus 1987, speed
To identify differences Prospective in walking speed & Cohort oxygen cost between walking with RGOs vs HKAFOs
T12: 4 L1: 4 L2: 3 L3: 10 L3-4: 2
Video analysis & questionnaire to assign score on ordinal scale: 1=primarily nonambulatory; 2=primarily ambulatory, uses wheelchair for distances >100 feet; 3=primarily ambulatory, does not use wheelchair in community; 4=always ambulatory, no diagnosis
To examine the Prospective Not stated relationship between cross-sectional lower extremity bone mineral density & level of ambulation
Categorized as: independent, partial (walking with crutches or walkers), nonambulatory PEDI & ambulation categorized as: independent, With walking aid, Nonambulant
To correlate ambulatory status with EMG findings
Tsai et al. 100 2002
MMC & LMMC
van den BergEmons et al. 2001101
MMC
Hoffer scale
Verhoef et al. 102 2004
SB
Modified Hoffer scale
RI PT
SC
6-13
M AN U
MMC & LMMC
Prospective Thoracic: 1 cross-sectional L1-2: 1 L3: 1 L4-S1: 19 S2-4: 12 No deficit: 13 To investigate Prospective No deficit: 15 functional cross-sectional Thoracic: 1 performance, & to L1-2: 3 correlate ambulatory L3: 2 status with PEDI score L4-S1: 24 S2-4: 18
4.4 (0.5-12)
47
Category: n Independent: 36 Partial: 2 Nonambulatory: 9
MMC = 4.60 + Not 3.18 (.58-12) stated; 63 LMMC= 3.81 + total 2.97 (.5- 11)
Mobility domain of PEDI: Norm standard scores MMC: 25.8 + 19.5 LMMC: 49.1 + 14.1 Scaled scores MMC: 57.6 + 28.3 LMMC: 72.7 + 29.5 MMC ambulatory status: n Independent: 6 With aid: 1 Non-ambulatory: 3 LMMC ambulatory status: n Independent: 30 Non-ambulatory: 2 To measure extent of Prospective Thoracic/lumbar: 2 18 + 4 (14-26) Not Hoffer: n hypoactivity in cross-sectional Lumbar: 3 stated; 14 Community: 3 adolescents & young Lumbar/sacral: 8 total Household: 6 adults with MMC with Sacral: 1 Nonambulators: 5 an activity monitor
AC C
EP
Tsai et al. 99 2001
TE D
Thompson et al. SB, CP, muscular 200098 dystrophy, spinal cancer, TD children
RGOs: 8.0 (4-15) HKAFO: 8.17 (5-13)
Independent ambulation with no assistive devices: 3 Independent ambulation with assistive device: 27 Ambulating with assistive device at home & wheelchair in community: 23 Wheelchair bound: 17 23 Category: n RGO: Community: 7 Household: 7 HKAFO: Community: 9 Speed: data over time reported in Table 2 9 with SB Category: n Level 1: 5 Level 2: 4 Level 3: 7 Level 4: all TD children
L2 & above: 73 To describe secondary Prospective health conditions in a cross-sectional L3-5: 68 Dutch adolescent S1 & below: 38 group with SB aperta & occulta
20. 75 + 2.92 (16-26)
Not Hoffer: n stated; Normal: 64 179 total Community: 28 Household: 17 Nonfunctional: 9 Nonambulatory: 61
PAS (ordinal scale), WeeFIM
As an outcome measure for treatment with nighttime electrical stimulation
Dutch version of SB, neuroWassenbergthe PEDI Severijnen et al. metabolic 105 disorders, 2003 osteo-genesis imperfecta, encephalopathy Williams et al. 106 1983
Myelodisplasia
Self selected & fast speeds over a level, 60.5m oval track
Williams et al. 107 2005
CP, SB
TUG
Winchester et al. 2002108
CP, ABI, SB, GMFM & speed DS & autism (10MWT)
Yngve et al. 109 1984
MMC, muscular dystrophy, SCI, CP, MS, spinal cord embolism or arterovenous malformation, LMMC
To establish the Prospective reliability of the Dutch psychometric version of the PEDI study
To document energy requirements for ambulation & wheelchair propulsion, & to compare these values with those of TD children To investigate the reliability & validity of the TUG in children with and without disabilities To evaluate a hippotherapy program
12.5 + 2.7 (716)
Thoracolumbar: 2 7 (4-12) Low lumbar/lumbosacral: 5
Not stated
Prospective Thoracic: 1 cross-sectional Lumbar: 11 Sacral: 3
80
7
Hoffer: n Community: 21 Household: 5 Therapeutic: 8 Nonambulators: 46 ASK (mean + 1SD): 62.4 + 25.6 All participants walked; No specific results reported for PAS or WeeFIM, only p-values
3.5 + 1.8 (0.58- 7 (53 7.33) total)
Mean scores on mobility domain of PEDI ranged 33.1 - 36.6
(5.1-12.7)
Speed (m/min): Self selected: 40.8 + 12.5 Fast: 69.4 + 16.6
15
Prospective Low level lesions, cross-sectional specific levels not stated
10.10 + 4.10 (5- 8 19)
Prospective Not stated cross-sectional
6.36
To evaluate the Prospective Thoracic: 3 effectiveness of a RGO cross-sectional L1: 1 L3: 3 L4: 6 L5: 1 L5-S1: 1 Sacral: 1
EP
Speed Ambulation categorized as: community, household, exercise, nonambulator
Prospective cohort
L1-3: 13 L4-5: 15 Sacral: 13
RI PT
MMC
To determine the Prospective relationship between cross-sectional spinal deformity & overall physical function & self perception
SC
Walker et al. 104 2011
ACCEPTED MANUSCRIPT T12 or higher: 39
Hoffer scale & ASK
M AN U
SB
TE D
Wai et al. 2005103
1.5-15
TUG (group with SB): 8 + 1.5 s
1 with SB GMFM: 42/42 (7 total) Speed (m/min): Pre-test: 16.6 Post-test 1: 17.9 Post-test 2: 28.8 16 Community: 4 Household: 4 Exercise: 5 Nonambulator: 2
AC C
Abbreviations: SD=standard deviation; SB=spina bifida; MMC=myelomeningocele; FIM=Functional Independence Measure; LMMC=lipomyelomeningocele; NEM=Necker-Enfants Malades; SCI=spinal cord injury; RGO=reciprocating gait orthosis; KAFO=knee-ankle-foot orthosis; AFO=ankle-foot orthosis; PEDI=Pediatric Evaluation of Disability Inventory; PCI=Physiological Cost Index; CP=cerebral palsy; FMS=Functional Mobility Scale; HKAFO=hip-knee-ankle-foot orthosis; 6MWT=6-minute walk test; AIS=American Spinal Injury Association Impairment Scale; MMT=manual muscle test; DMD=Duchenne Muscular Dystrophy; SMD=spinal muscular dystrophy; UMN=upper motor neuron; PM=plastic/metal; LM=leather/metal; CVA=cerebral vascular accident; TBI=traumatic brain injury; CNS=central nervous system; FAC=Functional Abmulation Categories; MC=meningocele; HRQOL=health-related quality of life; SMA=spinal muscular atrophy; HSP=hereditary spastic paresis; 10MWT=10-meter walk test; 10-minWT=10-minute walk test; NTD=neural tube defects; MABC=Movement Assessment Battery for Children; TD=typically-developing; EMG=electromyographic; ASK=Activities Scale for Kids; PAS=Progressive Ambulation Scale; TUG=Timed Up & Go test; ABI=acquired brain injury; DS=Downs syndrome; GMFM=Gross Motor Function Measure; MS=Multiple Sclerosis.
Appendix 3: Quality Assessment of Included Studies
ACCEPTED MANUSCRIPT
Interpretation: Participant characteristics : Adequate = All details provided; Partial = All details except gender; Inadequate = missing details. Sampling method : Convenience; Community-based; Population-based; Not stated. Inclusion/exclusion criteria : Stated = clear list of both provided; Limited = 1 or 2 points only; Not stated. Data Collection: Prospective = objective exam; Retrospective = chart review; Not stated.
Participant characteristics defined?
Sampling method Inclusion/ exclusion criteria stated?
Prospective or retrospective data collection?
Reliability of Validity of Interpretability or measure stated or measure stated or responsiveness of tested? tested? measure stated/tested?
Adequate
Convenience
Stated
Prospective
No
Agre et al. 1987
Adequate
Not stated
Not stated
Prospective
No
Al-Holou et al. 3 2009
Adequate
Convenience
Stated
Prospective
No
Alman et al. 1996
Inadequate
Convenience
Limited
Prospective
No
Ammerman et al. 5 1998
Adequate
Convenience
Not stated
Prospective
No
Apkon et al. 2009
Adequate
Convenience
Stated
Prospective
7
Adequate
Convenience
Limited
Prospective
8
Banta et al. 1991
Adequate
Convenience
Not stated
Bartnicki et al. 20129
Adequate
Not stated
Stated
Bartonek et al. 10 1999a
Adequate
Convenience
Stated
Bartonek et al. 11 1999b
Adequate
Not stated
Not stated
Bartonek et al. 12 1999c
Adequate
Convenience
Limited
Bartonek & Saraste Inadequate 13 2001
Convenience
Bartonek et al. 14 2002
Inadequate
Convenience
Bartonek et al. 200515
Inadequate
Convenience
Bartonek 201016
Inadequate
Convenience
Basobas et al. 200317
Inadequate
Convenience
Batavia et al. 200118
Inadequate
Convenience
6
Asher et al. 1983
No
Yes - speed expressed as % of normal
No
No
No
No
No
No
No
No
No
No
No
No
RI PT
No
SC
4
No
M AN U
2
Prospective
No
No
No
Prospective
No
No
No
Prospective
No
No
No
Prospective
No
No
No
Prospective
No
No
No
Limited
Prospective
No
No
No
Not stated
Prospective
No
No
No
Limited
Prospective
No
No
No
Limited
Prospective
No
No
No
Limited
Retrospective
No
No
No
Not stated
Prospective
No
No
No
No
TE D
1
Adzick et al. 2011
EP
Author & Year
Convenience
Stated
Retrospective
Benzer et al. 201220 Adequate
Convenience
Not stated
Retrospective
No
No
No
Bier et al. 200521
Adequate
Convenience
Not stated
Prospective
No
No
No
Adequate
Convenience
Stated
Prospective
No
No
No
Carbonari de Faria Partial et al. 201323
Convenience
Stated
Not stated
No
No
No
Chang et al. 200824 Adequate
Convenience
Limited
Prospective
No
No
No
Battibugli et al. 200719
Buffart et al. 200822
AC C Adequate
Yes - FIM: MRC= 0.92-0.95 (interrater, test-retest, equivalence) No
Cuddeford et al. 25 1997
Partial
Not stated
Not stated
Prospective
No
No
Yes - speed compared to normal value
Cuppen et al. 26 2013
Inadequate
Convenience
Stated
Prospective
No
No
No
ACCEPTED MANUSCRIPT Stated Retrospective No
Partial
Convenience
Danzer et al. 201128
Inadequate
Convenience
Not stated
Prospective
Yes - WeeFIM stated to be reliable
Yes - WeeFIM Yes - WeeFIM score stated to be valid compared to normal value
De Groot et al. 200829
Adequate
Convenience
Stated
Retrospective
No
No
Yes - 6MWT (expressed as % of predicted)
De Groot et al. 30 2009
Inadequate
Convenience
Stated
Prospective
No
No
No
De Groot et al. 201031
Adequate
Convenience
Stated
Prospective
Yes - speed: ICC=0.97 for testretest
No
Yes - speed: SEM=2.5 m/min; SDD=6.8 m/min
De Groot et al. 2011a32
Inadequate
Community-based Stated
Prospective
Yes - 6MWT: testretest stated excellent
No
No
De Groot et al. 33 2011b
Adequate
Convenience
Stated
Prospective
Yes - 6MWT: ICC=0.98 for testretest
No
Yes - 6MWT: SEM=13.1m; SDD=36.3m
De Souza & Carroll Partial 34 1976
Convenience
Limited
Prospective
No
No
No
Dudgeon et al. 35 1991
Adequate
Convenience
Stated
Retrospective
Duffy et al. 1996
36
Inadequate
Convenience
Limited
Prospective
Evaggelinou & 37 Drowatzky 1991
Inadequate
Community-based Not stated
Feldman et al. 200838
Inadequate
Community-based Stated
Findley et al. 198739
Adequate
Population-based
Limited
Findley & Agre 40 1988
Adequate
Not stated
Limited
Convenience
Gerritsma-Bleeker Adequate 42 et al. 1997
Convenience
No
SC No
No
No
No
Yes - speed compared to normal value No
M AN U
No
Prospective
No
No
Prospective
Yes - WeeFIM: interrater (Kappa 0.44-0.82) stated
Yes - WeeFIM Yes - WeeFIM: stated to be valid severity rating for total score given
Prospective
No
No
No
Prospective
No
No
No
TE D
Franks et al. 199141 Adequate
No
RI PT
Danzer et al. 200927
Stated
Prospective
No
No
No
Limited
Retrospective
No
No
No
Limited
Restrospective
No
No
No
No
No
Convenience
Guille et al. 2002
Adequate
Convenience
Limited
Retrospective
No
Hassan et al. 201045
Adequate
Not stated
Stated
Prospective
Yes - 6MWT: stated No to be reliable
Yes - 6MWT: score compared to normal values
AC C
44
EP
Inadequate
Grujic & Aparisi 198243
Hisaba et al. 201246 Partial
Convenience
Stated
Retrospective
No
No
No
Hoffer et al. 197347 Adequate
Convenience
Stated
Retrospective
No
No
No
Høiness & Kirkhus Inadequate 200948
Convenience
Limited
Retrospective
No
No
No
Hunt et al. 197349
Inadequate
Convenience
Limited
Prospective
No
No
No
Ilharreborde et al. Inadequate 200950
Convenience
Limited
Retrospective
No
No
No
Jaworek et al. 201351
Not stated
Not stated
Prospective
No
No
No
Convenience
Not stated
Not stated
No
No
No
Adequate
Kaiser & Rudeberg Inadequate 52 1986 Kramel-Ross et al. 53 1992
Partial
Not stated
Limited
Prospective
No
No
No
Katz et al. 199754
Adequate
Convenience
Limited
Prospective
No
No
No
55
ACCEPTED MANUSCRIPT Limited Prospective No
No
No
Labruyere et al. 56 2013
Adequate
Not stated
Stated
Prospective
No
No
No
Lemelle et al. 57 2006
Inadequate
Community-based Stated
Prospective
No
No
No
Lewis et al. 2004
Partial
Convenience
Limited
Retrospective
No
No
No
Lorente Molto & 59 Garrido 2005
Adequate
Convenience
Limited
Retrospective & prospective
No
No
No
Lough & Nielsen 60 1986
Adequate
Convenience
Not stated
Prospective
No
No
No
Magee & Kenny 200861
Adequate
Not stated
Not stated
Not stated
No
No
No
Marreiros et al. 62 2010
Inadequate
Convenience
Stated
Retrospective
No
McCall & Schmidt 63 1986
Adequate
Not stated
Limited
Prospective
No
McDonald et al. 64 1991
Inadequate
Not stated
Limited
Prospective
No
Moerchen et al. 65 2011
Adequate
Convenience
Stated
Prospective
Moore et al. 2001
Adequate
Not stated
Not stated
Prospective
Müller et al. 67 1992a
Adequate
Convenience
Limited
Müller et al. 1992b68
Inadequate
Convenience
Limited
Not stated
Stated Stated
66
Müller et al. 199469 Adequate Adequate
Convenience
Murdoch et al. 197971
Inadequate
Convenience
Norrlin et al. 72 2003
Adequate
Not stated
OkurowskaZawada et al. 201173
Adequate
Convenience
Ozaras et al. 2005
Adequate
Convenience
Pauly & Cremer 201375
Partial
Convenience
Petrikovsky et al. 200176
No
No
No
No
No
No
Yes - FMS: 50m functional for home & daycare
No
No
Yes - speed compared to control values
No
No
No
Prospective
No
No
No
Retrospective & prospective
No
No
No
Prospective
No
No
No
Limited
Prospective
No
No
No
Stated
Prospective
No
No
No
Stated
Prospective
No
No
No
Not stated
Prospective
No
No
No
Not stated
Retrospective
No
Yes - Hoffer: convergent validity shown
No
AC C
74
No
EP
Müller-Godeffroy 70 et al. 2008
No
Prospective
TE D
58
RI PT
Not stated
SC
Inadequate
M AN U
Krebs et al. 1988
Inadequate
Convenience
Not stated
Prospective
No
No
No
Phillips & Lindseth Inadequate 199277
Convenience
Limited
Retrospective
No
No
No
Phillips et al. 199578
Adequate
Convenience
Limited
Retrospective
No
No
No
Pirpiris et al. 200379 Adequate
Convenience
Stated
Retrospective & Prospective
Yes - 10 min WT: ICC=0.91 for testretest
Yes - speed compared with normal values
Rose et al. 198180
Inadequate
Convenience
Limited
Prospective
No
Yes - 10MWT & 10 min WT: convergent validity No
No
Ross et al. 2007
Adequate
Convenience
Limited
Retrospective
No
No
No
Samuelsson & 82 Skoog 1988
Adequate
Convenience
Stated
Prospective
No
No
No
Schiltenwolf et al. 83 1991
Inadequate
Not stated
Not stated
Prospective
No
No
No
81
ACCEPTED MANUSCRIPT Not stated Prospective No
Shoenmakers et al. Adequate 200384
Convenience
Shoenmakers et al. Adequate 200485
Convenience
Stated
Prospective
Schoenmakers et 86 al. 2005a
Adequate
Convenience
stated
Prospective
Shoenmakers et al. Adequate 87 2005b
Convenience
Limited
Prospective
Yes - PEDI: stated to be reliable
Yes - PEDI: stated No to be valid
Schoenmakers et 88 al. 2009
Adequate
Convenience
Stated
Prospective
No
No
Seitzberg et al. 200889
Adequate
Convenience
Stated
Retrospective
No
Sherk et al. 1991
Adequate
Convenience
Not stated
Prospective
No
Stallard et al. 91 1991
Inadequate
Convenience
Limited
Prospective
No
Stillwell et al. 198492
Adequate
Convenience
Limited
Prospective
No
Suson et al. 2010
Inadequate
Convenience
Not stated
Retrospective
Swank & Dias 94 1992
Adequate
Convenience
Stated
Retrospective
Swank & Dias 95 1994 Tezcan & Simsek 96 2013
Inadequate
Convenience
Limited
Adequate
Convenience
Stated
Thomas et al. 200197
Adequate
Convenience
Stated
Thompson et al. 98 2000
Inadequate
Community-based Limited
Tsai et al. 200199
Adequate
Convenience
Not stated
Tsai et al. 2002
Adequate
Convenience
van denBergEmons et al. 101 2001 Verhoef et al. 2004102
Adequate
Convenience
Adequate
Population-based
Wai et al. 2005103
Adequate
Convenience
Walker et al. 2011104
Adequate
Convenience
AC C
100
WassenbergSeverijnen et al. 2003105
RI PT
No No
No
No
No
No
No
No
SC
No
No
No
No
No
No
M AN U
No
No
Prospective
No
No
No
Prospective
No
No
No
Prospective
No
No
No
Prospective
No
No
No
Stated
Prospective
No
No
Yes - PEDI scores compared with normative data
Stated
Prospective
No
No
No
Stated
Prospective
No
No
No
Stated
Prospective
Yes - ASK: stated to Yes - ASK: stated be reliable to be valid
No
Stated
Prospective
No
No
No
Yes - PEDI Mobility No scale: ICC>0.94 for test-retest, interrater No No
No
Stated
Prospective
Adequate
Not stated
Not stated
Prospective
Williams et al. 2005107
Inadequate
Community-based Stated
Prospective
Winchester et al. 2002108
Inadequate
Not stated
Stated
Prospective
Adequate
Convenience
Stated
Prospective
109
Yes - PEDI: scores compared to normative values
No
Convenience
Yngve et al. 1984
Yes - MBAC & PEDI (normative data)
No
Inadequate
Williams et al. 1983106
Yes - MABC & PEDI: Yes - MABC: conreliability stated current validity (not specific to SB) stated (not specific to SB) Yes - PEDI: intra- & Yes - PEDI: content inter-interviewer & construct stated stated
No
Prospective
TE D
93
EP
90
No
Yes - speed compared to normal values
Yes - TUG: ICC=0.99 Yes - TUG: for within session convergent test-retest validity shown
Yes - TUG: responsiveness shown in TD children Yes - speed Yes - GMFM: stated Yes - GMFM: high intra- & stated valid in CP, compared to interrater in CP BI & DS normal values No
Abbreviations: FIM=Functional Independence Measure; MRC=median reliability coefficient; 6MWT=6-minute walk test;
No
Yes - speeds compared to community walking requirements
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ICC=intraclass correlations coefficient; SEM=standard error of measurement; SDD=smallest detectable difference; FMS=Functional Mobility Scale; 10 min WT=10-minute walk test; 10MWT=10-meter walk test; MABC=Movement Assessment Battery for Children; PEDI=Pediatric Evaluation of Disability Inventory; SB=spina bifida; ASK=Activities Scale for Kids; TUG=Timed Up & Go test; TD=typically-developing; GMFM=Gross Motor Function Measure; CP=cerebral palsy; BI=brain injury; DS=Down syndrome.
ACCEPTED MANUSCRIPT
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