DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY REVIEW

A common data language for clinical research studies:

the National Institute of Neurological Disorders and
Stroke and American Academy for Cerebral Palsy and
Developmental Medicine Cerebral Palsy Common
Data Elements Version 1.0 recommendations

VER ONICA SCHIARITI 1,2 | EILEEN FOWLER 3 | JOLINE E BRANDENBURG 4 | ERIC LEVEY 5 | SARAH MCINTYRE 6 |
THERESA SUKAL-MOULTON 7 | SHARON L RAMEY 8 | JESSICA ROSE 9 | SUSAN SIENKO 10 |
ELAINE STASHINKO 11 | LAURA VOGTLE 12 | ROBIN S FELDMAN 13 | JAMES I KOENIG 14
1 Division of Medical Sciences, University of Victoria, Victoria, BC; 2 Department of Pediatrics, University of British Columbia, Vancouver, BC, Canada. 3 University of
California Los Angeles, Los Angeles, CA; 4 Department of Physical Medicine and Rehabilitation, Department of Pediatric and Adolescent Medicine, Mayo Clinic,
Rochester, MN; 5 Health Services for Children with Special Needs, Inc., Washington, DC, USA. 6 Cerebral Palsy Alliance, University of Sydney, Sydney, NSW,
Australia. 7 Department of Physical Therapy and Human Movement Sciences, Feinberg School of Medicine, Northwestern University, Chicago, IL; 8 Virginia Tech
Carilion Research Institute, Virginia Polytechnic Institute, Roanoke, VA; 9 Stanford University School of Medicine, Stanford, CA; 10 Shriners Hospitals for Children,
Portland, OR; 11 Kennedy Krieger Institute, Baltimore, MD; 12 University of Alabama at Birmingham, Birmingham, AL; 13 The Emmes Corporation, Rockville, MD;
14 National Institute of Neurological Disorders and Stroke, National Institutes of Health, Bethesda, MD, USA.
Correspondence to Veronica Schiariti, Division of Medical Sciences, University of Victoria, PO Box 1700 STN CSC, BC V8W 2Y2, Canada. E-mail: [email protected]

On behalf of the American Academy for Cerebral Palsy and Developmental Medicine Cerebral Palsy Common Data Elements working groups.
This article is commented on by Fairhurst on page 968 of this issue.

To increase the efficiency and effectiveness of clinical research studies, cerebral palsy (CP)
PUBLICATION DATA specific Common Data Elements (CDEs) were developed through a partnership between the
Accepted for publication 17th January National Institute of Neurological Disorders and Stroke (NINDS) and the American Academy
2018. of Cerebral Palsy and Developmental Medicine (AACPDM). International experts reviewed
Published online 15th March 2018. existing NINDS CDEs and tools used in studies of children and young people with CP. CDEs
were compiled, subjected to internal review, and posted online for external public comment
ABBREVIATIONS in September 2016. Guided by the International Classification of Functioning, Disability and
AACPDM American Academy of Cerebral Health framework, CDEs were categorized into six domains: (1) participant characteristics; (2)
Palsy and Developmental health, growth, and genetics; (3) neuroimaging; (4) neuromotor skills and functional assess-
Medicine ments; (5) neurocognitive, social, and emotional assessments; and (6) engagement and qual-
CDE Common Data Element ity of life. Version 1.0 of the NINDS/AACPDM CDEs for CP is publicly available on the NINDS
CRF Case report form CDE and AACPDM websites. Global use of CDEs for CP will standardize data collection,
ICF International Classification of improve data quality, and facilitate comparisons across studies. Ongoing collaboration with
Functioning, Disability and international colleagues, industry, and people with CP and their families will provide mean-
Health ingful feedback and updates as additional evidence is obtained. These CDEs are recom-
NINDS National Institute of mended for NINDS-funded research for CP.
Neurological Disorders and
Stroke

In this era of information, with a vast number of outcome In 2006, the National Institute of Neurological Disor-
measures developed for complex neurological conditions, ders and Stroke (NINDS) initiated the development of
clinicians and researchers find it increasingly challenging CDEs to assist NINDS-funded investigators in collecting
to decide core concepts and how to most accurately mea- neuroscience clinical trial research data in a standard and
sure important areas of health. To guide standardization consistent fashion.1,2 The CDEs are content standards that
for data collection and outcome evaluation, the clinical and can be applied to various data collection models, are
research community recognized the need to develop Com- intended to be dynamic, and may evolve over time. The
mon Data Elements (CDEs). The overarching aim of CDE project is not a database, rather it is a collection of
developing condition-specific CDEs is to accelerate metadata and data standards. The CDEs consist of identifi-
research, to enable prompt uptake of evidence into clinical cation of common definitions, the standardization of case
practice, and, ultimately, to deliver the best quality care to report forms (CRFs), and tools. To date, the NINDS
individuals who require it. CDE website contains metadata and data standards for 23

976 DOI: 10.1111/dmcn.13723 © 2018 Mac Keith Press

neurological diseases for adult and paediatric populations What this paper adds
for NINDS-funded clinical studies. However, NINDS • This is the first comprehensive Common Data Elements (CDEs) for children
CDEs were not available for children and young people and young people with CP for clinical research.
with cerebral palsy (CP). Moreover, the need to develop a • The CDEs for children and young people with CP include common defini-
common data set of meaningful measurements for CP was tions, the standardization of case report forms, and measures.
identified as a priority area of the 2013–2017 strategic plan
• The CDE guides the standardization for data collection and outcome evalua-
tion in all types of studies with children and young people with CP.
developed by the American Academy of Cerebral Palsy and • The CDE ultimately improves data quality and data sharing.
Developmental Medicine (AACPDM).
CP describes a group of disorders that affect the devel- tools should be especially helpful to new investigators and
opment of movement and posture, causing activity limita- others working with limited budgets; (3) encourage focused
tions, which are attributed to non-progressive disturbances and simplified data collection to reduce burden on investi-
that occurred in the developing fetal or infant brain.3 The gators and practice-based clinicians to facilitate their par-
motor disorders of CP are often accompanied by distur- ticipation in clinical research; (4) improve data quality
bances of sensation, cognition, communication, perception, while controlling cost by providing uniform data descrip-
and/or behaviour and/or by a seizure disorder.3 tions and tools across NINDS-funded clinical studies.1
Since the 2001 publication of the International Classifi- The overall aim of this NINDS/AACPDM CDE project
cation of Functioning, Disability and Health (ICF)4 there was to standardize data collection and assessment in studies
has been increased interest in not only standardizing data of children and young people with CP by: (1) recommend-
collection, but also in understanding the functional abilities ing a set of CDEs and tools that comprehensively repre-
of individuals living with CP and the challenges they face sent functional profiles; (2) identifying valid and reliable
in performing everyday activities. After the publication of measures to be recommended for routine use across
the ICF for children and young people in 2007,5 ICF- NINDS-funded clinical studies and clinical practice; (3)
based tools – entitled ICF Core Sets6 – were developed for identifying CDEs/measurement gaps that could be
children and young people with CP worldwide. The ICF addressed in future research initiatives.
Core Sets serve as useful tools to standardize the descrip-
tion of the functional abilities and challenges children and METHOD
young people with CP have in performing everyday activi- Development of NINDS/AACPDM CDEs for children and
ties, and the contextual factors that facilitate or hinder young people with CP
functioning. Subsequently, an ICF-based toolbox of multi- The CDE project for CP began in 2015. An organizing
ple-item measures (e.g. questionnaires, standardized tests) committee with representatives from AACPDM and the
was proposed.7 This newly developed toolbox guides how NINDS was convened at an international meeting.
to measure the relevant areas of functioning, disability, and AACPDM leadership created a Steering Committee and
health included in the ICF Core Sets for CP. Hence, inter- worked with the AACPDM Research Committee to invite
national initiatives – such as the CDEs and ICF-based experts in CP into working groups, each of which was
tools – have recognized the importance of universal, stan- composed of five to seven members with knowledge and
dardized, and comprehensive data collection. Despite dif- experience relevant to CP-related health domains. These
ferences in methodological approaches, the CDEs and experts included epidemiologists, clinicians, clinical
ICF-based tools put special emphasis on measures for researchers, educators, and clinical trial experts. The work-
CP.6,7 ing groups were organized into the following domains: (1)
Previous reviews have compared measures used in CP, participant characteristics; (2) health, growth, genetics,
providing valuable information on measurement comorbidities, and labs; (3) neuroimaging diagnostics; (4)
characteristics and theoretical backgrounds of a range of neuromotor skill and functional assessments; (5) neurocog-
measures.8–14 A recent systematic review of outcome mea- nitive, social, and emotional assessments; (6) engagement
sures used in studies with children and young people with and quality of life assessments. Moreover, the integrated
CP showed that clinicians and researchers used different across working group was created to oversee the data.
measures to assess the same concept/construct.15 This find- Composition of each working group is provided in
ing demonstrates the variability in practice and lack of con- Appendix S1, online supporting information.
sensus on data collection, challenging the interpretation of The Chair of each working group, together with their
results. To address this issue, the AACPDM partnered Steering Committee liaison, constituted an advisory team
with the NINDS CDE team to create the paediatric CDEs that communicated throughout the development phase to
for CP. This paper reviews the process by which experts coordinate goals and identify shared solutions. Each work-
gathered in working groups and developed the first version ing group was tasked with identifying existing CDEs and/
of CDEs for children and young people with CP. or tools in the assigned domain and providing recommen-
The goals of the NINDS CDE project are to: (1) dis- dations for their use in clinical studies with children and
seminate standards for data collected from participants young people with CP. The working groups developed
enrolled in studies of neurological diseases; (2) create easily CRFs by selecting the most relevant items from existing
accessible tools for clinical study data collection – these NINDS CDEs and tools for other diagnoses, or identified

Review 977
and recommended the use of copyrighted tools for CP. The following selection criteria were applied by all
When necessary, they developed new CDEs and recom- working groups: (1) applicable to children and young peo-
mendations de novo. Brief details of how this was accom- ple aged 0 to 18 years; (2) represents relevant areas of
plished in individual working groups are described later. study in CP; and (3) has documented validity and reliabil-
Assessment measures and/or outcome measures were ity. Each of the working groups proceeded with slightly
selected based on the reliability and validity of the tool in different approaches that were largely dependent on the
children and young people with CP. Tools could be a sin- status of existing data standards and elements (see ‘Individ-
gle question (single-item measure), a questionnaire (multi- ual methods of each of the working groups’). CDEs and
ple-item measure), a score obtained through physical tools had to meet all selection criteria to be included in
examination, a laboratory measurement, or a score the final set for CP.
obtained through observation of an image. After compiling the CDEs and definition tables, recom-
mendations, and CRFs from each of the working groups
Terminology of NINDS CDEs representing the six CDE domains, the draft documents
Consistent with guidance across the NINDS CDE project, were sent to the integrated across working group to look
the working groups were charged with classifying each rec- for gaps, overlaps, and to make recommendations for the
ommended CDE and tools as ‘Core’, ‘Supplemental – inclusion of CDEs and tools. Descriptive analysis was con-
Highly Recommended’, ‘Supplemental’, or ‘Exploratory’ ducted to remove duplications and identify distribution of
(Table I). CDEs by level of recommendation. Subsequently, all the
measures recommended as ‘Supplemental – Highly Recom-
Conceptual framework mended’ were mapped into the ICF components and chap-
Managing the depth and breadth of data related to CP ters. Revised documents were then disseminated for
required an organizational framework to help visualize the internal review by the full panel of working group experts,
essential data categories. Given the multidimensional the Steering Committee, and the NINDS CDE project
impact of having CP on developmental trajectories of chil- team (June 2016). The draft CDEs for CP were posted on
dren and young people and their families,16–20 it was essen- the NINDS CDE website for public review from Septem-
tial to use a comprehensive framework such as the ICF.4 ber 2016 to October 2016. The final version, Version 1.0
Hence, NINDS domains were populated with the most NINDS CDEs for CP, was posted on the NINDS CDE
appropriate CDEs and measures incorporating the ICF website on December 15th, 2016 after the incorporation of
components (Fig. 1). comments received from the public review (Fig. 1).

CDE revision process and selection criteria Individual methods of each of the working groups
Individual working groups met via teleconference Participant characteristics
monthly for 6 to 9 months. As a starting point, all This working group conducted a review of common data
working groups reviewed the existing list of CDEs and variables collected by CP registers from around the
tools previously defined from other diagnostic groups, world,21 most notably demographic and disease classifica-
including Friedrich’s ataxia, stroke, epilepsy, Duchenne tion data elements, to ensure that the CDEs for this initia-
muscular dystrophy/Becker muscular dystrophy, spinal tive were inclusive. All CDEs and tools selected were
muscular atrophy, and traumatic brain injury, in the reviewed and discussed by the members of the working
NINDS CDE project. groups to determine the classification (core or supplement)
based on the NINDS classification criteria.

Table I: National Institute of Neurological Disorders and Stroke (NINDS) Health, growth, genetics, comorbidities, and labs
Common Data Elements (CDEs) classification criteria Each working group member independently reviewed 500
already established CDEs to determine if it applied to CP
CDE classification Definitions
and to the health, growth, genetics, comorbidities, or labs
Core A data element for recording essential topic areas. Each element was reviewed by two working
information applicable to any CP study,
including therapeutic areas and study group members; in the case of disagreement, the element
designs; consistent with all NINDS disease- was discussed by the working group and a resolution was
specific CDE sets reached. Working group members independently drafted
Supplemental – A data element recommended for use
Highly whenever applicable modifications to existing CRFs for other diseases, or cre-
Recommended ated new CRFs if needed, and the draft was discussed by
Supplemental A data element that has some evidence of the group during a teleconference to create updated ver-
validity and is commonly collected in clinical
studies in CP; use depends upon study sions of each CRF.
design
Exploratory A data element that is emerging or that Engagement and quality of life assessments/data
requires further validation in CP
The working group developed a list of measures for review
CP, cerebral palsy. by surveying systematic reviews and reviewing the

978 Developmental Medicine & Child Neurology 2018, 60: 976–986

 NINDS CDEs domains for CP

Health,
Neuromotor Neurocognitive, Engagement
growth,
Participant Neuroimaging skills and social, and and quality
genetics,
characteristics diagnostics functional emotional of Life
comorbidities,
assessmets assessments assessments
and labs

6 WGs proposed CDEs+1 WG compared CDEs across WGs

ICF framework
CDEs = CRFs Tools

CDEs for children and young people with CP – Version 0.0

Internal review Public feedback

CDEs for children and young people with CP – Version 1.0

Input from stakeholders Final revision

Figure 1: National Institute of Neurological Disorders and Stroke (NINDS) Common Data Elements (CDEs) for Cerebral Palsy (CP) Project: workflow and
conceptual framework. Tools include multiple-item measures, single-item measures, classifications systems, a score obtained through physical examina-
tion, a laboratory measurement, or a score obtained through observation of an image. Composition of each working group (WG) is provided in
Appendix S1, online supporting information. CRFs, case report forms; ICF, International Classification of Functioning, Disability and Health. [Colour figure
can be viewed at wileyonlinelibrary.com].

literature on engagement and quality of life tools used with This working group focused on developing CDEs and a
children with CP. The list of tools was distributed among CRF to document the characteristics and findings obtained
the working group members for review and additional using conventional clinical MRI of the brain. The working
measures were added based on clinical expertise. A litera- group decided to defer the development of CDEs or CRFs
ture review was performed for each measure by a member for diffusion tensor imaging, although diffusion tensor
of the working group using the NINDS/AACPDM- imaging is a promising neuroimaging tool for studying
defined selection criteria. Preference was given to validated brain microstructure in children with CP. The working
measures that were developed for children with CP. Using group did not specifically address use of ultrasound, com-
the tabulated information, all measures were reviewed and puted tomography, or functional MRI.
discussed to determine the appropriate classification based
on the NINDS classification criteria. Neuromotor skills and functional assessments
The Chair of the working group reviewed CDEs and tools
Neuroimaging diagnostics related to physical and neurologic examination, neuromotor
The working group searched for existing tools that were skills, physical function assessments, rehabilitation thera-
developed for documenting or interpreting neuroimaging pies, and adaptive equipment that were included for other
findings in children with CP and infants and children at disease groups. The entire working group reviewed these
risk of developing CP. While there were no commonly CDEs and tools, and narrowed this list to include those
accepted tools/tools for neuroimaging in CP, several neu- that were applicable to CP. Working group members were
roimaging assessment tools were identified and included. then assigned to CDE areas that matched their areas of
Published reports of abnormal brain magnetic resonance expertise and each member performed an electronic search
imaging (MRI) findings in children with CP were reviewed. for systematic and narrative reviews to identify tools that

Review 979
were deemed most reliable and psychometrically sound. Participant characteristics
Tools were selected that were psychometrically sound, The few CDEs classified as ‘Core’ included date of birth,
commonly used in CP research for children aged 0 to country of birth, country of residence, and sex. Only four
18 years, filled gaps in outcome measures/functional assess- tools were specific to individuals with CP. CDEs classified
ments, or were promising. Many CDEs that were already as ‘Supplemental – Highly Recommended’ were maternal
in use for other disease groups were modified to fit the date of birth, level of education attained, health insurance,
accepted nomenclature for CP as determined through a postneonatal onset (exact cause), gestational age, birth-
review of literature. When the working groups could not weight, multiple birth, predominant, and secondary motor
identify a CDE that captured the important information, a type. Many CDEs identified by this working group were
new CDE was created. When questions arose, consensus classified as ‘Supplemental’.
was reached through discussion. A particularly critical focus
for this working group was to identify and collate occupa- Health, growth, genetics, comorbidities, and labs
tional therapy, physical therapy, and speech and language The ‘Supplemental’ elements proposed are expected to be
pathology CDEs and tools. Specifically, occupational ther- used depending on the focus of a given clinical trial.
apy, physical therapy, and speech and language pathology Efforts were made to give general and supporting addi-
CDEs were selected based on gaps and needs in rehabilita- tional levels of detail that may be appropriate for a given
tion research (the need to determine the association study. Checklists and tables were used for the history
between frequency, intensity, timing, and type of rehabilita- forms to facilitate efficient data collection. There are a few
tion intervention and outcomes), published literature, and ‘Supplemental – Highly Recommended’ variables that
clinical expertise. The CDEs and created CRFs were based should be prioritized if collecting data related to those
on three key articles,22–24 reviewed by three or more thera- constructs. In the review of existing CDEs, many relevant
pists in each discipline, and modified to reflect CP practice. CDEs were identified but were validated in diseases other
than CP; therefore, these were classified as ‘Exploratory’.
Neurocognitive, social, and emotional assessments
This working group reviewed CDEs from other paediatric Engagement and quality of life assessments
diagnoses, including spinal cord injury – paediatrics, mito- No tools reviewed by the group were classified as ‘Core’.
chondrial disease – paediatrics, Duchenne muscular dystro- Only three quality of life measures were classified as ‘Sup-
phy/Becker muscular dystrophy, epilepsy (diagnosis by age plemental – Highly Recommended (Disease Specific)’. The
included), and traumatic brain injury – including early remaining quality of life measures were classified as
childhood. Like CP, these diagnoses are predominately ‘Exploratory’ owing to their limited use in children with
motor diseases often with some communicative and cogni- CP and limited age range. For participation measures, only
tive impairment. The working group finalized specific three measures were classified as ‘Supplemental – Highly
domains considered to be of central importance for CP. Recommended’ as they have been widely used in the
Outcome measures for the domains: language – speech, assessment of participation in children with CP and have
expressive, and receptive; cognitive and neurocognitive demonstrated good psychometric properties in individuals
executive function; intelligence and general cognition; with CP. Many of the tools reviewed were not specific for,
executive function and attention; adaptive behaviour; devel- or differentiated between, types or levels of CP motor
opmental milestones for the infant/toddler age range; impairment.
social–emotional development; and behavioural problems
were considered for inclusion/exclusion. In discussions, this Neuroimaging and diagnostics
working group strongly advocated that clinical and longitu- This working group included a comprehensive list of
dinal studies of individuals with CP be considered, which CDEs that would allow for coding and documentation of
included several assessments that capture important aspects lesions that would be found in any of the types of CP, i.e.
of cognitive and social–emotional development. spastic CP (cortical lesions, periventricular white matter
injury, and diffuse white matter injury), dyskinetic CP
RESULTS (basal ganglia lesions), ataxic CP (cerebellar lesions), or
The complete list of CDEs and tools and recommendation mixed types. The CRF developed allows for documenta-
for their use can be found on the NINDS CDEs (https:// tion of both acute and chronic changes on a single form.
www.commondataelements.ninds.nih.gov/#page=Default) Future efforts should focus on classification of structural
and AACPDM (http://www.aacpdm.org/) websites. Over- MRI findings, interpretation, and prognostication based on
all, few CDEs were classified as ‘Core’ and ‘Supplemental those findings.
– Highly Recommended’. None of the measures was classi-
fied as ‘Core’. A summary of CDEs classified as ‘Core and Neuromotor skills and functional assessments
Supplemental – Highly Recommended’ is available in A total of 92 tools were included (Table II). No tools were
Appendix S2, online supporting information. A brief classified as ‘Core’. Twelve tools inclusive of ICF domains
description of the working group findings is provided in of body structures and functions and activities and partici-
the following subsections. pation were categorized as ‘Supplemental – Highly

980 Developmental Medicine & Child Neurology 2018, 60: 976–986

Table II: Number of tools and case report forms (CRFs) reviewed and
System, Manual Ability Classification System, and Com-
recommended by working groups
munication Function Classification System – were included
within the list of ‘Supplemental – Highly Recommended’
Number tools by different working groups. In addition, 11 CRFs
Working group of tools CRFs
were recommended by the working groups (Table II). An
Participant characteristics 8 3 example of a recommended CRF can be found in
Health, growth, genetics, comorbidities, 28 11
Appendix S3, online supporting information.
and labs
Neuromotor skill and functional 92 6 Table III shows the tools recommended as ‘Supplemen-
assessments tal – Highly Recommended’ by the working groups. In
Neurocognitive, social, and emotional 66 NA
addition, tools are organized by NINDS domains, ICF
assessments
Engagement and quality of life 32 NA components, and ICF chapters. The tools highly repre-
assessments sented the ICF component body functions followed by the
Neuroimaging NA 1
component activities and participation; many measures
Total 226 20
covered areas of neuromusculoskeletal and movement-
NA, not applicable. related functions, intellectual functions, general tasks and
demands, and mobility.

Recommended’ (Table III). Specific tolls included in the DISCUSSION

‘Supplemental – Highly Recommended’ group included The NINDS/AACPDM CDEs for children and young
Gross Motor Function Classification System – Expanded people with CP (Version 1.0) are now available for clinical
and Revised, Manual Ability Classification System, Eating practice and research. The proposed CDEs for CP are the
and Drinking Ability Classification System, and Communi- starting point to achieving standardization and universal
cation Function Classification System. Sixty-nine tools data collection across NINDS-funded clinical studies. Ben-
were classified as ‘Supplemental’. Eleven tools were catego- efits of the widespread use of these CDEs include facilitat-
rized as ‘Exploratory’. ing data sharing across a wide range of study types.
Importantly, sharing CDEs and cross-referencing with
Neurocognitive, social, and emotional assessments other international measurement initiatives for CP will
This working group included standardized assessments of enable a common language across the full spectrum of
neurocognitive and social–emotional development that cap- clinical research studies worldwide. While many of the
ture the full developmental potential of children with CP, CDEs for CP are used across other domains and diseases,
from the youngest ages through to and including adult- some of the CDEs were created de novo and will require
hood. The working group identified many standardized further scrutiny regarding use for CP research. In addition,
tools designed originally for children who are typically measures for CP continue to be developed and others
developing that are included in studies of children with CP improved upon. With the creation of the CDEs for chil-
but have not necessarily been subjected to formal psycho- dren and young people with CP (Version 1.0), we hope
metric inquiry. Further, tests for younger children have that this will encourage researchers to continue this pro-
often been used with older children with CP when their cess along with creating and validating measures to fill
developmental stage seems matched to the research ques- gaps in research for children and young people with CP.
tion (e.g. children who are age-delayed in language acquisi- Following the CDE recommendations for CP, research-
tion). In many but not all study samples, the assessors ers and clinicians can select the most appropriate set of
make reasonable accommodations as to how they adminis- CDEs to best design and conduct their research or clinical
ter the test or the timing of the items, adjusting to the study. Tutorials demonstrating how to access and apply
individual profile of a child with CP. To what extent this the CDEs are available on the NINDS CDE website. In
alters the reported reliability and validity estimates is sel- addition, a demonstration video was created to illustrate
dom known with high levels of confidence. how to access, select, and apply the CDEs for CP (https://
www.youtube.com/watch?v=C9CqmFAHtvE).
Integrated across working group
The integrated across working group compiled and Challenges creating CDEs for CP
reviewed all the tools that the six other working groups The biggest challenge when creating the CDEs for CP
recommended (Table II). As expected, a vast number of was the heterogeneity of the condition, with regards to
measures were identified by the working groups (n=226). both clinical presentation and aetiology.
Of the initial 226 tools reviewed by the working groups,
122 (54%) were proposed for inclusion in Version 1.0 CP Health, growth, genetics, comorbidities, and labs
CDEs. Of these, 34 were classified as ‘Supplemental – There is no genetic or other test to biologically confirm
Highly Recommended’; 61 as ‘Supplemental’; and 31 as the diagnosis, and laboratory tests that may be related to
‘Exploratory’. Of note, some valuable classification systems comorbidities were thought to be more extensive than rea-
specific for CP – Gross Motor Function Classification sonable for the first iteration of CDE recommendations.

Review 981
Table III: List of ‘Supplemental – Highly Recommended’ tools (including measures and classifications) by National Institute of Neurological Disorders
and Stroke (NINDS) domains and International Classification of Functioning, Disability and Health (ICF) components/chapters

Speech,
Spasticity/ language, and
NINDS domain ICF component/chapters Classification Motor function movement communication
25,26
Neuromotor skill Body functions: neuromusculoskeletal EDACS AIMS27,28 BADS29 PPVT-430
and functional and movement-related functions GMFCS-ER31 COPM32,33 Tardieu Scale34
assessments Mental functions MACS35 GMFM-88, GMFM-6636–38
Functions of digestive system CFCS39 Prechtl’s Assessment of
Activities and participation: mobility General Movements
communication (General Movement
Assessments)40,41
TIMP42

Speech,
language, and Cognitive and Executive Social–
NINDS domain ICF component/chapters communication emotional status functioning Memory emotional

Neurocognitive, Body functions: mental functions PPVT-430 ASQ-343 BRIEF-P44 CVLT-C45 BITSEA46
social, and Neuromusculoskeletal and movement- BRIEF-247 SCQ48,49
emotional related functions Bayley III, BSID50,51 BRIEF-A52
assessments Voice and speech functions MSEL53
Activities and participation: general tasks WAIS-IV54 Conners CPT 355
and demands D-KEFS56
Learning and applying knowledge WISC-V57
Communication

NINDS domain ICF component/chapters QoLa Participation

58
Engagement and Activities and participation: general CPCHILD LIFE-H59
quality of life tasks and demands CP QOL60 Child Engagement
assessments Mobility in Daily Life61
Self-care Interpersonal interactions PedsQL62, 63 PEM-CY64–66
and relationships PedsQL-CP67 YC-PEM68,69
Learning and Applying knowledge
Environmental factors: Attitudes
and supports
a
Not included in the ICF. EDACS, Eating and Drinking Ability Classification System; AIMS, Alberta Infant Motor Scale; BADS, Barry-Albright
Dystonia Scale; PPVT-4, Peabody Picture Vocabulary Test, Fourth Edition; GMFCS-ER, Gross Motor Function Classification System –
Expanded and Revised; COPM, Canadian Occupational Performance Measure; MACS, Manual Ability Classification System; GMFM, Gross
Motor Function Measure; CFCS, Communication Function Classification System; TIMP, Test of Infant Motor Performance; ASQ-3, Ages &
Stages Questionnaire 3; BRIEF-P, Brief Rating Inventory of Executive Function – Preschool version; CVLT-C, California Verbal Learning Test
for Children; BITSEA, Brief Infant Toddler Social Emotional Assessment; BRIEF-2, Behavior Rating Inventory of Executive Function – Sec-
ond edition; SCQ, Social Communication Questionnaire; BSID, Bayley Scale of Infant Development; BRIEF-A, Behavior Rating Inventory of
Executive Function – Adult version; MSEL, Mullen Scales of Early Learning; WAIS-IV, Wechsler Adult Intelligence Scale – Fourth edition;
Conners CPT 3, Conners Continuous Performance Test, Third Edition; D-KEFS, Delis–Kaplan Executive Function System; WISC-V, Wechsler
Intelligence Scale for Children – Fifth Edition; QoL, quality of life; CPCHILD, Caregiver Priorities and Child Health Index of Life with Disabili-
ties; LIFE-H, Assessment of Life Habits; CP QOL, Cerebral Palsy Quality of Life; PedsQL, Pediatric Quality of Life Inventory; PEM-CY, Partici-
pation and Environment Measure for Children and Youth; PedsQL-CP, Pediatric Quality of Life Inventory Cerebral Palsy Module; YC-PEM,
Young Children’s Participation and Environment Measure.

Engagement and quality of life assessments/data collection of any relevant findings. Comprehensive evalua-
Identifying one engagement or quality of life tool was diffi- tion is important for analysis and meta-analysis in future
cult. Many of the tools reviewed were not specific for chil- studies to better understand the neural structure function
dren with CP. For individuals with CP who are non-verbal relations vital to improving function in CP. Also, it is chal-
or who have cognitive impairments as a component of lenging to allow for documentation of both acute findings
their CP, assessment of quality of life is a difficult con- (recent injury) and chronic findings that would be relevant
struct to measure, and validated proxy and self-report to development of CP. This working group developed a
options are limited. useful CRF that can be utilized for documenting both
acute and chronic findings among the different types of
Neuroimaging diagnostics CP.
In contrast to CP, CRFs developed for other neurological
disorders and populations are more homogeneous and the Neuromotor skills and functional assessments
types of findings are narrower. CP has many aetiologies CP is a heterogeneous disorder in aetiology and pheno-
and there can be a wide range of findings; therefore, it is type. Therefore, identifying tools that (1) spanned the CP
important that a neuroimaging CRF allows for data type and functional ability; (2) were psychometrically

982 Developmental Medicine & Child Neurology 2018, 60: 976–986

sound; and (3) were specific to individuals with CP was international standards, specifically the ICF Core Sets for
challenging. Furthermore, while therapies are key interven- children and young people with CP,6 which highlight what
tions in CP, there has not been a standardized method for areas of functioning, disability, and health should be mea-
documenting and reporting on therapy interventions. sured in this population, we found commonalities and dif-
Therefore, this working group created CRFs for therapies ferences. The landmark characteristics of CP are captured
(physical, occupational, speech/language) to facilitate sys- by both standards, including movement and posture disor-
tematic reporting on specific therapeutic interventions, ders, communication, cognition, and musculoskeletal chal-
duration, and intensity of interventions. lenges, among others. However, some relevant areas are
under-represented by the current version of the CDEs, for
Comparison to other international CP standards example sensation of pain, sensory functions, sleep func-
CP registers throughout the world were used as a starting tions, support and attitude of peers, role of products and
point.21 Where there was full agreement amongst registers, technology – including assistive devices for daily living,
those items were included and measured in the same way. education, and recreation. When we compared the how to
Medical, surgical, and family history forms are consistent measure relevant areas in CP – proposed by the NINDS/
with the type of information collected by CP registries AACPDM set of tools – to the recently developed ICF-
around the world. based toolbox of measures for CP,7 we found that the
In terms of CDEs related to neuroimaging, conventional NINDS/AACPDM includes a much more comprehensive
MRI of the brain is the imaging modality of choice for set of tools, including single-item and multiple-item mea-
clinical diagnostic evaluation of children with CP and is sures. However, the NINDS/AACPDM recommended
consistent with the practice parameter ‘Diagnostic Assess- tools highly represent the ICF component body functions,
ment of the Child with CP’ of the American Academy of whereas the ICF-based toolbox of measures focuses mainly
Neurology,70 as well as the practice parameters of other on the ICF component activities and participation.
international groups. Moreover, the Surveillance of Cere-
bral Palsy in Europe group published a classification sys- Considerations for future CDE revisions
tem for standardizing the abnormal findings of brain MRI There is currently no definitive ‘classification of aetiology’
in children with CP.71 Their approach was different from of CP owing to the heterogeneous nature of the disorder
the NINDS CDE project in that they tried to define cate- and because there is very rarely one definitive cause, rather
gories of patterns of involvement for research studies. The a causal pathway that contains any number of risk factors.
NINDS CDE for CP approach was to promote coding If classifications for aetiology are developed, they should
and documentation of all abnormal findings in a consistent be incorporated into the CP CDEs. In addition, further
manner but with less focus on interpretation and categoriz- expansion of commonly used laboratory tests should be
ing patterns of involvement. The tool developed by considered in future revisions.
Surveillance of Cerebral Palsy in Europe would be useful Coding systems for evaluating different structures in the
for research and complementary to the CRF developed for brain by neuroimaging will gradually become more refined.
this project, and both could be used together. The various aetiologies of CP will be increasingly corre-
The NINDS CDEs for CP are comparable to CDEs for lated with specific neuroimaging findings. This research
other medical conditions, including Friedrich’s ataxia, will need to be incorporated into the CDE framework for
stroke, epilepsy, Duchenne muscular dystrophy/Becker neuroimaging in CP. CDEs for newer neuroimaging
muscular dystrophy, spinal muscular atrophy, and trau- modalities (e.g. diffusion tensor imaging and functional
matic brain injury. However, terminology used for these MRI) relevant to CP will need to be developed.
other diagnoses is not consistent with currently accepted Owing to the wide spectrum of abilities and limitations
terminology based on published consensus statements of of individuals with CP, the development of tools specific
experts.72 For example, while hemiplegia, diplegia, and to anatomical distribution, unilateral or bilateral, or the
quadriplegia are frequently used in other disorders, there functional level of individuals with CP would be beneficial.
can be imprecision with this classification in CP.73 Thus, While there are well-known tools for spasticity and dys-
we sought to also encourage the use of ‘unilateral’ and ‘bi- tonia assessments in CP, there are as yet no tools to clas-
lateral’ CP, as recommended by expert consensus.72 Tools sify hypotonia. Furthermore, tools to assess a child’s
were reviewed from systematic and narrative reviews in the engagement in therapy and measure the impact that partic-
CP literature, when those existed.15,74–82 Occupational ipation has on the outcomes of therapy are needed. Finally,
therapy, physical therapy, and speech and language pathol- given the breadth of the CDEs and instruments, the work-
ogy recommendations are based on published data ele- ing groups did not explore CDEs or tools to assess surgical
ments (modified to reflect CP practice) and are aligned interventions and surgical outcomes in CP. Creation of an
with discipline-specific standards of practice and current interdisciplinary surgical working group for the next ver-
research.22,24,83 sion of the CDEs for CP is recommended.
When we compared the what and the how to measure From an ICF perspective, future revisions should include
common information proposed by the NINDS/AACPDM all relevant areas of functioning, disability, and health
CDEs for CP (Version 1.0) to other newly developed highlighted in the ICF Core Sets for children and young

Review 983
people with CP, incorporating measures addressing func- interact dynamically across ages and stages of develop-
tional impact of chronic pain and sleep disturbances in ment of children and young people with CP. Ongoing
day-to-day functioning. Further expansion of self-report international collaboration will help ensure that the
measures capturing activities and participation constructs CDEs are updated and reviewed as additional evidence is
should be considered in future revisions. obtained.
Finally, although CP is a life-long disability, many mea-
sures for CP are focused on paediatrics. Measures for chil- A CK N O W L E D G E M E N T S
dren and young people with CP have been used to The views expressed here are those of the authors and do not rep-
evaluate adults with CP, but the psychometric properties resent those of the National Institutes of Health, the National
have not been studied. In addition, measures developed for Institute of Neurological Disorders and Stroke (NINDS), or the
adults with other types of disability may be useful but have US Government. Logistics support for this project was provided,
not been validated for adults with CP. The development of in part, through National Institutes of Health contract
a working group to examine this need is recommended. HHSN271201200034C. The development of the NINDS CP
CDEs was made possible thanks to the great investment of time
Future directions and effort of working group members and the members of the
Of note, Version 1.0 of NINDS CDEs for CP is the start- NINDS Common Data Elements Project team participating from
ing point; CDEs are dynamic and will evolve over time. It 2015 to present. Dr. Ver onica Schiariti was the recipient of a
is expected that future revisions will incorporate the valu- NeuroDevNet & Child and Family Research Institute Postdoc-
able perspective of parents of children with CP, as well as toral Fellowship Award (2014-2016), supporting this study
adults with CP. Furthermore, NINDS has created an over-
sight committee to continuously review the NINDS CDEs SUPPORTING INFORMATION
for CP. The following additional material may be found online:
Appendix S1: National Institute of Neurological Disorders
CONCLUSION and Stroke/American Academy of Cerebral Palsy and Develop-
The global adoption of standardized data collection, mental Medicine Common Data Element Project working
whenever feasible, will increase opportunities for con- groups.
ducting secondary data analyses and meta-analyses that Appendix S2: Cerebral palsy start-up resource document.
could advance knowledge about how brain behaviour, Appendix S3: Cerebral palsy motor developmental history case
functional abilities, and genetics–biology–environment report form.

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986 Developmental Medicine & Child Neurology 2018, 60: 976–986

 DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY REVIEW

RESUMEN
UN LENGUAJE DE DATOS COMUNES PARA ESTUDIOS DE INVESTIGACION
CL
INICA PARA PARALISIS

CEREBRAL:
RECOMENDACIONES DEL NATIONAL INSTITUTE OF NEUROLOGICAL DISORDERS AND STROKE Y LA AMERICAN ACADEMY FOR

1.0
CEREBRAL PALSY AND DEVELOPMENTAL MEDICINE - ELEMENTOS DE DATOS COMUNES VERSION
Para aumentar la eficiencia y la efectividad de los estudios de investigacio
n cl
ınica, se desarrollaron elementos de datos comunes
(CDEs sigla en ingle
s) espec
ıficos para la para
lisis cerebral (PC), mediante una colaboracio
n entre la Academia Estadounidense de
Para
lisis Cerebral y Medicina del Desarrollo (AACPDM) y el Instituto Nacional de Trastornos Neurolo
gicos y Accidentes
Cerebrovasculares (NINDS). Expertos internacionales revisaron los CDEs existentes y las herramientas de medicio
n del NINDS
utilizadas en estudios de nin ~ os y jo
venes con PC. Los CDEs se compilaron, se sometieron a una revisio
n interna y se publicaron
en l
ınea para recibir comentarios pu
blicos externos en septiembre de 2016. Guiados por el marco teo
rico de la Clasificacio
n
Internacional del Funcionamiento, de la Discapacidad y de la Salud, los CDE se categorizaron en seis dominios: (1) caracter
ısticas
de los participantes; (2) salud, crecimiento y gene
tica; (3) neuroimagen; (4) habilidades neuromotoras y evaluaciones funcionales;
(5) evaluaciones neurocognitivas, sociales y emocionales; y (6) compromiso y calidad de vida. La versio
n 1.0 de los CDE del
NINDS / AACPDM para PC esta
pu
blicamente disponible en los sitios web del NINDS CDE y AACPDM. El uso global de CDE para
PC estandarizara
la recopilacio
n de datos, mejorara
la calidad de los datos, y facilitara
las comparaciones entre los estudios. La
colaboracio
n continua con colegas internacionales, la industria, y las personas con PC y sus familias proporcionara

retroalimentacio
n y actualizaciones significativas a medida que se obtenga evidencia adicional. Por el momento, la aplicacio
n de
estos CDEs se recomienda para estudios cl
ınicos y de investigacio
n - financiados por el NINDS - en individuos con PC.

RESUMO
UMA LINGUAGEM DE DADOS COMUNS PARA ESTUDOS DE PESQUISA CL
INICA: VERSAO
~ 1.0 DAS RECOMENDAC ~
ß OES DOS
ELEMENTOS DE DADOS COMUNS DO NATIONAL INSTITUTE OF NEUROLOGICAL DISORDERS AND STROKE E DA AMERICAN
ACADEMY FOR CEREBRAL PALSY AND DEVELOPMENTAL MEDICINE
Para aumentar a eficie ^ncia e efetividade dos estudos de pesquisas cl
ınicas, Elementos de dados comuns (EDC) espec
ıficos para
paralisia cerebral (PC) foram desenvolvidos por meio de parceria com a Academia Americana de Paralisia Cerebral e Medicina do
Desenvolvimento (AACPDM) e do Instituto Nacional de Desordens Neurolo
gicas e Derrame (NINDS). Especialistas internacionais
revisaram os EDCs do NINDS e os instrumentos usados em estudos de criancßas e jovens com PC. EDCs foram compilados,
submetidos a revisa ~o interna, e postados online para comenta
rio do pu
blico externo em setembro de 2016. Guiados pela
estrutura da Classificacßa~ o internacional de funcionalidade, incapacidade e sau
de, os EDC foram categorizados em seis dom
ınios:
1) caracter
ısticas dos participantes; 2) sau
de, crescimento e gene
tica; 3) neuroimagem; 4) habilidades neuromotoras e avaliacßo ~ es
funcionais; 5) avaliacßo~ es neurocognitivas, sociais e emocionais; e 6) engajamento e qualidade de vida. A versa ~o 1.0 dos EDC
NINDS/AACPDM para PC esta
dispon
ıvel para o pu
blico nos websites do NINDS EDC e da AACPDM. O uso global de EDCs para

padronizar a coleta de dados, melhorar a qualidade dos dados, e facilitar comparacßo
PC ira ~ es entre estudos. Colaboracßo~ es em
andamento com colegas internacionais, indu
stria e pessoas com PC e suas fam
ılias ira ~o fornecer informacßo~ es significativas e
~ es conforme evide
atualizacßo ^ncias adicionais forem obtidas. Estes EDCs sa ~o recomentados para pesquisas em PC financiadas pelo
NINDS.