- Email: [email protected]
Toolbox of multiple-item measures aligning with the ICF Core Sets for children and youth with cerebral palsy
Accepted Manuscript Toolbox of multiple-item measures aligning with the ICF Core Sets for children and youth with Cerebral Palsy Verónica Schiariti, MD MHSc PhD, Sandy Tatla, MOT, MSc., Karen Sauve, BSc. PT, MSc, Maureen O’Donnell, MD MSc FRCPC PII:
S1090-3798(16)30191-X
DOI:
10.1016/j.ejpn.2016.10.007
Reference:
YEJPN 2136
To appear in:
European Journal of Paediatric Neurology
Received Date: 18 October 2015 Revised Date:
6 October 2016
Accepted Date: 20 October 2016
Please cite this article as: Schiariti V, Tatla S, Sauve K, O’Donnell M, Toolbox of multiple-item measures aligning with the ICF Core Sets for children and youth with Cerebral Palsy, European Journal of Paediatric Neurology (2016), doi: 10.1016/j.ejpn.2016.10.007. 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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Toolbox of multiple-item measures aligning with the ICF Core Sets for children and youth with Cerebral Palsy
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Karen (First name) Sauve (Lastname), BSc. PT, MSc b; c Children’s Hospital, 4480 Oak Street Vancouver, British Columbia, V6H 3V4, Canada
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Sandy (First name) Tatla (Lastname), MOT, MSc. b; c Children’s Hospital, 4480 Oak Street Vancouver, British Columbia, V6H 3V4, Canada
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Verónica (First name) Schiariti (Lastname), MD, MHSc, PhD a,b; a Department of Pediatrics, University of British Columbia, Room F614, 4480 Oak Street Vancouver, British Columbia, V6H 3V4, Canada b ;Division of Medical Sciences, University of Victoria, PO Box 1700 STN CSC, Victoria, British Columbia, V8W 2Y2, Canada Email:[email protected]
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Maureen (First name) O’Donnell (Lastname), MD MSc FRCPC a,b. a Department of Pediatrics, University of British Columbia, Vancouver, British Columbia, Canada; b BC Children’s Hospital, 4480 Oak Street Vancouver, British Columbia, V6H 3V4 Canada
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Corresponding author: Verónica Schiariti MD MHSc PhD Department of Pediatrics University of British Columbia Room F614, 4480 Oak Street Vancouver, British Columbia, V6H 3V4 Email:[email protected] Phone: 604-875-2000 Fax: 604-875-3569
Word count: Abstract 250 words, Manuscript 4745 words 2 tables, 3 figures for printing version Additional online supplementary material: 1 table, 3 appendices Manuscript category: Systematic review Running title: ICF Core Sets-based toolbox for CP
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Abstract Selecting appropriate measure(s) for clinical and/or research applications for children and youth with Cerebral Palsy (CP) poses many challenges. The newly developed International Classification of Functioning, Disability and Health (ICF) Core Sets for children and youth with CP serve as universal guidelines for assessment, intervention and follow-up. The aims of this study were: 1) to identify valid and reliable measures used in studies with children and youth with CP, 2) to characterize the content of each measure using the ICF Core Sets for children and youth with CP as a framework, and finally 3) to create a toolbox of psychometrically sound measures covering the content of each ICF Core Set for children and youth with CP. All clearly defined multiple-item measures used in studies with CP between 1998 and 2015 were identified. Psychometric properties were extracted when available. Construct of the measures were linked to the ICF Core Sets. Overall, 83 multiple-item measures were identified. Of these, 68 measures (80%) included reliability and validity testing. The majority of the measures were discriminative, generic and designed for school-aged children. The degree to which measures with proven psychometric properties represented the ICF Core Sets for children and youth with CP varied considerably. Finally, 25 valid and reliable measures aligned highly with the content of the ICF Core Sets, and as such, these measures are proposed as a novel ICF Core Sets-based toolbox of measures for CP. Our results will guide professionals seeking appropriate measures to meet their research and clinical needs worldwide. Keywords: cerebral palsy, measure, child, ICF, core sets, toolbox
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Highlights • First ICF-based toolbox of measures for children and youth with CP is proposed. • The twenty-five multiple-item measure toolbox operationalizes the ICF Core Sets for children and youth with CP. • No single psychometrically robust measure fully represents the content of the ICF Core Sets for CP. • The toolbox guides professionals seeking comprehensive valid and reliable measures.
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Assisting Hand Assessment (AHA) Assessment of Motor and Process Skills (AMPS) Activities Scale for Kids (ASK) Bimanual fine motor function (BFMF) Bruininks Oseretsky Test of Motor Proficiency (BOTMP) Behaviour Rating Inventory of Executive Functioning (BRIEF) The Bayley Scales of Infant Development (2nd edn;) (BSID-II) Children's Assessment of Participation and Enjoyment (CAPE) Child Behaviour Checklist (CBCL) Child engagement in daily life (CDEL) Canadian Occupational Performance Measure (COPM) Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD) Quality of life of children with CP (CPQOL) Drooling Impact Scale (DIS) Dimension of Mastery Questionnaire (DMQ) Developmental Test of Visual Perception 2nd ed (DTVP-2) Gillette Functional Assessment Questionnaire (FAQ) Functional Independence Measure (FIM) Family Support Scale (FSS) Gross Motor Function Measure (GMFM) Gross Motor Performance Measure (GMPM) Harter-SPPC-CP (HSPPC-CP) Health Utilities Index (HUI3) Jebsen-Taylor Test of Hand Function (JTHF) KIDSCREEN Life-Habits questionnaire (LHQ) Measurement of Activities of Daily Living questionnaire (MADL) Melbourne Assessment of Upper Limb Function (MAULF) Mobility Questionnaire (MobQues) Participation and Environmental Measure children and youth Peabody Developmental Motor Scales (PDMS-2) Pediatric Evaluation of Disability Inventory (PEDI) Quality of Function Measure (QFM) Preschool Language Scales, Fifth edition (PLS-5) Pediatric Outcomes Data Collection Inventory (PODCI) Pediatric Volitional Questionnaire (PVQ) Quality of Life Instrument for People With Developmental Disabilities-children version (QOLDD) Quality of Upper Extremity Skills Test (QUEST) The Revised Children's Manifest Anxiety Scale (RCMAS) School Function Assessment (SFA) Self-Perception Profile for College Students (SPPCS) TNO-AZL questionnaire for Children's HRQOL (TACQOL) Toddler Infant Motor Evaluation (TIME) Test of Nonverbal Intelligence, 2nd edition (TONI-2) Vineland Adaptive Behavior Scales (VABS) Functional Independence Measure for children (WEEFIM)
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1. Introduction Cerebral palsy (CP) is a life-long neurodevelopmental disorder that impacts children’s and youth’s functional abilities and developmental trajectories. 1 As children and youth with CP transition from birth to adulthood, their needs and functional abilities change. As such, it is imperative to select appropriate measures to best capture these age-specific needs and to evaluate their response to interventions over time.
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Selecting appropriate measures for clinical and/or research applications for children and youth with CP poses many challenges. Several factors influence the selection of a suitable measure; particularly, the research question and/or the clinical purpose, the type of intervention, the psychometric properties, the availability of a client/proxy version, the age range, and the specific condition under investigation. Previous reviews have compared the characteristics of measures used in CP, providing useful information on measurement characteristics and theoretical backgrounds of a range of instruments.2-7
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Despite the breadth of information currently available, there remains a lack of consensus regarding which measures are best used in clinical practice and/or research studies with this population. Further, inconsistencies in the application of available measures call into question the ability to evaluate responses to interventions and to compare results across studies worldwide. Furthermore, a need has been identified to compile data on disability, including the functional impact of chronic conditions, such as CP. It has been recommended that such data be made internationally comparable in order to benchmark and monitor national and international progress on disability policies (The WHO Global Disability Action Plan 2014-2021). 8 Specific actions have been proposed, including the implementation of valid and reliable tools, consistent with the International Classification of Functioning, Disability and Health (ICF). 9
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Our research group conducted a content analysis of all multiple-item measures used as outcome measures in interventional and/or observational studies with children and youth with CP using the ICF. 10 Our previous work characterized the content of each measure used in CP based on the entire pediatric ICF classification. 11 From our findings, no multiple-item measure alone fully represents the components of the ICF; therefore, a combination of measures seemed most appropriate if the goal is to comprehensively describe the functional profile of a child or youth with CP. Due to the extensiveness of the ICF classification, it is recommended to apply ICFbased tools, such as the ICF Core Sets for specific health conditions or functional profiles, when available. An ICF Core Set is a shortlist of ICF categories that are considered most relevant for describing the functioning of an individual with a particular health condition. 12, 13 In keeping with the specific aims of the ICF ─ to establish a universal language for describing functional abilities, to permit comparison of data across countries, andto provide a systematic coding scheme for health information systems, among others ─ the newly developed ICF Core Sets for children and youth with CP 14 serve as universal guidelines for assessment, intervention and follow up. Following a rigorous multi-step methodology endorsed by the WHO, our research team ─ in collaboration with the ICF Research Branch of the WHO German Collaborating Centre for the Family of International Classifications ─ and international CP experts ─ representing all WHO health regions ─ developed five ICF Core Sets14 : 1) 4
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a comprehensive set covering all the developmental stages from birth up to 18 years of age consisting of 135 ICF categories; 2) a brief set consisting of 25 ICF categories that are common to describe children and youth with CP up to 18 years of age; 3) a 31-category brief set for children and youth with CP below 6 years of age; 4) a 35-category brief set for children and youth with CP ≥6 and <14 years of age; and 5) a 37-category brief set for children and youth with CP ≥14 and 18 years of age. To ensure international applicability, the development of the ICF Core Sets for children and youth with CP involved the international community; specifically the development methodology incorporated the perspectives of international health, education and social sciences professionals as well as the valuable perspectives of children and youth with CP and their caregivers. 10, 15-18 Finally, the five ICF Core Sets for children and youth with CP capture the developmental trajectory that children and youth with CP transition from birth to young adults, facilitating the characterization of the functional trajectory of this population over time. 19 Above all, the ICF Core Sets can guide professionals in selecting the most appropriate outcome measures to comprehensively capture functional information regarding children and youth with CP.
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When selecting an outcome measure, it is important to examine the psychometric properties of the measures, the conceptualization of constructs, and the content used to measure these. While numerous scales exist to measure patient outcomes, few measures have undergone rigorous psychometric testing to support the interpretations based on their use. Marshal et al., 20 conducted a systematic review of over 300 randomized control trials and found that studies were 40% more likely to report that treatment was effective when they used unpublished scales, rather than validated ones 20; thus, demonstrating the implications of measurement selection for patient care. In the field of CP, Hoare & Imms 21 reviewed 15 studies evaluating the effects of Botolinum toxin in the upper limb, and found that only 4 studies had used valid and reliable measures, which compromise the accuracy of the results. Contemporary validity researchers reason that psychometric considerations are relevant to researchers and consumers of measures, alike 22 As such; one must be critical and judicious when selecting measures to determine if they are valid and reliable for their intended use. Furthermore, since the publication of the ICF, clinicians and researchers are also encouraged to consider the “ICF mapping” of the content of available measures.
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Our research group have demonstrated that the content of multiple-item measures –defined as those measures comprising of several items, where each item is a single question, observation, or a statement that needs to be evaluated –varied considerably based on theirs ICF content representation, highlighting the differences between multiple-item measures and the importance of adding the ICF content information 10 Notably, we found that multiple-item measures highly capture functional domains identified as important areas of functioning by international experts, and most importantly, by children and youth with CP and their families. 10, 18 To that end, the focus of this study is to build upon our previous work in providing content analysis of multipleitem measures used with children and youth with CP10, and to recommend a minimum set of multiple-item measures that comprehensively represent the functional profile of this population. Specific objectives of this study were: 1) to examine the psychometric properties of measures used with children and youth with CP, 2) to map the content of each multiple-item measure using the ICF Core Sets for children and youth with CP as a framework, and finally 3) to propose a toolbox of valid and reliable measures aligning with the ICF Core Sets. 5
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2. Methods In this study, we followed a four-step methodology. Firstly we identified multiple-item measures applicable to children and youth with CP. Secondly, we categorized the purpose of each measure and its psychometric properties. Thirdly, we mapped the content of multiple-item measures ─with proven psychometric characteristics─ to the ICF Core Sets for children and youth with CP and identified a toolbox of measures representing the ICF Core Sets. Finally, we extracted the clinical utility characteristics of the measures included in the toolbox. Figure 1describes the methodology overview of this study.
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2.1 Step 1- Identifying multiple-item measures We used two sources for retrieving multiple-item measures used in CP. Source 1, a systematic review of the literature including original studies on children and youth with CP with a clearly defined outcome measure published between 1998-2012, detailed description of the methodology is available.10 Source 2, we conducted a consultation with clinical and research experts and a target literature search to identify newly developed and/or revised multiple-items measures published between January 2013 and March 2015. Overall, we identified 83 multipleitem measures, 78 measures were retrieved from source 110 and 5 measures were identified from source 2.
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2.2 Step 2- Measures categorization and psychometric analysis We categorized measures according to age-groups, generic and/or CP-condition specific, proxy and/or self-reported, and whether they were descriptive, evaluative or predictive. Next, we evaluated the psychometric properties of each instrument, focusing specifically on whether studies objectively reported the presence of reliability data, including: internal consistency, testretest, inter-rater and intra-rater reliability, and responsiveness, and validity data, including: content, construct, criterion, concurrent, and convergent validity. We considered psychometrically sound measures to be those measures that underwent both reliability and validity testing of at least four psychometric properties with adequate to excellent values ( ≥ 0.6, arbitrary criteria). Reliability here was defined as the consistency in properties of test responses, indicating the extent to which differences are true, as opposed to measuring error.23 Validity was defined as the degree to which evidence and theory support the interpretation of measurement results.23 See full glossary in Appendix A (online). 2.3 Step 3a- Mapping content to the ICF Core Sets for children and youth with CP We followed the ICF linking guidelines proposed by Cieza et al 24, which have been applied to a variety of outcome measures in adults and pediatric measures (e.g. DISABKIDS, cancer measures, CP disease-specific measures, and generic patient-reported outcomes). 10, 25-27The content of the measure is linked to the two parts of the ICF. Part 1 covers functioning and disability and includes the components of body functions (b) and structure (s) and activities and participation (d). Part 2 covers contextual factors and includes the components environmental factors (e) and personal factors (pf). In the classification, the letters b, s, d and e, which refer to the component of the classification, are followed by a numeric code starting with the chapter 6
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number adding one digit, (e.g d9-community life), followed by a second level code adding two digits (e.g d920-recreation and leisure), third or fourth level code by adding one digit (e.g d9200play). This linking procedure was performed separately by two trained assessors (VS, KS). Subsequently, disagreements were resolved by consensus. Inter-coders reliability of the linking process was 0.74 (95% confidence interval 0.73-0.75). Before linking each measure, the purpose of the measure and its manual were reviewed, a decision was made as to whether the full content (individual items) could be linked to the ICF Core Sets or only the main domains could be linked, for example the main domains of the AMPS were linked as the detailed information contained in the individual items would not have been capture by the ICF coding system resulting in redundant ICF information. Only original versions of the measures were linked. Finally, the full content of some multiple-item measures were unavailable because of access fees, as described in the articles, therefore the main domains were linked (e.g. WeeFIM).
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Step 3b- ICF-based Toolbox of multiple-item measures Once the content of all psychometrically sound measures were linked to the five ICF Core Sets, those measures whose content represent areas of “functioning” as defined by the ICF Core Sets for children and youth with CP were included in the ICF-based toolbox. Specifically, the toolbox inclusion criteria consisted of: 1) multiple-item measure with at least 4 tested psychometric properties of both reliability and validity with adequate to excellent values and 2) content of measure mapping ICF categories of the ICF Core Sets at the ICF second level (e.g. d920recreation and leisure).
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2.4 Step 4-Clinical Utility- Toolbox of multiple-item measures Finally, we collected information regarding the format (observation, proxy/self-report, performance, etc), required qualifications (Masters level, etc), estimated time for completion, and associated fees of each measure included in the ICF-based toolbox.
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2.5 Data analysis Descriptive analysis was conducted in SPSS. For each of the identified instruments, we calculated the frequency with which its items/domains addressed b, s, d, e and pf. If an ICFcategory was assigned repeatedly in a measure, the category was counted only once.
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3. Results 3.1 Multiple-item measures characteristics We found, 68 out of 83 multiple-item measures used in studies with children and youth with CP demonstrating evidence of validity and reliability. (Figure 1) The majority of the measures were discriminative, generic, proxy reported, and designed for school-aged children. 3.2 Multiple-item measures meeting study’s psychometric criteria, n=46 Table 1(online) shows the forty six multiple-item measures meeting the study’s psychometric criteria – measures that reported at least four tested psychometric properties within both reliability and validity domains with adequate to excellent values. On average, five psychometric properties were tested, with a range of four to nine.
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Measures demonstrating the greatest number of psychometrics, included: AMPS, FIM, GMFM88, MAULF, PDMS-2, PEDI, SFA, TONI-2, VABS, and WEEFIM. Generic functional measures appeared to have the highest number of reported psychometric properties (e.g. PEDI, FIM, and AMPS).
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When we assessed the purpose of the 46 psychometrically sound measures, 24 (52%) were discriminative, 7 (15%) were evaluative, 14 (30%) were both discriminative and evaluative, and only 3 were predictive measures. S measures were CP-specific (CPCHILD, CPQOL, GMFM, GMPM, QFM, and QUEST) and among them only one (CPQOL) was self-reported.
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3.3 Mapping to the ICF Core Sets for children and youth with CP n=42 measures We were able to link the content of 42 measures with proven psychometrics at the domain and item level. We were unable to link the content of 4 measures due to lack of access to the measures. The 42 measures varied considerably in the degree to which their content represented the ICF Core Sets for CP. Primarily, measures covered the ICF components of activities and participation. Self-care, mobility, and recreational and leisure were the most frequent areas represented by the measures. Overall, individual measures few categories comprising the ICF Core Sets. A single measure (PEM-CY) covered the majority of the environmental factors included in the ICF Core Sets.
Mapping to Comprehensive ICF Core Set for children and youth with CP (N=135 categories)
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The comprehensive ICF Core Set allows the complete and detailed description of functional abilities and relevant contextual factors in children and youth with CP aged 0 to 18 years. This Core Set is useful in interdisciplinary assessments because it provides extensive detail, offering exhaustive assessment that can distributed among multi-professional team members. 14
Mapping to Common Brief ICF Core Set for children and youth with CP (N=25 categories)
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Evaluative measures demonstrating the highest coverage of ICF categories included in this Core Set were: ASK, CPCHILD, COPM, FIM, PEDI, PODCI, SFA, and WEEFIM. Discriminative measures that aligned with the Comprehensive ICF Core Set include: ASK, CBCL, CPQOL, CDEL, KIDSCREEN, PEDI, PEM-CY, PLS-5, and VABS.
The Common Brief ICF Core Set describes the most common areas of functioning in children and youth aged 0 to 18 years with CP. This Core Set can be employed in regular clinical encounters where only a brief assessment is necessary, and in clinical and epidemiological research. This Core Set can be used by a single professional or an inter-disciplinary team. A key characteristic of the Common Brief Core Set is that it allows the description of the level of functioning of children and youth with CP over time, from birth to transition to adulthood. Therefore, this Core Set can be considered as a minimum for any “minimal data set” which describes children and youth with CP across their lifespan. 14
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The evaluative measures with the most coverage of the ICF categories included in this Core Set are: AMPS, CPCHILD, GMFM, PEDI, PDMS-2, PODCI, SFA, and WEEFIM. The discriminative measures that predominantly aligned with the Common Brief Core Set are: AMPS, CDEL, CPQOL, LHQ, PEM-CY, PEDI, PODCI, TACQOL, and VABS. The content of the PEM-CY mapped to all the environmental factors of the Common Brief ICF Core Set.
Mapping to Age-Specific Brief ICF Core Sets (N=31, 35, and 37 categories, respectively)
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Figure 2 illustrates the mapping of psychometrically sound measures to the Common Brief ICF Core Set. Few measures containing multiple attributes comprehensively cover the range of categories included in this Core Set (CPCHILD, CPQOL, PEDI, SFA, PEM-CY, and VABS). Two measures cover the components of body functions, activities and participation, and environmental factors (CPQOL and SFA). As shown in figure 2, the coverage of each ICF category included in the Core Set varies significantly across measures. The categories most commonly represented by the measures are: d550-eating, d530-toiletting, d450-walking, and d410 maintaining a body position.
Each of the age-specific sets includes the ICF categories of the Common Brief ICF Core Set in addition to ICF categories unique to each age-group: 0 to <6, 6≥ to <14 and 14≥ to 18 years of age. Each age-specific ICF Core Set describes the most common areas of functioning in children and youth with CP for the associated age-group. The Age-Specific ICF Core Sets can be applied in clinical or research context where additional detail is desirable in caring for or describing a particular age-group of children and youth with CP. These ICF Core Sets can be used by a single professional or an interdisciplinary team.
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The age range for which the measures were developed varies significantly. The content of few measures aligned with the Brief ICF Core Set for children <6 years of age; in particular for children < 3 years (n=l9/42). Similarly, fewer psychometrically sound measures aligned with the transition to adulthood Brief ICF Core Set, specifically for teenagers ≥ 16 years of age. A large number of measures addressed the school-aged Brief ICF Core Set.
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3.4 Proposed ICF Core Sets-based Toolbox of measures for children and youth with CP Table 2 shows the 25 measures included in the ICF-based toolbox. Measures are applicable to children and youth with CP aged 0 to 18 years, will all levels of functioning and all types of CP. The vast majority focused on school-aged children. For example, as shown in table 2, only two measures (CDEL and PDMS-2) are specific for children < 6 years of age. Three measures are specific for school aged and the transition to adulthood youth (CAPE, CBCL, and KIDSCREEN). Table 3 illustrates the characteristics and the representation of each component of the ICF Core Sets by the 25 measures included in the toolbox. As shown in table 3, the propose toolbox highly represents the component activities and participation, followed by the components body functions and environmental factors. All in all, the toolbox embraces functional areas included in the ICF Core Sets. (For full description of measures please see Appendix B, online). 3.5 Clinical utility: Clinical utility data were extracted for the 25 measures included in the toolbox. (Appendix C, online). Sixteen (67%) measures are administered via proxy report (parent/caregiver), and 13 9
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(54%) include a self-report version through which children and youth can share their perspectives. Eleven (44%) measures are based on observation by a clinician or teacher and seven (29%) measures require an interview with parent/caregiver and/or child.
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In terms of complexity and time to administer, the AMPs, CAPE, CBCL, COPM, FIM, GMFM, LHQ, PDMS-2, PEDI, PEM-CY, and QUEST require at least 30 minutes to administer and are more complex in terms of set up and/or scoring and administration. Nonetheless, these measures provide more comprehensive information regarding the child's functioning across various domains. Simple measures reflected those requiring between 10-20 minutes for administration, and included: AHA, ASK, CDEL, CPCHILD, CPQOL, FAQ, HUI3, KIDSCREEN, PODCI, and SFA (depending on the number of scales administered- SFA can fit in easy or more complex). Importantly, the toolbox includes measures applicable in different contexts - home, school, and community.
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4. Discussion This study identified a set of valid and reliable multiple-item measures assessing the relevant areas of functioning highlighted in the ICF Core Sets for children and youth with CP. The diverse purposes of the measures demonstrate the broad spectrum of functional impact of CP on children and youth. The majority of the measures were discriminative, proxy reported, and for school-aged children. By using the ICF Core Sets for CP as a common framework, clinicians and researchers can systematically incorporate an integrated functional approach when selecting appropriate measures for clinical practice and/or research studies.
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4.1 ICF Core Sets-based toolbox of multiple-item measures for CP Our work describes the first attempt to create an “ICF-bassed toolbox of measures for children and youth with CP” consisting of clinically feasible, psychometrically robust measures, across all levels of functioning and developmental stages. Using this toolbox, clinicians and researchers can choose the measures that best fit the purpose/s of their clinical and/or research applications and are also appropriate for each specific child or youth with CP they see. The measures included in this toolbox operationalize the implementation of the ICF Core Sets for children and youth with CP in clinical practice and research. The systematic draw of measures from this toolbox will bring international consensus on measurement approach and reporting of functional strengths and challenges of the pediatric CP population. In clinical practice, benefits of applying a set of measures from this toolbox includes a unified language for monitoring functional status in CP, ensuring each child's or youth’s needs are being met comprehensively, Although our work has summarized the extensive information on measures available today, and has narrowed the vast list of measures to a manageable ICF-based toolbox. The selection of the most appropriate measure still relies on clinical judgement of professionals and researchers. Given the heterogeneous nature of CP, selecting appropriate measures to meet the assessment needs of a specific child/youth can be challenging in clinical practice. We propose an overarching algorithm to guide professionals navigate the selection of the best fit measure. (Figure 3) It is expected that professionals will select one or a combination of measures out of the entire toolbox, depending on the clients’ goals of interventions, resources, availability of trained professionals, and access to instruments.
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As described by Wright and Majnemer 28 several factors need to be considered when creating a toolbox for children and youth with CP. Specifically, the purpose and scope of the measures, their psychometric characteristics, the inclusion of child and family’ perspectives, and the developmental consideration of each child and youth with CP. We made special efforts to incorporate all these key factors in our toolbox selection with the essential application of the ICF as the overarching framework. It is hoped that this novel toolbox will ease the overwhelming endeavour of selecting appropriate measures that clinicians and researchers face every day. Furthermore, our proposed toolbox, based on extensive data analysis, includes several of Wright’s and Majnemer’s suggested candidate outcome measures for a toolbox, in particular in the areas of activities and participation (multi domain) and motor function. 28
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Our findings are not limited to guiding the systematic assessment of functional abilities and/or challenges in children and youth with CP, but also allow the comparison of functional data worldwide. The latter can be achieved by encouraging researchers to refer to the detailed content analysis provided in this study (Appendix B, online) and by applying these guidelines globally. For example, if the purpose of the assessment is to capture information on “d710-basic interpersonal interactions” using the Common Brief ICF Core Set as a framework, professionals have the options of using the: CPQOL, KIDSCREEN, LHQ ,PEDI, PEM-CY, SFA, VABS and WEEFIM. (Figure 2)
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4.2 Comments on psychometrics properties We found that multiple-item measures lacked evaluation of responsiveness with only 25% of measures demonstrating some degree of responsivity. This is an important consideration for clinical care, as health care practitioners are often interested in monitoring functional progression or decline over time in order to guide intervention planning and care, which points to the need for further evaluation of measures in this area. We have noticed that some measures have been used as evaluative measures when they have not been tested for responsiveness to change (e.g. BSID).When using a measure as an evaluative tool, professionals should be aware of this important psychometric property to avoid misuse and misinterpretation of results.
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Notably, some measures are fairly new (e.g. Mini-AHA, Ad-AHA, PEDI-CAT, and QFM). Psychometric properties remain under investigation in these early stages and this is not to preclude their application and appropriateness for children and youth with CP. In addition, new measures might become available in the future and might be suitable candidates for the proposed toolbox. 4.3 Coverage of the ICF Core Sets for children and youth with CP by the toolbox Although the vast majority of ICF categories included in the five ICF Core Sets are represented by the toolbox. The representation varies within each ICF Core Set. Overall, the areas more frequently covered by our suggested toolbox are d4-mobility, d5-self-care, d7-interpersonal interactions and relationships, d8-major life areas, and d9-community, social and civil life. Some relevant areas of functioning are lacking measures with a high degree of psychometric testing with the CP population. For example, we identified a paucity of measures to assess cognitive abilities and communication skills (verbal and non-verbal). These aspects of functioning are critical to planning transition to adulthood services. Furthermore, few measures included in the toolbox assess “b280-sensation of pain”, none of them are pain-specific measures. Pain 11
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prevalence has been frequently reported in pediatric studies in CP, ranging from 14% to 73%; as such, it is imperative to incorporate into the routine functional assessment of children and youth with CP. 29
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Few measures offer a broad overview of environmental factors included in the ICF Core Sets for CP (KIDSCREEN, PODCI, SFA and PEM-CY). The PEM-CY is the only measure that addresses all the environmental factors categories included in the Brief ICF Core Sets and the majority of the Comprehensive ICF Core Set. Of note, the PEM-CY is a discriminative measure which helps identify important environmental factors that influence participation; however, there is a need for an evaluative tool that measures response to environmental interventions.
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4.4 Identified Gaps The paucity of self-reported measures for children and youth with CP is a major deficit. The ICF incorporates the convention of the rights of children’s and promotes the active inclusion of the child and/or youth’s perspective when assessing functional abilities. The children’s subjective experiences are as essential - or arguably greater - as more objective information, such as observed performance, to plan individualized family-centered care. Our results also point to the need for more measures to evaluate the needs of youth with CP, as this segment of the population is less represented by existing measures. Nonetheless, the age specific core set for this group (e.g. >14 years) can be used as a guide to capture functional areas using informal assessment (direct questions, observation, etc).
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We chose not to use the COSMIN (Consensus-based Standards for the selection of health status Measurement Instruments) checklist 30- to evaluate the clinimetric properties of each instrument as we used an adapted clinical utility description (Appendix C, online). The focus of this paper was to map psychometrically sound measures to the ICF Core Sets for CP, rather than to provide extensive clinimetric evaluation. Further studies can build upon these findings to conduct further analysis.
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4.5 Limitations Limiting studies to measures, which are in English, is a further limitation of our review. We encourage others who are familiar with non-English language measures to add to this body of literature by completing a similar content analysis exercise. Moreover, while the measures included in this review have been widely used with the CP population, psychometric analyses for some measures were completed with other populations (e.g. attention-deficit hyper active disorder); further research is needed to evaluate psychometric properties in children with CP, specifically. 4.6 Future directions Future work may broaden the scope of this toolbox including single-item measures to cover areas misrepresented by this multiple-item toolbox, such as muscle tone, mobility of joints, etc. In addition, future work can build upon these findings by developing self-report measures which include all categories of the ICF Core Sets. In the absence of such measures, professionals can gather information by interview, clinical examination, observation or technical testing and remediate the gaps of current standardized measures. 12
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4.7 Conclusion: This study used a robust methodology in order to identify a set of multiple-item measures for children and youth with CP. Using the ICF Core Sets as a framework, we recommend a toolbox which guides “how to measure” the most relevant areas of functioning in CP. The systematic application of this proposed toolbox of measures will standardize the measurement and report of key functional information in CP, facilitating international comparisons of findings across studies. Finally, our results will guide professionals seeking appropriate measures to meet children’s and youth’s with CP needs comprehensively.
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Acknowledgements Veronica Schiariti is the recipient of a NeuroDevNet & Child and Family Research Institute Postdoctoral Fellowship Award. This study was partially financially supported by a 2013 research grant from the American Academy of Cerebral Palsy and Developmental Medicine (AACPDM).
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We express our thanks to all the developers of measures who kindly shared their measures with us. We would also like to thank Child Health BC for its administrative support.
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Disclosure: The authors have stated that they had no interests which might be perceived as posing a conflict or bias.
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References
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1. Rosenbaum P, Rosenbloom L. What is Cerebral Palsy? Hart H (ed) Cerebral Palsy from Diagnosis to Adult Life. London: Mac Keith Press, 2012: 3-13. 2. Harvey A, Robin J, Morris ME, Graham HK, Baker R. A systematic review of measures of activity limitation for children with cerebral palsy. Dev Med Child Neurol 2008; 50: 190-198. 3. Waters E, Davis E, Ronen GM, Rosenbaum P, Livingston M, Saigal S. Quality of life instruments for children and adolescents with neurodisabilities: how to choose the appropriate instrument. Dev Med Child Neurol 2009; 51: 660-669. 4. Debuse D, Brace H. Outcome measures of activity for children with cerebral palsy: a systematic review. Pediatric Phys Ther 2011 Fall;23(3):221-312011; 23: 221-231. 5. Carlon S, Shields N, Yong K, Gilmore R, Sakzewski L, Boyd R. A systematic review of the psychometric properties of Quality of Life measures for school aged children with cerebral palsy. BMC Pediatrics 2010; 10. 6. Thomas C. Body and Society: Some Reflections on the Concepts of ’Disability’ and 'Impairment’. Watson N, Cunningham-Burley S (eds) Reframing the Body. Basingstoke, Hampshire: Palgrave, 2001: 47-62. 7. James S, Ziviani J, Boyd R. A systematic review of activities of daily living measures for children and adolescents with cerebral palsy. Dev Med Child Neurol 2014; 56: 233-244. 8. World Health Organization. Disability Action Plan 2014-2021. Geneva: WHO; 2014. 9. World Health Organization. International Classification of Functioning, Disability and Health: Children & Youth version. Geneva: WHO; 2001. 10. Schiariti V, Klassen AF, Cieza A, et al. Comparing contents of outcome measures in cerebral palsy using the international classification of functioning (ICF-CY): A systematic review. Europ J Paediatr Neurol 2014; 18: 1-12. 11. World Health Organization. International Classification of Functioning, Disability and Health: Children & Youth version. Geneva: WHO; 2007. 12. Cieza A, Ewert T, Ustun TB, Chatterji S, Kostanjsek N, Stucki G. Development of ICF Core Sets for patients with chronic conditions. J Rehabil Med 2004; (44 Suppl): 9-11. 13. Fayed N, Cieza A, Bickenbach J. Illustrating child-specific linking issues using the Child Health Questionnaire. Am J Phys Med Rehabil 2012; 91: S189-98. 14. Schiariti V, Selb M, Cieza A, O'Donnell M. International Classification of Functioning, Disability and Health Core Sets for children and youth with cerebral palsy: a consensus meeting. Dev Med Child Neurol 2015; 57: 149-158. 15. Schiariti V, Sauve K, Klassen AF, O'Donnell M, Cieza A, Masse LC. 'He does not see himself as being different': The perspectives of children and caregivers on relevant areas of functioning in cerebral palsy. Dev Med Child Neurol 2014; 56: 853-861. 16. Schiariti V, Masse LC, Cieza A, et al. Toward the development of the International Classification of Functioning Core Sets for children with cerebral palsy: a global expert survey. J Child Neurol 2014; 29: 582-591. 17. Schiariti V, Masse LC. Relevant areas of functioning in children with cerebral palsy based on the international classification of functioning, disability and health coding system: A clinical perspective. J Child Neurol 2015; 30: 216-222. 18. Schiariti V, Masse LC. Identifying relevant areas of functioning in children and youth with Cerebral Palsy using the ICF-CY coding system: From whose perspective?. Europ J Paediatr Neurol 2014; 18: 609-617. 15
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19. Schiariti V, Selb M, Cieza A, O'Donnell M. International Classification of Functioning, Disability and Health Core Sets for children and youth with CP: contributions to clinical practice. Dev Med Child Neurol 2015; 57: 203-204. 20. Marshall M, Lockwood A, Bradley C, Adams C, Joy C, Fenton M. Unpublished rating scales: A major source of bias in randomised controlled trials of treatments for schizophrenia. British Journal of Psychiatry 2000; 176: ate of Pubaton: 2000. 21. Hoare B.J., Imms C. Upper-limb injections of botulinum toxin-A in children with cerebral palsy: A critical review of the literature and clinical implications for occupational therapists. Am J Occup Ther 2004; 58: 389-397. 22. Hoyt WT, Warbasse RE, Chu EY. Construct validation in counseling psychology research. Counsel Psych 2006; 34 (6): 769-805. 23. Furr M, F., Bacharach, V.,R. Psychometrics: An Introduction. . United States of America: Sage Publications, Inc., 2008. 24. Cieza A. ICF linking rules: an update based on lessons learned. J Rehab Med 2005; 37: 212. 25. Fava L, Muehlan H, Bullinger M. Linking the DISABKIDS modules for health-related quality of life assessment with the International Classification of Functioning, Disability and Health (ICF). Disab Rehab 2009; 31: 1943-1954. 26. Fayed N, Schiariti V, Bostan C, Cieza A, Klassen A. Health status and QOL instruments used in childhood cancer research: deciphering conceptual content using World Health Organization definitions. Qual Life Res 2011; 20: 1247-1258. 27. Schiariti V, Fayed N, Cieza A, Klassen A, O'Donnell M. Content comparison of healthrelated quality of life measures for cerebral palsy based on the International Classification of Functioning. Disab RehaB 2011; 33: 1330-1339. 28. Wright FV, Majnemer A. The concept of a toolbox of outcome measures for children with cerebral palsy: Why, what, and how to use?. J Child Neurol 2014; 29: 1055-1065. 29. Penner M, Xie WY, Binepal N, Switzer L, Fehlings D. Characteristics of pain in children and youth with cerebral palsy. Pediatrics 2013; 132: e407-e413. 30. Mokkink LB, Terwee CB, Knol DL, Stratford PW, Alonso J, Patrick DL, Bouter LM, de Vet HCW. The COSMIN checklist for evaluating the methodological quality of studies on measurement properties: A clarification of its content. BMC Medical Research Methodology 2010;10:22.
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Figures attached as separate files, please see titles belows Figure 1. Overview of study methodology Figure 2. Psychometrically sound measures aligned with the Common Brief ICF Core Set for CY with CP aged 0 to 18 years
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Footnote Figure 2 *Measures with highest coverage shown in this Figure, for full descriptions please see Appendix B online. Full names of measures and references provided on Table 1, online. NOTE: The figure shows only the categories that aligned with the Common Brief ICF Core Set.
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Figure 3. ICF-based Toolbox algorithm
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Tables 1, 2, 3 attached as separate files
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Table 2. ICF Core Sets-based Toolbox for children and youth with CP
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below 6 ≥6 to 14 ≥14 to 18
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Assisting Hand Assessment (Kids-AHA) Assessment of Motor and Process Skills (AMPS) Activities Scale for Kids (ASK) Children's Assessment of Participation and Enjoyment (CAPE) Child Behaviour Checklist (CBCL) Child engagement in daily life (CDEL) Canadian Occupational Performance Measure (COPM) Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD)
Age-Specific ICF Core Sets
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Valid and Reliable measures that aligned with the Comprehensive and Common Brief Core Sets for children and youth with CP
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Quality of life of children with CP (CPQOL) Gillette Functional Assessment Questionnaire (FAQ) Functional Independence Measure (FIM) Gross Motor Function Measure (GMFM) Health Utilities Index (HUI3) KIDSCREEN Life-Habits questionnaire (LHQ) Peabody Developmental Motor Scales (PDMS-2) Pediatric Evaluation of Disability Inventory (PEDI) Participation and Environmental Measure children and youth (PEM-CY) Preschool Language Scales, Fifth edition (PLS-5) Pediatric Outcomes Data Collection Inventory (PODCI) Quality of Upper Extremity Skills Test (QUEST) School Function Assessment (SFA) TNO-AZL questionnaire for Children's HRQOL (TACQOL) Vineland Adaptive Behavior Scales (VABS) WeeFIM (WEEFIM)
Full reference shown on Table 1 online
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Generic vs CP specific
d.e d ,e d,e d,e d d e e d d, e e d, e d,e,p d d d, e d, e
G¥ G G G G G G CP CP G G CP G G G G G
5-15y 0-7.11y
d d
G G
PODCI QUEST SFA* TACQOL VABS WEEFIM*
2- 18y 1.5-8y 5-13 y 5-18y 0-18y 0.5-18y
d, e e e d d d,e
G CP G G G G
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Observation Observation Child report Self report/Interview Parent report Parent/Caregiver Report Semi-structured interview Caregiver proxy Caregiver proxy and child report Self or proxy measure Observation Observation Observ. And/or self or proxy report Self/Proxy Interview/Proxy/Self-Questionnaire Observation Observation/Parent/Caregiver interview or Self Report Parent/Caregiver Report Observation/Parent/Caregiver interview Parent/Caregiver/Self-report Observation Proxy report (Clinician/Teacher) Parent/Caregiver/Self-report Interview/Teacher form Proxy report (Clinician/Caregiver)
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PEM-CY PLS-5
Content representation by ICF Components within the ICF Core Sts Body Activities & Environmental Functions Participation Factors
Format
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Asses Purpose
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18m-12y AHA > 2y AMPS*ª 5-15y ASK 6-21y CAPE 6-18y CBCL¶ 1.5-5y CDEL All COPM* 5-18y CPCHILD 4-12y CPQOL 2-17y+ FAQ 0.5 -18y FIM 5m-18y GMFM-88 5y+ HUI3 KIDSCREEN 8-18y 3-18y LHQ* 0-5y PDMS-2 0.5-7.5y PEDIª
Type of ICF Core Set covering age range of measure
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Age Range
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Toolbox measures
*Content linked to the ICF Core Sets at the domain level; ¶ no information available on psychometric studies including CY with CP; ª small representation of CY with CP in psychometric studies ; G, generic;G¥, unilateral disability; CP, CP-specific; d, discriminative; e, evaluative; p, predictive. Comprehensive ICF Core Set; Common Brief ICF Core Set; AgeSpecific Brief ICF Core Set for children <6y; Age-Specific Brief ICF Core Set for children and youth; Age-Specific Brief ICF Core Set for youth aged 14 to 18y ICF linking representation, Content of measure includes:
mild,
moderate,
high,
For full names of measures please refer to abbreviation section, references shown in Table 1 online
very high
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Highlights • First ICF-based toolbox of measures for children and youth with CP is proposed. • The twenty-five multiple-item measure toolbox operationalizes the ICF Core Sets for children and youth with CP. • No single psychometrically robust measure fully represents the content of the ICF Core Sets for CP. • The toolbox guides professionals seeking comprehensive valid and reliable measures.
S1090-3798(16)30191-X
DOI:
10.1016/j.ejpn.2016.10.007
Reference:
YEJPN 2136
To appear in:
European Journal of Paediatric Neurology
Received Date: 18 October 2015 Revised Date:
6 October 2016
Accepted Date: 20 October 2016
Please cite this article as: Schiariti V, Tatla S, Sauve K, O’Donnell M, Toolbox of multiple-item measures aligning with the ICF Core Sets for children and youth with Cerebral Palsy, European Journal of Paediatric Neurology (2016), doi: 10.1016/j.ejpn.2016.10.007. 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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Toolbox of multiple-item measures aligning with the ICF Core Sets for children and youth with Cerebral Palsy
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Karen (First name) Sauve (Lastname), BSc. PT, MSc b; c Children’s Hospital, 4480 Oak Street Vancouver, British Columbia, V6H 3V4, Canada
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Sandy (First name) Tatla (Lastname), MOT, MSc. b; c Children’s Hospital, 4480 Oak Street Vancouver, British Columbia, V6H 3V4, Canada
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Verónica (First name) Schiariti (Lastname), MD, MHSc, PhD a,b; a Department of Pediatrics, University of British Columbia, Room F614, 4480 Oak Street Vancouver, British Columbia, V6H 3V4, Canada b ;Division of Medical Sciences, University of Victoria, PO Box 1700 STN CSC, Victoria, British Columbia, V8W 2Y2, Canada Email:[email protected]
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Maureen (First name) O’Donnell (Lastname), MD MSc FRCPC a,b. a Department of Pediatrics, University of British Columbia, Vancouver, British Columbia, Canada; b BC Children’s Hospital, 4480 Oak Street Vancouver, British Columbia, V6H 3V4 Canada
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Corresponding author: Verónica Schiariti MD MHSc PhD Department of Pediatrics University of British Columbia Room F614, 4480 Oak Street Vancouver, British Columbia, V6H 3V4 Email:[email protected] Phone: 604-875-2000 Fax: 604-875-3569
Word count: Abstract 250 words, Manuscript 4745 words 2 tables, 3 figures for printing version Additional online supplementary material: 1 table, 3 appendices Manuscript category: Systematic review Running title: ICF Core Sets-based toolbox for CP
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Abstract Selecting appropriate measure(s) for clinical and/or research applications for children and youth with Cerebral Palsy (CP) poses many challenges. The newly developed International Classification of Functioning, Disability and Health (ICF) Core Sets for children and youth with CP serve as universal guidelines for assessment, intervention and follow-up. The aims of this study were: 1) to identify valid and reliable measures used in studies with children and youth with CP, 2) to characterize the content of each measure using the ICF Core Sets for children and youth with CP as a framework, and finally 3) to create a toolbox of psychometrically sound measures covering the content of each ICF Core Set for children and youth with CP. All clearly defined multiple-item measures used in studies with CP between 1998 and 2015 were identified. Psychometric properties were extracted when available. Construct of the measures were linked to the ICF Core Sets. Overall, 83 multiple-item measures were identified. Of these, 68 measures (80%) included reliability and validity testing. The majority of the measures were discriminative, generic and designed for school-aged children. The degree to which measures with proven psychometric properties represented the ICF Core Sets for children and youth with CP varied considerably. Finally, 25 valid and reliable measures aligned highly with the content of the ICF Core Sets, and as such, these measures are proposed as a novel ICF Core Sets-based toolbox of measures for CP. Our results will guide professionals seeking appropriate measures to meet their research and clinical needs worldwide. Keywords: cerebral palsy, measure, child, ICF, core sets, toolbox
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Highlights • First ICF-based toolbox of measures for children and youth with CP is proposed. • The twenty-five multiple-item measure toolbox operationalizes the ICF Core Sets for children and youth with CP. • No single psychometrically robust measure fully represents the content of the ICF Core Sets for CP. • The toolbox guides professionals seeking comprehensive valid and reliable measures.
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Abbreviations:
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Assisting Hand Assessment (AHA) Assessment of Motor and Process Skills (AMPS) Activities Scale for Kids (ASK) Bimanual fine motor function (BFMF) Bruininks Oseretsky Test of Motor Proficiency (BOTMP) Behaviour Rating Inventory of Executive Functioning (BRIEF) The Bayley Scales of Infant Development (2nd edn;) (BSID-II) Children's Assessment of Participation and Enjoyment (CAPE) Child Behaviour Checklist (CBCL) Child engagement in daily life (CDEL) Canadian Occupational Performance Measure (COPM) Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD) Quality of life of children with CP (CPQOL) Drooling Impact Scale (DIS) Dimension of Mastery Questionnaire (DMQ) Developmental Test of Visual Perception 2nd ed (DTVP-2) Gillette Functional Assessment Questionnaire (FAQ) Functional Independence Measure (FIM) Family Support Scale (FSS) Gross Motor Function Measure (GMFM) Gross Motor Performance Measure (GMPM) Harter-SPPC-CP (HSPPC-CP) Health Utilities Index (HUI3) Jebsen-Taylor Test of Hand Function (JTHF) KIDSCREEN Life-Habits questionnaire (LHQ) Measurement of Activities of Daily Living questionnaire (MADL) Melbourne Assessment of Upper Limb Function (MAULF) Mobility Questionnaire (MobQues) Participation and Environmental Measure children and youth Peabody Developmental Motor Scales (PDMS-2) Pediatric Evaluation of Disability Inventory (PEDI) Quality of Function Measure (QFM) Preschool Language Scales, Fifth edition (PLS-5) Pediatric Outcomes Data Collection Inventory (PODCI) Pediatric Volitional Questionnaire (PVQ) Quality of Life Instrument for People With Developmental Disabilities-children version (QOLDD) Quality of Upper Extremity Skills Test (QUEST) The Revised Children's Manifest Anxiety Scale (RCMAS) School Function Assessment (SFA) Self-Perception Profile for College Students (SPPCS) TNO-AZL questionnaire for Children's HRQOL (TACQOL) Toddler Infant Motor Evaluation (TIME) Test of Nonverbal Intelligence, 2nd edition (TONI-2) Vineland Adaptive Behavior Scales (VABS) Functional Independence Measure for children (WEEFIM)
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1. Introduction Cerebral palsy (CP) is a life-long neurodevelopmental disorder that impacts children’s and youth’s functional abilities and developmental trajectories. 1 As children and youth with CP transition from birth to adulthood, their needs and functional abilities change. As such, it is imperative to select appropriate measures to best capture these age-specific needs and to evaluate their response to interventions over time.
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Selecting appropriate measures for clinical and/or research applications for children and youth with CP poses many challenges. Several factors influence the selection of a suitable measure; particularly, the research question and/or the clinical purpose, the type of intervention, the psychometric properties, the availability of a client/proxy version, the age range, and the specific condition under investigation. Previous reviews have compared the characteristics of measures used in CP, providing useful information on measurement characteristics and theoretical backgrounds of a range of instruments.2-7
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Despite the breadth of information currently available, there remains a lack of consensus regarding which measures are best used in clinical practice and/or research studies with this population. Further, inconsistencies in the application of available measures call into question the ability to evaluate responses to interventions and to compare results across studies worldwide. Furthermore, a need has been identified to compile data on disability, including the functional impact of chronic conditions, such as CP. It has been recommended that such data be made internationally comparable in order to benchmark and monitor national and international progress on disability policies (The WHO Global Disability Action Plan 2014-2021). 8 Specific actions have been proposed, including the implementation of valid and reliable tools, consistent with the International Classification of Functioning, Disability and Health (ICF). 9
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Our research group conducted a content analysis of all multiple-item measures used as outcome measures in interventional and/or observational studies with children and youth with CP using the ICF. 10 Our previous work characterized the content of each measure used in CP based on the entire pediatric ICF classification. 11 From our findings, no multiple-item measure alone fully represents the components of the ICF; therefore, a combination of measures seemed most appropriate if the goal is to comprehensively describe the functional profile of a child or youth with CP. Due to the extensiveness of the ICF classification, it is recommended to apply ICFbased tools, such as the ICF Core Sets for specific health conditions or functional profiles, when available. An ICF Core Set is a shortlist of ICF categories that are considered most relevant for describing the functioning of an individual with a particular health condition. 12, 13 In keeping with the specific aims of the ICF ─ to establish a universal language for describing functional abilities, to permit comparison of data across countries, andto provide a systematic coding scheme for health information systems, among others ─ the newly developed ICF Core Sets for children and youth with CP 14 serve as universal guidelines for assessment, intervention and follow up. Following a rigorous multi-step methodology endorsed by the WHO, our research team ─ in collaboration with the ICF Research Branch of the WHO German Collaborating Centre for the Family of International Classifications ─ and international CP experts ─ representing all WHO health regions ─ developed five ICF Core Sets14 : 1) 4
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a comprehensive set covering all the developmental stages from birth up to 18 years of age consisting of 135 ICF categories; 2) a brief set consisting of 25 ICF categories that are common to describe children and youth with CP up to 18 years of age; 3) a 31-category brief set for children and youth with CP below 6 years of age; 4) a 35-category brief set for children and youth with CP ≥6 and <14 years of age; and 5) a 37-category brief set for children and youth with CP ≥14 and 18 years of age. To ensure international applicability, the development of the ICF Core Sets for children and youth with CP involved the international community; specifically the development methodology incorporated the perspectives of international health, education and social sciences professionals as well as the valuable perspectives of children and youth with CP and their caregivers. 10, 15-18 Finally, the five ICF Core Sets for children and youth with CP capture the developmental trajectory that children and youth with CP transition from birth to young adults, facilitating the characterization of the functional trajectory of this population over time. 19 Above all, the ICF Core Sets can guide professionals in selecting the most appropriate outcome measures to comprehensively capture functional information regarding children and youth with CP.
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When selecting an outcome measure, it is important to examine the psychometric properties of the measures, the conceptualization of constructs, and the content used to measure these. While numerous scales exist to measure patient outcomes, few measures have undergone rigorous psychometric testing to support the interpretations based on their use. Marshal et al., 20 conducted a systematic review of over 300 randomized control trials and found that studies were 40% more likely to report that treatment was effective when they used unpublished scales, rather than validated ones 20; thus, demonstrating the implications of measurement selection for patient care. In the field of CP, Hoare & Imms 21 reviewed 15 studies evaluating the effects of Botolinum toxin in the upper limb, and found that only 4 studies had used valid and reliable measures, which compromise the accuracy of the results. Contemporary validity researchers reason that psychometric considerations are relevant to researchers and consumers of measures, alike 22 As such; one must be critical and judicious when selecting measures to determine if they are valid and reliable for their intended use. Furthermore, since the publication of the ICF, clinicians and researchers are also encouraged to consider the “ICF mapping” of the content of available measures.
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Our research group have demonstrated that the content of multiple-item measures –defined as those measures comprising of several items, where each item is a single question, observation, or a statement that needs to be evaluated –varied considerably based on theirs ICF content representation, highlighting the differences between multiple-item measures and the importance of adding the ICF content information 10 Notably, we found that multiple-item measures highly capture functional domains identified as important areas of functioning by international experts, and most importantly, by children and youth with CP and their families. 10, 18 To that end, the focus of this study is to build upon our previous work in providing content analysis of multipleitem measures used with children and youth with CP10, and to recommend a minimum set of multiple-item measures that comprehensively represent the functional profile of this population. Specific objectives of this study were: 1) to examine the psychometric properties of measures used with children and youth with CP, 2) to map the content of each multiple-item measure using the ICF Core Sets for children and youth with CP as a framework, and finally 3) to propose a toolbox of valid and reliable measures aligning with the ICF Core Sets. 5
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2. Methods In this study, we followed a four-step methodology. Firstly we identified multiple-item measures applicable to children and youth with CP. Secondly, we categorized the purpose of each measure and its psychometric properties. Thirdly, we mapped the content of multiple-item measures ─with proven psychometric characteristics─ to the ICF Core Sets for children and youth with CP and identified a toolbox of measures representing the ICF Core Sets. Finally, we extracted the clinical utility characteristics of the measures included in the toolbox. Figure 1describes the methodology overview of this study.
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2.1 Step 1- Identifying multiple-item measures We used two sources for retrieving multiple-item measures used in CP. Source 1, a systematic review of the literature including original studies on children and youth with CP with a clearly defined outcome measure published between 1998-2012, detailed description of the methodology is available.10 Source 2, we conducted a consultation with clinical and research experts and a target literature search to identify newly developed and/or revised multiple-items measures published between January 2013 and March 2015. Overall, we identified 83 multipleitem measures, 78 measures were retrieved from source 110 and 5 measures were identified from source 2.
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2.2 Step 2- Measures categorization and psychometric analysis We categorized measures according to age-groups, generic and/or CP-condition specific, proxy and/or self-reported, and whether they were descriptive, evaluative or predictive. Next, we evaluated the psychometric properties of each instrument, focusing specifically on whether studies objectively reported the presence of reliability data, including: internal consistency, testretest, inter-rater and intra-rater reliability, and responsiveness, and validity data, including: content, construct, criterion, concurrent, and convergent validity. We considered psychometrically sound measures to be those measures that underwent both reliability and validity testing of at least four psychometric properties with adequate to excellent values ( ≥ 0.6, arbitrary criteria). Reliability here was defined as the consistency in properties of test responses, indicating the extent to which differences are true, as opposed to measuring error.23 Validity was defined as the degree to which evidence and theory support the interpretation of measurement results.23 See full glossary in Appendix A (online). 2.3 Step 3a- Mapping content to the ICF Core Sets for children and youth with CP We followed the ICF linking guidelines proposed by Cieza et al 24, which have been applied to a variety of outcome measures in adults and pediatric measures (e.g. DISABKIDS, cancer measures, CP disease-specific measures, and generic patient-reported outcomes). 10, 25-27The content of the measure is linked to the two parts of the ICF. Part 1 covers functioning and disability and includes the components of body functions (b) and structure (s) and activities and participation (d). Part 2 covers contextual factors and includes the components environmental factors (e) and personal factors (pf). In the classification, the letters b, s, d and e, which refer to the component of the classification, are followed by a numeric code starting with the chapter 6
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number adding one digit, (e.g d9-community life), followed by a second level code adding two digits (e.g d920-recreation and leisure), third or fourth level code by adding one digit (e.g d9200play). This linking procedure was performed separately by two trained assessors (VS, KS). Subsequently, disagreements were resolved by consensus. Inter-coders reliability of the linking process was 0.74 (95% confidence interval 0.73-0.75). Before linking each measure, the purpose of the measure and its manual were reviewed, a decision was made as to whether the full content (individual items) could be linked to the ICF Core Sets or only the main domains could be linked, for example the main domains of the AMPS were linked as the detailed information contained in the individual items would not have been capture by the ICF coding system resulting in redundant ICF information. Only original versions of the measures were linked. Finally, the full content of some multiple-item measures were unavailable because of access fees, as described in the articles, therefore the main domains were linked (e.g. WeeFIM).
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Step 3b- ICF-based Toolbox of multiple-item measures Once the content of all psychometrically sound measures were linked to the five ICF Core Sets, those measures whose content represent areas of “functioning” as defined by the ICF Core Sets for children and youth with CP were included in the ICF-based toolbox. Specifically, the toolbox inclusion criteria consisted of: 1) multiple-item measure with at least 4 tested psychometric properties of both reliability and validity with adequate to excellent values and 2) content of measure mapping ICF categories of the ICF Core Sets at the ICF second level (e.g. d920recreation and leisure).
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2.4 Step 4-Clinical Utility- Toolbox of multiple-item measures Finally, we collected information regarding the format (observation, proxy/self-report, performance, etc), required qualifications (Masters level, etc), estimated time for completion, and associated fees of each measure included in the ICF-based toolbox.
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2.5 Data analysis Descriptive analysis was conducted in SPSS. For each of the identified instruments, we calculated the frequency with which its items/domains addressed b, s, d, e and pf. If an ICFcategory was assigned repeatedly in a measure, the category was counted only once.
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3. Results 3.1 Multiple-item measures characteristics We found, 68 out of 83 multiple-item measures used in studies with children and youth with CP demonstrating evidence of validity and reliability. (Figure 1) The majority of the measures were discriminative, generic, proxy reported, and designed for school-aged children. 3.2 Multiple-item measures meeting study’s psychometric criteria, n=46 Table 1(online) shows the forty six multiple-item measures meeting the study’s psychometric criteria – measures that reported at least four tested psychometric properties within both reliability and validity domains with adequate to excellent values. On average, five psychometric properties were tested, with a range of four to nine.
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Measures demonstrating the greatest number of psychometrics, included: AMPS, FIM, GMFM88, MAULF, PDMS-2, PEDI, SFA, TONI-2, VABS, and WEEFIM. Generic functional measures appeared to have the highest number of reported psychometric properties (e.g. PEDI, FIM, and AMPS).
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When we assessed the purpose of the 46 psychometrically sound measures, 24 (52%) were discriminative, 7 (15%) were evaluative, 14 (30%) were both discriminative and evaluative, and only 3 were predictive measures. S measures were CP-specific (CPCHILD, CPQOL, GMFM, GMPM, QFM, and QUEST) and among them only one (CPQOL) was self-reported.
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3.3 Mapping to the ICF Core Sets for children and youth with CP n=42 measures We were able to link the content of 42 measures with proven psychometrics at the domain and item level. We were unable to link the content of 4 measures due to lack of access to the measures. The 42 measures varied considerably in the degree to which their content represented the ICF Core Sets for CP. Primarily, measures covered the ICF components of activities and participation. Self-care, mobility, and recreational and leisure were the most frequent areas represented by the measures. Overall, individual measures few categories comprising the ICF Core Sets. A single measure (PEM-CY) covered the majority of the environmental factors included in the ICF Core Sets.
Mapping to Comprehensive ICF Core Set for children and youth with CP (N=135 categories)
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The comprehensive ICF Core Set allows the complete and detailed description of functional abilities and relevant contextual factors in children and youth with CP aged 0 to 18 years. This Core Set is useful in interdisciplinary assessments because it provides extensive detail, offering exhaustive assessment that can distributed among multi-professional team members. 14
Mapping to Common Brief ICF Core Set for children and youth with CP (N=25 categories)
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Evaluative measures demonstrating the highest coverage of ICF categories included in this Core Set were: ASK, CPCHILD, COPM, FIM, PEDI, PODCI, SFA, and WEEFIM. Discriminative measures that aligned with the Comprehensive ICF Core Set include: ASK, CBCL, CPQOL, CDEL, KIDSCREEN, PEDI, PEM-CY, PLS-5, and VABS.
The Common Brief ICF Core Set describes the most common areas of functioning in children and youth aged 0 to 18 years with CP. This Core Set can be employed in regular clinical encounters where only a brief assessment is necessary, and in clinical and epidemiological research. This Core Set can be used by a single professional or an inter-disciplinary team. A key characteristic of the Common Brief Core Set is that it allows the description of the level of functioning of children and youth with CP over time, from birth to transition to adulthood. Therefore, this Core Set can be considered as a minimum for any “minimal data set” which describes children and youth with CP across their lifespan. 14
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The evaluative measures with the most coverage of the ICF categories included in this Core Set are: AMPS, CPCHILD, GMFM, PEDI, PDMS-2, PODCI, SFA, and WEEFIM. The discriminative measures that predominantly aligned with the Common Brief Core Set are: AMPS, CDEL, CPQOL, LHQ, PEM-CY, PEDI, PODCI, TACQOL, and VABS. The content of the PEM-CY mapped to all the environmental factors of the Common Brief ICF Core Set.
Mapping to Age-Specific Brief ICF Core Sets (N=31, 35, and 37 categories, respectively)
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Figure 2 illustrates the mapping of psychometrically sound measures to the Common Brief ICF Core Set. Few measures containing multiple attributes comprehensively cover the range of categories included in this Core Set (CPCHILD, CPQOL, PEDI, SFA, PEM-CY, and VABS). Two measures cover the components of body functions, activities and participation, and environmental factors (CPQOL and SFA). As shown in figure 2, the coverage of each ICF category included in the Core Set varies significantly across measures. The categories most commonly represented by the measures are: d550-eating, d530-toiletting, d450-walking, and d410 maintaining a body position.
Each of the age-specific sets includes the ICF categories of the Common Brief ICF Core Set in addition to ICF categories unique to each age-group: 0 to <6, 6≥ to <14 and 14≥ to 18 years of age. Each age-specific ICF Core Set describes the most common areas of functioning in children and youth with CP for the associated age-group. The Age-Specific ICF Core Sets can be applied in clinical or research context where additional detail is desirable in caring for or describing a particular age-group of children and youth with CP. These ICF Core Sets can be used by a single professional or an interdisciplinary team.
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The age range for which the measures were developed varies significantly. The content of few measures aligned with the Brief ICF Core Set for children <6 years of age; in particular for children < 3 years (n=l9/42). Similarly, fewer psychometrically sound measures aligned with the transition to adulthood Brief ICF Core Set, specifically for teenagers ≥ 16 years of age. A large number of measures addressed the school-aged Brief ICF Core Set.
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3.4 Proposed ICF Core Sets-based Toolbox of measures for children and youth with CP Table 2 shows the 25 measures included in the ICF-based toolbox. Measures are applicable to children and youth with CP aged 0 to 18 years, will all levels of functioning and all types of CP. The vast majority focused on school-aged children. For example, as shown in table 2, only two measures (CDEL and PDMS-2) are specific for children < 6 years of age. Three measures are specific for school aged and the transition to adulthood youth (CAPE, CBCL, and KIDSCREEN). Table 3 illustrates the characteristics and the representation of each component of the ICF Core Sets by the 25 measures included in the toolbox. As shown in table 3, the propose toolbox highly represents the component activities and participation, followed by the components body functions and environmental factors. All in all, the toolbox embraces functional areas included in the ICF Core Sets. (For full description of measures please see Appendix B, online). 3.5 Clinical utility: Clinical utility data were extracted for the 25 measures included in the toolbox. (Appendix C, online). Sixteen (67%) measures are administered via proxy report (parent/caregiver), and 13 9
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(54%) include a self-report version through which children and youth can share their perspectives. Eleven (44%) measures are based on observation by a clinician or teacher and seven (29%) measures require an interview with parent/caregiver and/or child.
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In terms of complexity and time to administer, the AMPs, CAPE, CBCL, COPM, FIM, GMFM, LHQ, PDMS-2, PEDI, PEM-CY, and QUEST require at least 30 minutes to administer and are more complex in terms of set up and/or scoring and administration. Nonetheless, these measures provide more comprehensive information regarding the child's functioning across various domains. Simple measures reflected those requiring between 10-20 minutes for administration, and included: AHA, ASK, CDEL, CPCHILD, CPQOL, FAQ, HUI3, KIDSCREEN, PODCI, and SFA (depending on the number of scales administered- SFA can fit in easy or more complex). Importantly, the toolbox includes measures applicable in different contexts - home, school, and community.
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4. Discussion This study identified a set of valid and reliable multiple-item measures assessing the relevant areas of functioning highlighted in the ICF Core Sets for children and youth with CP. The diverse purposes of the measures demonstrate the broad spectrum of functional impact of CP on children and youth. The majority of the measures were discriminative, proxy reported, and for school-aged children. By using the ICF Core Sets for CP as a common framework, clinicians and researchers can systematically incorporate an integrated functional approach when selecting appropriate measures for clinical practice and/or research studies.
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4.1 ICF Core Sets-based toolbox of multiple-item measures for CP Our work describes the first attempt to create an “ICF-bassed toolbox of measures for children and youth with CP” consisting of clinically feasible, psychometrically robust measures, across all levels of functioning and developmental stages. Using this toolbox, clinicians and researchers can choose the measures that best fit the purpose/s of their clinical and/or research applications and are also appropriate for each specific child or youth with CP they see. The measures included in this toolbox operationalize the implementation of the ICF Core Sets for children and youth with CP in clinical practice and research. The systematic draw of measures from this toolbox will bring international consensus on measurement approach and reporting of functional strengths and challenges of the pediatric CP population. In clinical practice, benefits of applying a set of measures from this toolbox includes a unified language for monitoring functional status in CP, ensuring each child's or youth’s needs are being met comprehensively, Although our work has summarized the extensive information on measures available today, and has narrowed the vast list of measures to a manageable ICF-based toolbox. The selection of the most appropriate measure still relies on clinical judgement of professionals and researchers. Given the heterogeneous nature of CP, selecting appropriate measures to meet the assessment needs of a specific child/youth can be challenging in clinical practice. We propose an overarching algorithm to guide professionals navigate the selection of the best fit measure. (Figure 3) It is expected that professionals will select one or a combination of measures out of the entire toolbox, depending on the clients’ goals of interventions, resources, availability of trained professionals, and access to instruments.
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As described by Wright and Majnemer 28 several factors need to be considered when creating a toolbox for children and youth with CP. Specifically, the purpose and scope of the measures, their psychometric characteristics, the inclusion of child and family’ perspectives, and the developmental consideration of each child and youth with CP. We made special efforts to incorporate all these key factors in our toolbox selection with the essential application of the ICF as the overarching framework. It is hoped that this novel toolbox will ease the overwhelming endeavour of selecting appropriate measures that clinicians and researchers face every day. Furthermore, our proposed toolbox, based on extensive data analysis, includes several of Wright’s and Majnemer’s suggested candidate outcome measures for a toolbox, in particular in the areas of activities and participation (multi domain) and motor function. 28
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Our findings are not limited to guiding the systematic assessment of functional abilities and/or challenges in children and youth with CP, but also allow the comparison of functional data worldwide. The latter can be achieved by encouraging researchers to refer to the detailed content analysis provided in this study (Appendix B, online) and by applying these guidelines globally. For example, if the purpose of the assessment is to capture information on “d710-basic interpersonal interactions” using the Common Brief ICF Core Set as a framework, professionals have the options of using the: CPQOL, KIDSCREEN, LHQ ,PEDI, PEM-CY, SFA, VABS and WEEFIM. (Figure 2)
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4.2 Comments on psychometrics properties We found that multiple-item measures lacked evaluation of responsiveness with only 25% of measures demonstrating some degree of responsivity. This is an important consideration for clinical care, as health care practitioners are often interested in monitoring functional progression or decline over time in order to guide intervention planning and care, which points to the need for further evaluation of measures in this area. We have noticed that some measures have been used as evaluative measures when they have not been tested for responsiveness to change (e.g. BSID).When using a measure as an evaluative tool, professionals should be aware of this important psychometric property to avoid misuse and misinterpretation of results.
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Notably, some measures are fairly new (e.g. Mini-AHA, Ad-AHA, PEDI-CAT, and QFM). Psychometric properties remain under investigation in these early stages and this is not to preclude their application and appropriateness for children and youth with CP. In addition, new measures might become available in the future and might be suitable candidates for the proposed toolbox. 4.3 Coverage of the ICF Core Sets for children and youth with CP by the toolbox Although the vast majority of ICF categories included in the five ICF Core Sets are represented by the toolbox. The representation varies within each ICF Core Set. Overall, the areas more frequently covered by our suggested toolbox are d4-mobility, d5-self-care, d7-interpersonal interactions and relationships, d8-major life areas, and d9-community, social and civil life. Some relevant areas of functioning are lacking measures with a high degree of psychometric testing with the CP population. For example, we identified a paucity of measures to assess cognitive abilities and communication skills (verbal and non-verbal). These aspects of functioning are critical to planning transition to adulthood services. Furthermore, few measures included in the toolbox assess “b280-sensation of pain”, none of them are pain-specific measures. Pain 11
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prevalence has been frequently reported in pediatric studies in CP, ranging from 14% to 73%; as such, it is imperative to incorporate into the routine functional assessment of children and youth with CP. 29
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Few measures offer a broad overview of environmental factors included in the ICF Core Sets for CP (KIDSCREEN, PODCI, SFA and PEM-CY). The PEM-CY is the only measure that addresses all the environmental factors categories included in the Brief ICF Core Sets and the majority of the Comprehensive ICF Core Set. Of note, the PEM-CY is a discriminative measure which helps identify important environmental factors that influence participation; however, there is a need for an evaluative tool that measures response to environmental interventions.
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4.4 Identified Gaps The paucity of self-reported measures for children and youth with CP is a major deficit. The ICF incorporates the convention of the rights of children’s and promotes the active inclusion of the child and/or youth’s perspective when assessing functional abilities. The children’s subjective experiences are as essential - or arguably greater - as more objective information, such as observed performance, to plan individualized family-centered care. Our results also point to the need for more measures to evaluate the needs of youth with CP, as this segment of the population is less represented by existing measures. Nonetheless, the age specific core set for this group (e.g. >14 years) can be used as a guide to capture functional areas using informal assessment (direct questions, observation, etc).
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We chose not to use the COSMIN (Consensus-based Standards for the selection of health status Measurement Instruments) checklist 30- to evaluate the clinimetric properties of each instrument as we used an adapted clinical utility description (Appendix C, online). The focus of this paper was to map psychometrically sound measures to the ICF Core Sets for CP, rather than to provide extensive clinimetric evaluation. Further studies can build upon these findings to conduct further analysis.
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4.5 Limitations Limiting studies to measures, which are in English, is a further limitation of our review. We encourage others who are familiar with non-English language measures to add to this body of literature by completing a similar content analysis exercise. Moreover, while the measures included in this review have been widely used with the CP population, psychometric analyses for some measures were completed with other populations (e.g. attention-deficit hyper active disorder); further research is needed to evaluate psychometric properties in children with CP, specifically. 4.6 Future directions Future work may broaden the scope of this toolbox including single-item measures to cover areas misrepresented by this multiple-item toolbox, such as muscle tone, mobility of joints, etc. In addition, future work can build upon these findings by developing self-report measures which include all categories of the ICF Core Sets. In the absence of such measures, professionals can gather information by interview, clinical examination, observation or technical testing and remediate the gaps of current standardized measures. 12
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4.7 Conclusion: This study used a robust methodology in order to identify a set of multiple-item measures for children and youth with CP. Using the ICF Core Sets as a framework, we recommend a toolbox which guides “how to measure” the most relevant areas of functioning in CP. The systematic application of this proposed toolbox of measures will standardize the measurement and report of key functional information in CP, facilitating international comparisons of findings across studies. Finally, our results will guide professionals seeking appropriate measures to meet children’s and youth’s with CP needs comprehensively.
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Acknowledgements Veronica Schiariti is the recipient of a NeuroDevNet & Child and Family Research Institute Postdoctoral Fellowship Award. This study was partially financially supported by a 2013 research grant from the American Academy of Cerebral Palsy and Developmental Medicine (AACPDM).
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We express our thanks to all the developers of measures who kindly shared their measures with us. We would also like to thank Child Health BC for its administrative support.
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Disclosure: The authors have stated that they had no interests which might be perceived as posing a conflict or bias.
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1. Rosenbaum P, Rosenbloom L. What is Cerebral Palsy? Hart H (ed) Cerebral Palsy from Diagnosis to Adult Life. London: Mac Keith Press, 2012: 3-13. 2. Harvey A, Robin J, Morris ME, Graham HK, Baker R. A systematic review of measures of activity limitation for children with cerebral palsy. Dev Med Child Neurol 2008; 50: 190-198. 3. Waters E, Davis E, Ronen GM, Rosenbaum P, Livingston M, Saigal S. Quality of life instruments for children and adolescents with neurodisabilities: how to choose the appropriate instrument. Dev Med Child Neurol 2009; 51: 660-669. 4. Debuse D, Brace H. Outcome measures of activity for children with cerebral palsy: a systematic review. Pediatric Phys Ther 2011 Fall;23(3):221-312011; 23: 221-231. 5. Carlon S, Shields N, Yong K, Gilmore R, Sakzewski L, Boyd R. A systematic review of the psychometric properties of Quality of Life measures for school aged children with cerebral palsy. BMC Pediatrics 2010; 10. 6. Thomas C. Body and Society: Some Reflections on the Concepts of ’Disability’ and 'Impairment’. Watson N, Cunningham-Burley S (eds) Reframing the Body. Basingstoke, Hampshire: Palgrave, 2001: 47-62. 7. James S, Ziviani J, Boyd R. A systematic review of activities of daily living measures for children and adolescents with cerebral palsy. Dev Med Child Neurol 2014; 56: 233-244. 8. World Health Organization. Disability Action Plan 2014-2021. Geneva: WHO; 2014. 9. World Health Organization. International Classification of Functioning, Disability and Health: Children & Youth version. Geneva: WHO; 2001. 10. Schiariti V, Klassen AF, Cieza A, et al. Comparing contents of outcome measures in cerebral palsy using the international classification of functioning (ICF-CY): A systematic review. Europ J Paediatr Neurol 2014; 18: 1-12. 11. World Health Organization. International Classification of Functioning, Disability and Health: Children & Youth version. Geneva: WHO; 2007. 12. Cieza A, Ewert T, Ustun TB, Chatterji S, Kostanjsek N, Stucki G. Development of ICF Core Sets for patients with chronic conditions. J Rehabil Med 2004; (44 Suppl): 9-11. 13. Fayed N, Cieza A, Bickenbach J. Illustrating child-specific linking issues using the Child Health Questionnaire. Am J Phys Med Rehabil 2012; 91: S189-98. 14. Schiariti V, Selb M, Cieza A, O'Donnell M. International Classification of Functioning, Disability and Health Core Sets for children and youth with cerebral palsy: a consensus meeting. Dev Med Child Neurol 2015; 57: 149-158. 15. Schiariti V, Sauve K, Klassen AF, O'Donnell M, Cieza A, Masse LC. 'He does not see himself as being different': The perspectives of children and caregivers on relevant areas of functioning in cerebral palsy. Dev Med Child Neurol 2014; 56: 853-861. 16. Schiariti V, Masse LC, Cieza A, et al. Toward the development of the International Classification of Functioning Core Sets for children with cerebral palsy: a global expert survey. J Child Neurol 2014; 29: 582-591. 17. Schiariti V, Masse LC. Relevant areas of functioning in children with cerebral palsy based on the international classification of functioning, disability and health coding system: A clinical perspective. J Child Neurol 2015; 30: 216-222. 18. Schiariti V, Masse LC. Identifying relevant areas of functioning in children and youth with Cerebral Palsy using the ICF-CY coding system: From whose perspective?. Europ J Paediatr Neurol 2014; 18: 609-617. 15
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19. Schiariti V, Selb M, Cieza A, O'Donnell M. International Classification of Functioning, Disability and Health Core Sets for children and youth with CP: contributions to clinical practice. Dev Med Child Neurol 2015; 57: 203-204. 20. Marshall M, Lockwood A, Bradley C, Adams C, Joy C, Fenton M. Unpublished rating scales: A major source of bias in randomised controlled trials of treatments for schizophrenia. British Journal of Psychiatry 2000; 176: ate of Pubaton: 2000. 21. Hoare B.J., Imms C. Upper-limb injections of botulinum toxin-A in children with cerebral palsy: A critical review of the literature and clinical implications for occupational therapists. Am J Occup Ther 2004; 58: 389-397. 22. Hoyt WT, Warbasse RE, Chu EY. Construct validation in counseling psychology research. Counsel Psych 2006; 34 (6): 769-805. 23. Furr M, F., Bacharach, V.,R. Psychometrics: An Introduction. . United States of America: Sage Publications, Inc., 2008. 24. Cieza A. ICF linking rules: an update based on lessons learned. J Rehab Med 2005; 37: 212. 25. Fava L, Muehlan H, Bullinger M. Linking the DISABKIDS modules for health-related quality of life assessment with the International Classification of Functioning, Disability and Health (ICF). Disab Rehab 2009; 31: 1943-1954. 26. Fayed N, Schiariti V, Bostan C, Cieza A, Klassen A. Health status and QOL instruments used in childhood cancer research: deciphering conceptual content using World Health Organization definitions. Qual Life Res 2011; 20: 1247-1258. 27. Schiariti V, Fayed N, Cieza A, Klassen A, O'Donnell M. Content comparison of healthrelated quality of life measures for cerebral palsy based on the International Classification of Functioning. Disab RehaB 2011; 33: 1330-1339. 28. Wright FV, Majnemer A. The concept of a toolbox of outcome measures for children with cerebral palsy: Why, what, and how to use?. J Child Neurol 2014; 29: 1055-1065. 29. Penner M, Xie WY, Binepal N, Switzer L, Fehlings D. Characteristics of pain in children and youth with cerebral palsy. Pediatrics 2013; 132: e407-e413. 30. Mokkink LB, Terwee CB, Knol DL, Stratford PW, Alonso J, Patrick DL, Bouter LM, de Vet HCW. The COSMIN checklist for evaluating the methodological quality of studies on measurement properties: A clarification of its content. BMC Medical Research Methodology 2010;10:22.
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Figures attached as separate files, please see titles belows Figure 1. Overview of study methodology Figure 2. Psychometrically sound measures aligned with the Common Brief ICF Core Set for CY with CP aged 0 to 18 years
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Footnote Figure 2 *Measures with highest coverage shown in this Figure, for full descriptions please see Appendix B online. Full names of measures and references provided on Table 1, online. NOTE: The figure shows only the categories that aligned with the Common Brief ICF Core Set.
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Figure 3. ICF-based Toolbox algorithm
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Tables 1, 2, 3 attached as separate files
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Table 2. ICF Core Sets-based Toolbox for children and youth with CP
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below 6 ≥6 to 14 ≥14 to 18
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Assisting Hand Assessment (Kids-AHA) Assessment of Motor and Process Skills (AMPS) Activities Scale for Kids (ASK) Children's Assessment of Participation and Enjoyment (CAPE) Child Behaviour Checklist (CBCL) Child engagement in daily life (CDEL) Canadian Occupational Performance Measure (COPM) Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD)
Age-Specific ICF Core Sets
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Valid and Reliable measures that aligned with the Comprehensive and Common Brief Core Sets for children and youth with CP
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Quality of life of children with CP (CPQOL) Gillette Functional Assessment Questionnaire (FAQ) Functional Independence Measure (FIM) Gross Motor Function Measure (GMFM) Health Utilities Index (HUI3) KIDSCREEN Life-Habits questionnaire (LHQ) Peabody Developmental Motor Scales (PDMS-2) Pediatric Evaluation of Disability Inventory (PEDI) Participation and Environmental Measure children and youth (PEM-CY) Preschool Language Scales, Fifth edition (PLS-5) Pediatric Outcomes Data Collection Inventory (PODCI) Quality of Upper Extremity Skills Test (QUEST) School Function Assessment (SFA) TNO-AZL questionnaire for Children's HRQOL (TACQOL) Vineland Adaptive Behavior Scales (VABS) WeeFIM (WEEFIM)
Full reference shown on Table 1 online
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Generic vs CP specific
d.e d ,e d,e d,e d d e e d d, e e d, e d,e,p d d d, e d, e
G¥ G G G G G G CP CP G G CP G G G G G
5-15y 0-7.11y
d d
G G
PODCI QUEST SFA* TACQOL VABS WEEFIM*
2- 18y 1.5-8y 5-13 y 5-18y 0-18y 0.5-18y
d, e e e d d d,e
G CP G G G G
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Observation Observation Child report Self report/Interview Parent report Parent/Caregiver Report Semi-structured interview Caregiver proxy Caregiver proxy and child report Self or proxy measure Observation Observation Observ. And/or self or proxy report Self/Proxy Interview/Proxy/Self-Questionnaire Observation Observation/Parent/Caregiver interview or Self Report Parent/Caregiver Report Observation/Parent/Caregiver interview Parent/Caregiver/Self-report Observation Proxy report (Clinician/Teacher) Parent/Caregiver/Self-report Interview/Teacher form Proxy report (Clinician/Caregiver)
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Content representation by ICF Components within the ICF Core Sts Body Activities & Environmental Functions Participation Factors
Format
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Asses Purpose
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18m-12y AHA > 2y AMPS*ª 5-15y ASK 6-21y CAPE 6-18y CBCL¶ 1.5-5y CDEL All COPM* 5-18y CPCHILD 4-12y CPQOL 2-17y+ FAQ 0.5 -18y FIM 5m-18y GMFM-88 5y+ HUI3 KIDSCREEN 8-18y 3-18y LHQ* 0-5y PDMS-2 0.5-7.5y PEDIª
Type of ICF Core Set covering age range of measure
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Toolbox measures
*Content linked to the ICF Core Sets at the domain level; ¶ no information available on psychometric studies including CY with CP; ª small representation of CY with CP in psychometric studies ; G, generic;G¥, unilateral disability; CP, CP-specific; d, discriminative; e, evaluative; p, predictive. Comprehensive ICF Core Set; Common Brief ICF Core Set; AgeSpecific Brief ICF Core Set for children <6y; Age-Specific Brief ICF Core Set for children and youth; Age-Specific Brief ICF Core Set for youth aged 14 to 18y ICF linking representation, Content of measure includes:
mild,
moderate,
high,
For full names of measures please refer to abbreviation section, references shown in Table 1 online
very high
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Highlights • First ICF-based toolbox of measures for children and youth with CP is proposed. • The twenty-five multiple-item measure toolbox operationalizes the ICF Core Sets for children and youth with CP. • No single psychometrically robust measure fully represents the content of the ICF Core Sets for CP. • The toolbox guides professionals seeking comprehensive valid and reliable measures.