Physical & Occupational Therapy In Pediatrics

ISSN: 0194-2638 (Print) 1541-3144 (Online) Journal homepage: http://www.tandfonline.com/loi/ipop20

Measures of Self-Care Independence for Children

with Osteochondrodysplasia: A Clinimetric Review
Penelope Ireland & Leanne M. Johnston
To cite this article: Penelope Ireland & Leanne M. Johnston (2012) Measures of Self-Care
Independence for Children with Osteochondrodysplasia: A Clinimetric Review, Physical &
Occupational Therapy In Pediatrics, 32:1, 80-96
To link to this article: http://dx.doi.org/10.3109/01942638.2011.593619

Published online: 06 Jul 2011.

Submit your article to this journal

Article views: 221

View related articles

Full Terms & Conditions of access and use can be found at

http://www.tandfonline.com/action/journalInformation?journalCode=ipop20
Download by: [dessy rahma]

Date: 13 November 2015, At: 01:56

Physical & Occupational Therapy in Pediatrics, 32(1):8096, 2012

C 2012 by Informa Healthcare USA, Inc.
Available online at http://informahealthcare.com/potp
DOI: 10.3109/01942638.2011.593619

Measures of Self-Care Independence for Children

with Osteochondrodysplasia: A Clinimetric Review
Penelope Ireland1,2 & Leanne M. Johnston2
1

Downloaded by [dessy rahma] at 01:56 13 November 2015

Queensland Paediatric Rehabilitation Service, Royal Childrens Hospital, Brisbane,

Australia, 2 Division of Physiotherapy, School of Health and Rehabilitation Sciences,
The University of Queensland, Brisbane, Australia

ABSTRACT. This systematic review evaluates the validity, reliability, and clinical utility
of outcome measures used to assess self-care skills among children with congenital musculoskeletal conditions and assesses the applicability of these measures for children with
osteochondrodysplasia aged 012 years. Electronic databases were searched to identify
self-care assessments that addressed the self-care domain as defined by of the International Classification of Function Disability and included children with osteochondrodysplasia. Ten measures were identified and three met the inclusion criteria: the Functional
Independence Measure for Children (WeeFIM), the Activities Scale for Kids (ASK),
and the Pediatric Evaluation of Disability Inventory (PEDI). Although psychometric
data specific to children with osteochondrodysplasia are limited, adequate to excellent
reliability and evidence of validity were reported for all three instruments for children
with physical disabilities. Further evaluation of psychometric properties of self-care instruments specifically for children with osteochondrodysplasia would be beneficial to
help identify instruments that will assist with improved assessment and management.
KEYWORDS. Activities of daily living, functional performance, measurement, osteochondrodysplasias, short stature

Osteochondrodysplasia incorporates over 350 conditions of abnormal growth and

remodeling of cartilage and bone with a collective incidence of approximately 24.7
per 10,000 live births (Baitner, Maurer, Gruen, & Di Cesare, 2000). This group of
disorders can be subclassified as osteodysplasias, disorders of abnormal bone density with proportionate short stature, or chondrodysplasias which result in deformations and malformations with short trunk or short limb short stature (Tuysuz, 2004).
It is widely acknowledged that these complex abnormalities of musculoskeletal development are associated with limitations in physical activity and functioning in
everyday tasks (Tuysuz, 2004). Disproportionate shortening of the trunk and/or
limbs and restricted positioning and handling in the first year of life may contribute
to delayed motor development and independence in self-care. Although delays in
Address correspondence to: Penelope Ireland, Queensland Paediatric Rehabilitation Service Level 2, Coles
Medical Building, Royal Childrens Hospital, HERSTON 4029 (E-mail: Penny ireland@health.qld.gov.au).
(Received 29 July 2010; accepted 31 May 2011.)

80

Downloaded by [dessy rahma] at 01:56 13 November 2015

Self-Care Measures in Osteochondrodysplasia

81

self-care independence are observed clinically, there is little literature regarding

activity limitations in self-care and guidelines for physical and occupational therapy assessment and management. A lack of information regarding assessment and
intervention for children with achondroplasia, the most common form of osteochondrodysplasia, may have contributed to omission of physical and occupational
therapy in the international consensus guidelines for management of the condition (Horton, Hall, & Hecht, 2007). Knowledge of reliable and valid assessments is
necessary in order that well-timed and purposeful intervention can be provided to
improve independence in self-care (Damignani, Young, Cole, Anthony, & Badley,
2004).
The ability of currently available assessments to measure self-care limitations
arising from the characteristic musculoskeletal impairments associated with osteochondrodysplasia has not been determined. Although systematic reviews have
addressed activity and participation measures for children with developmental disabilities (Harvey, Robin, Morris, Graham, & Baker, 2008) including cerebral palsy
(Sakzewski, Boyd, & Ziviani, 2007), the results may not be applicable to children
with osteochondrodysplasia. According to COsensus-based Standards for the selection of health status Measurement INstruments (COSMIN) recommendations
(Mokkink et al., 2010), three essential clinimetric properties are validity (the degree
to which each assessment tool measures the construct of self-care), reliability (the
level of reproducibility of the assessment results), and clinical utility (the feasibility
and usefulness of the tool for the osteochondrodysplasia population).
For the purpose of this review, the construct of self-care includes functional
tasks that are expected to develop during childhood that have been described by
the International Classification of Functioning, Disability and Health (ICF, World
Health Organization, 2007). In the ICF, the combined Activity and Participation
component lists the domain of d5 Self-Care, which includes categories of relevant self-care tasks for children and adolescents: including washing oneself (d510);
caring for body parts (d520); toileting (d530); dressing (d540); eating (d550); drinking (d560); looking after ones health (d570); and looking after ones safety (d571;
World Health Organization, 2007). Competence with tasks in these categories can
be evaluated with respect to (a) a childs capacity, i.e., what a child can do in a standardized environment, (b) a childs capability, i.e., what a child can do in his/her
natural environment, and/or (c) a childs performance, i.e., what a child actually
does do in his/her natural environment/s. Assessment of capacity in a standard
environment addresses activity, while assessment of capability and performance
in the childs natural environments addresses participation (Holsbeeke, Ketelaar,
Schoemaker, & Gorter, 2009). For each component, the degree of physical assistance needed to perform the task and the type and contribution of assistive devices
are important qualifying factors. The aims of this systematic review are the following:
1. to identify measures appropriate for assessing self-care capacity, capability, or
performance in children with osteochondrodysplasia less than 12 years of age;
2. to evaluate the reliability, validity, and utility of the measures; and
3. to provide recommendations for practice and research.

82

Ireland and Johnston

METHOD

Downloaded by [dessy rahma] at 01:56 13 November 2015

Search Strategy
Articles were identified through systematic searches of five computerized
databases, including Medline (1966July 2010), CINAHL (1982July 2010), PsychINFO (1806July 2010), PubMed (1951July 2010), and the Cochrane Library (July
2010). The search strategy used the MeSH terms and text words for osteochondrodysplasia or short stature combined with self-care, or activities of daily
living or functional outcome with searches limited to articles involving children
from birth to 12 years and published up to November 2010. To address the possibility that conditions included under osteochondrodysplasia or short stature varied
across databases, all conditions listed as MeSH terms under osteochondrodysplasia
in each database were also searched separately as keywords (Achondroplasia;
Acquired Hypertosis Syndrome; Camurati-Engelmann Syndrome; Chrondrodysplasia Punctata; Rhizomelic Cleidocranial dysplasia; Ellis-Can Crevold Syndrome;
Erchondromatosis; Fibrous Dysplasia of Bone; Hyperostosis, Cortical, Congenital;
Hyperostosis Frontalis Interno; Langer-Giedian Syndrome; Osteochondroma;
Osteogenesis Imperfecta; Osteosclerosis; Short-Rib Polydactyly Syndrome;
Thanatophonic Dysplasia). Subsequent searches were performed using the names
of identified assessments and their authors and manual checking of the reference
lists. Citation tracking of included articles was performed up to November 2010.
Inclusion/Exclusion Criteria
Articles included in the systematic review were those that (a) were written in English; (b) included children aged 012 years with a form of osteochondrodysplasia
or related congenital musculoskeletal condition; (c) measured at least one of the
ICF d5 self-care domain categories of washing oneself, caring for body parts, toileting, dressing, eating or drinking, or at least one independent self-care subscale;
and (d) included data on one or more clinimetric properties.
Articles were excluded if they (a) focused on populations of children with conditions not characterized by a primary musculoskeletal impairment (e.g., Down
syndrome or cerebral palsy) or intellectual impairments; (b) reported on self-care
domains of looking after ones health (d570) or looking after ones safety
(d571) as these were deemed more complex skills; and (c) reported individualized
goal setting tools (e.g., Goal Attainment Scaling).
Titles and abstracts of the articles identified by the literature searches were
screened independently by the two authors (Penelope Ireland and Leanne
M. Johnston) to identify studies for inclusion. Both authors were physiotherapists
with over 15 years experience regarding outcome assessment and research with children with developmental and neurological disorders. Full-text articles were sourced
where the title and abstract did not provide enough information. Articles selected
for review were independently rated as meeting criteria by the two therapists. There
were no disagreements on article selection.
Data Extraction and Quality Assessment
The checklist for COSMIN developed by Mokkink et al. (2010) was used to
evaluate the methodological quality of those studies reporting on measurement

Downloaded by [dessy rahma] at 01:56 13 November 2015

Self-Care Measures in Osteochondrodysplasia

83

properties for the identified assessments. The criteria for assessing reliability, content, structural, criterion, and cross-cultural validity and responsiveness are outlined in the COSMIN checklist manual. Using the checklist, each study was evaluated for general design and statistical methods using yes/no responses. Specific
instructions and explanations are provided in the COSMIN manual to assist with
scoring and interpretation. Further information is available from the checklist manual (Mokkink et al., 2009) and the COSMIN Web site (http://www.cosmin.nl). Both
the authors (Penelope Ireland and Leanne M. Johnston) completed these checklists
independently. Conflicting viewpoints were discussed until consensus was reached.
A data extraction sheet was modified from the CanCHILD Outcomes Measures
Rating Form (Law, 2004). This was used to record and compare the clinimetric
properties of each assessment, looking specifically at clinical utility, reliability, and
validity. Clinical utility was evaluated through review of test manuals, administration guidelines for scoring and interpretation, and formal training requirements
(Law, 2004). Reliability is the determination that the procedure measures findings
in a manner that is both reproducible and consistent, i.e., the repeatability of the
findings (Dawson & Trapp, 2004; Guyatt, Walter, & Norman, 1987; Law, 2004).
Intratester, intertester, equivalence, and testretest reliability were reviewed and
recorded. Results of intraclass correlation coefficient (ICC) analyses greater than
0.80 were considered excellent, 0.600.79 as adequate, and less than 0.60 as poor
(Law, 2004). Validity refers to how well an instrument measures what it was designed to measure (Dawson & Trapp, 2004; Guyatt et al., 1987). Content validity,
criterion validity, and responsiveness to change were evaluated.
RESULTS
Search Strategy Yield
Electronic searches yielded 120 articles. Review of these articles identified 21 articles that reported on 10 assessments of self-care. Three assessments, and the articles they were cited in, were excluded because they did not include any measures
of self-care: (a) the Gillette Functional Assessment Questionnaire Walking Scale
(Furlong, Barr, Feeny, & Yandow, 2005), (b) The Health Utilities Index (Furlong
et al., 2005), and (c) the Pediatric Inventory of Quality of Life (Furlong et al.,
2005). A further four assessments, and the articles they were cited in, were excluded as they contained only individual self-care items that were not independently administrable as a self-care subscale, (d) the Child Health Questionnaire
Parent Form 50 (Collins et al., 2005; Kelly, Brillante, Kushner, Gehron Robey,
& Collins, 2005), (5) the Health-Related Quality of Life (Apajasalo, Sintonen,
Rautonen, & Kaitila, 1998), (6) the Lifestyle Assessment Questionnaire (Jessen,
Colver, Mackie, & Jarvis, 2003), and (7) the Pediatric Outcomes Data Collection
Instrument (Huang, Ambrose, Sullivan, & Haynes, 2006; Leet et al., 2006).
The three assessments that met the selection criteria are the Functional Independence Measure for Children (WeeFIM; Uniform Data System for Medical Rehabilitation, 2006), the Pediatric Evaluation of Disability Inventory (PEDI; Haley,
Costner, Ludlow, Haltiwanger, & Andrellos, 1992), and the Activities Scale for Kids
(ASK; Young, Yoshida, Williams, Bombardier, & Wright, 1995). These three measures are summarized in Table 1.

84
WeeFIM (Braun et al., 1991)

ASK (Young et al., 1995)

Children with physical or general

Children with physical limitations due to
developmental limitations or restrictions
musculoskeletal disorders
6 months to 7.5 years or >7.5 years if delayed 6 months to 7 years or >7 years if
515 years
functional ability
delayed functional ability.
Adolescents and adults may use the
Functional Independence Measure
Self-care, mobility, and social function
Self-care, mobility, and cognition
Self-care, locomotion, standing skills, transfers,
play, and other skills
197 functional skills over 3 domains
18 caregiver assistance items
ASK capability version = 30 items
20 caregiver assistance skills
ASK performance version = 30 items
Capability performance
Performance
Capability performance
Washing oneself (d510: 1 item), caring for Washing oneself (d510: 1 item), caring for body
Washing oneself (d510: 6 items), caring for
parts (d520: 1 item), toileting (d530: 1 item),
body parts (d520: 1 item), toileting
body parts (d520: 20 items), toileting (d530:
and dressing (d540: 4 items)
(d530: 3 items), dressing (d540: 2
18 items), dressing (d540: 22 items), eating
items), eating (d550), and drinking
(d550: 10 items), and drinking (d560: 5 items)
(d560: 1 item)
Manual$122.25
ManualApply though
Manualnot required
www.udsmr.org/cost on application
Score sheets$42.75 for 25 sheets
Score sheetsable to develop own
Score sheetscan be downloaded via the ASK
and/or photocopy score sheets
Web. Charges may apply depending on
required usage
4560 min
1015 min
59 min
Direct observation by examiner
Direct observation by examiner
Child report via booklet questionnaire. Parents
are able to read questions to child or provide
a proxy report if child is unable
Structured interview with primary caregiver
Face-to-face or telephone interview of a
Able to be administered by postal contact
primary caregiver
Discipline free
Discipline free
Discipline free
Training recommended to ensure appropriate
Training recommended to ensure
No specific training required to administer or
administration and scoring
appropriate administration and scoring
score

Children with physical cognitive limitations

PEDI (Haley et al., 1992)

Note: PEDI, Pediatric Evaluation of Disability Inventory; WeeFIM, Functional Independence Measures for Children; ASK, Activities Scales for Kids; ICF, International Classification of
Functioning, Disability and Health.

Examiner training

Administration time
Administration methods

Manual

ICF construct
ICF domain categories
and number of items
for self-care

Number of items

Test domains

Age range

Target population

Instrument

TABLE 1. Description of the Three Measures Included in the Systematic Review

Downloaded by [dessy rahma] at 01:56 13 November 2015

Downloaded by [dessy rahma] at 01:56 13 November 2015

Self-Care Measures in Osteochondrodysplasia

85

A total of eleven articles were identified from the literature search that used
the three assessments in children with osteochondrodysplasia (Amako et al., 2004;
Damignani et al., 2004; Engelbert et al., 2004; Engelbert et al., 1997a; Engelbert
et al., 1997b; Land, Rauch, Montpetit, Ruck-Gibbis, & Glorieux, 2006; Leet et al.,
2006; Sakkers et al., 2004; Seikaly et al., 2005; Sperle, Ottenbacher, Braun, Lane
& Nockajski, 1997; Ziviani et al., 2001). The titles of the three assessments were
used as key words to search for additional studies involving children with osteochondrodysplasia, primary musculoskeletal impairments, or typically developing
controls. The searches yielded a total of 16 articles and 2 assessment manuals that
provided detailed information on one or more clinimetric properties. A description
of each article and COSMIN quality review of each study are reported in Table 2.
Three of the studies specifically reported on children with osteochondrodysplasia (Engelbert et al., 1997a; Pencharz et al., 2001; Wassenberg-Severijnen et al.,
2003). A further nine studies (Chen et al., 2005; Haley et al., 1992; Nichols &
Case-Smith, 1996; Ottenbacher et al., 1996, 1997, 1999, 2000; Sperle et al., 1997;
Ziviani et al., 2001) made reference to inclusion of children with developmental
disabilities including genetic or chromosomal abnormalities but were not specific
about condition type. Three studies included children with primary musculoskeletal
impairments (Feldman et al., 1990; Young et al., 1995, 2000). The remaining three
studies were performed using a typically developing reference group (Berg et al.,
2004; Jongjit et al., 2006; Wong et al., 2002). Children were recruited through early
intervention, school-, hospital-, or rehabilitation-based programs.
Reliability
Scores on the COSMIN checklist indicated that 6 of the 12 studies addressed all
components of reliability (Table 2). Eleven studies included children with physical disabilities, with two studies specifically including children with osteochondrodysplasia (Engelbert et al., 1997b; Wassenberg-Severijnen et al., 2003; Table
2). Adequate to excellent interrater and intrarater reliability (ICC = 0.61.0) for
children with congenital or genetic abnormalities were reported for the WeeFIM
(Ottenbacher et al., 1996, 1997; Sperle et al., 1997; Ziviani et al., 2001) and the PEDI
(Engelbert et al., 1997b; Nichols & Case-Smith, 1996; Wassenberg-Severijnen et al.,
2003; Ziviani et al., 2001; Table 3). The number of children with osteochondrodysplasia, however, was not always specified. Engelbert et al. (1997b) was the one study
involving only children with osteochondrodysplasia; however, ICC for reliability
were not reported. Young et al. (1995, 2000) reported excellent reliability (ICC >
0.8) for the ASK when completed by children with a variety of musculoskeletal conditions (Table 3). Neither study indicated whether children with osteochondrodysplasia were among the participants.
Validity
Criterion Validity
Scores on the COSMIN checklist indicated that four of the six studies addressed
all components of validity (Table 2). Moderate to high concurrent validity was reported for all three measures (Feldman et al., 1990; Nichols & Case-Smith, 1996;
Ottenbacher et al., 1999; Pencharz et al., 2001; Young et al., 1995; Ziviani et al.,

86

Sample type, age, and source

Ottenbacher et al.
(1999)

Ottenbacher et al.
(1997)

Ottenbacher et al.
(1996)

Jongjit et al. (2006)

Sample size and representation

Children with disabilities Age,

1187 months
Sourceearly intervention
service, school-based
recruitment

Age, 1276 months

Sourceearly intervention
services, school-based
recruitment
Children with disabilities Age,
1187 months
Sourceearly intervention
services, school-based
recruitment

Proportional

Not stated

Convenience

Sampling

Cohort = typical: 37 (55%); neuromotor: 19

(28%); nonspecific: 11 (17%);
osteochondro-dysplasia (not stated or
excluded)
Proportional
N = 205
Cohort = cerebral palsy, Down syndrome,
spina bifida, developmental disabilities,
mental retardation, congenital impairments
(% not specified); osteochondrodysplasia
(not stated or excluded)
Proportional
N = 20
Cohort = cerebral palsy, prematurity,
Down syndrome, spina bifida, genetic
impairments (% not specified);
osteochondrodysplasia (not stated or
excluded)

N = 814
Cohort = ABI: 448 (55%), cerebral palsy: 90
(11%), multiple trauma: 57 (7%), other
(including congenital musculoskeletal):
219 (27%); osteochondrodysplasia (not
stated or excluded)
Typically developing Thai children N = 569
Age, 6100 months
Cohort = typical: 569 (100%);
Sourcecommunity volunteers
osteochondrodysplasia (no)
Typically developing children and N = 67
children with disabilities

WeeFIM
Chen, Bode, Granger, Inpatient rehabilitation patients
& Heinemann (2005) Age, not stated
Sourcepatient database

Study

TABLE 2. Studies of the Clinimetric Properties of the WeeFIM, PEDI, and ASK

Downloaded by [dessy rahma] at 01:56 13 November 2015

Criterion validity

Missing B5
administration
independence
Missing B9 test
conditions

All items addressed

All items addressed

Missing B9 test
conditions

All items addressed

All items addressed

COSMIN checklist

D
E

Interrater reliability

Interrater and
intrarater
reliability
Testretest and
equivalence
reliability

Content validity
Structural validity

Psychometrics

87

Children with disabilities

Age, 1971 months
Source3 education facilities

Rehabilitation program patients

Age, 1.69.5 years
Sourceclient database

Sperle, Ottenbacher,
Braun, Lane, &
Nochajski (1997)

Ziviani et al. (2001)

Engelbert et al. (1997)

PEDI
Berg, Jahnsen,
Froslie, & Hussain
(2004)

Typically developing Norwegian

children
Age, 15 years
Sourceregistry of inhabitants
Children with osteogenesis
imperfecta
Age, 018 years
Sourcehospital clinic

Wong, Wong, Chan, & Typically developing Chinese

Wong (2002)
children
Age, 6 months to 7 years
Sourcecommunity volunteers

Children with disabilities

Age, 1187 months
Source3 rehabilitation facilities

Ottenbacher et al.
(2000)

Cohort = osteogenesis imperfecta (a

subcategory of osteochondro-dysplasia):
61 (100%)

Cohort = typical: 30 (100%);

osteochondrodysplasia (no)
N = 61

N = 30

Cohort = typical: 445 (100%);

osteochondrodysplasia (no)

N = 174
Cohort = cerebral palsy: 41 (24%), Down
syndrome: 12 (7%), communication
disorder: 72 (41%), developmental
disorder: 32 (18%), congenital impairment:
9 (5%); osteochondrodysplasia (not stated
or excluded)
N = 30
Cohort = cerebral palsy: 12 (40%), Down
syndrome: 6 (20%), developmental delay:
7 (23%), and congenital abnormalities: 5
(17%); osteochondrodysplasia (not stated
or excluded)
N=4
Cohort = ABI: 16 (39%); spina bifida: 19
(46%); genetic/chromosomal
abnormalities : 6 (15%); osteochondrodysplasia (not stated or excluded)
N = 445
Interrater reliability

Criterion validity
Interrater reliability

Equivalence
reliability

Responsiveness

Convenience

Interrater and
intrarater
reliability

Proportional
Interrater and
and conveintrarater
nience
reliability

Proportional

Convenience

Convenience

Proportional

Downloaded by [dessy rahma] at 01:56 13 November 2015

Missing B5
administration
independence and
B11 use of ICC

All items addressed

Missing B5
administration
independence, B8
time intervals, and
B9 test conditions

All items addressed

All items addressed

All items addressed

All items addressed

(Continued on next page)

H
B

88

Typically developing children

and children with disabilities
Age, 28 years
Sourcehospital-based
clinics
Children with disabilities
Age, not stated
Sourceclinical sites (not
specific)

Feldman, Haley, &

Coryell (1990)

Ziviani et al. (2001)

Children in rehabilitation
programs
Age, 1.69.5 years
Sourceclient database

Typically developing Dutch

Wassenbergchildren and children with
Severijnen, Custers,
disabilities
Hox, Vermeer, &
Age, 788 months
Helders (2003)
Sourcecommunity
volunteers and childrens
hospital

Nichols & Case-Smith Children with disabilities

(1996)
Age, 1382 months
Sourceclinical sites (not
specific)

Haley et al. (1992)

Sample type, age, and source

Study
Convenience

Sampling

Convenience

Cohort = typical: 63 (54%),

Neurometabolic: 29 (25%), spina
bifida: 7 (6%), osteogenesis
imperfecta (a subcategory of
osteochondrodysplasia): 11 (9%),
infantile encephalopathy: 6 (6%)
N = 41
Cohort = ABI: 16 (39%); spina bifida: 19
(46%); genetic/chromosomal
abnormalities: 6 (15%);
osteochondrodysplasia (not stated or
excluded)

Interrater, intrarater
and
interrespondent
reliability

Convenience

N = 116

Intrarater reliability

Criterion validity

Intrarater and
interrespondent
reliability

Criterion validity

Content validity
Responsiveness

Criterion validity

Psychometrics

Cohort = cerebral palsy: 30 (46%),

developmental delay: 15 (23%), other
(including chromosomal anomalies):
20 (31%); osteochondrodysplasia (not
stated or excluded)

Cohort = typical: 20 (50%); spina bifida:

10 (25%), arthritic conditions: 10
(25%); osteochondrodysplasia (no)
N = 46
Convenience
Cohort = ABI: 23 (50%), multiple,
significant disabilities: 23 (50%).
Breakdown not specified;
osteochondrodysplasia (not stated or
excluded)
N = 65
Convenience

N = 40

Sample size and representation

TABLE 2. Studies of the Clinimetric Properties of the WeeFIM, PEDI, and ASK (Continued)

Downloaded by [dessy rahma] at 01:56 13 November 2015

D
I

All items addressed

All items addressed

Missing H3: small sample

size
Missing B5 administration
independence, B9 test
conditions, B8 short
interval between tests,
(<2 weeks) and B3
small sample size
Missing B9 test conditions

All items addressed

Missing I6 interim period
description, I7
proportion of patients
changed

Missing H3: small sample

size

COSMIN checklist

89

Children with disabilities

Age, 517 years
Sourcehospital clinics

Pencharz, Young,
Owen, & Wright
(2001)

N = 166
Cohort = fractures: 35 (21%); neuromuscular:
42 (25%); spinal: 31 (19%); other
musculoskeletal: 58 (35%; including skeletal
dysplasia 2)

Consecutive

N = 28
Not stated
Cohort = rheumatology: 6 (21%); orthopaedics:
6 (21%); physical therapy: 3 (11%); spina
bifida: 4 (14%) and amputee: 9 (32%);
osteochondrodysplasia (not stated or
excluded)
N = 200
Selective
Cohort = neuromuscular: 88 (44%);
orthopaedic: 71 (35%); arthritic conditions:
41 (21%); osteochondrodysplasia (not stated
or excluded)
Interrater reliability
Responsiveness
Criterion validity

Validity (all)

Testretest reliability
Content validity

D
E
G
H
B
I
H

B
D

All items addressed

All items addressed
All items addressed
All items addressed
All items addressed
All items addressed
All items addressed

All items addressed

All items addressed

Note: Measurement properties identified in the COSMIN checklist: B = reliability; D = content validity; E = structural validity; G = cross-cultural validity; H = criterion validity; I =
responsiveness.

Children with disabilities

Age, 515 years
Sourcehospital clinics

Children with disabilities

Age, 515 years
Sourcehospital clinics

Young, Williams,
Yoshida, & Wright
(2000)

ASK
Young et al. (1995)

Downloaded by [dessy rahma] at 01:56 13 November 2015

90

ASK
Children with physical
stabilities

Both

Children with and

PEDIwithout disabilities
Typically developing
children
Children with physical
disabilities

Children with physical

disabilities

WeeFIM
Typically developing
children

Tool

ICC = 0.99 (Young et al., 2000)

ICC = 0.99 for all domains

(Wassenberg-Severijnen
et al., 2003)

ICC = 0.950.99 (Berg et al.,

2004)
ICC = 0.810.95 (Ziviani et al.,
2001)
Spearman rank coefficient >0.9
(Engelbert et al., 1997)

ICC = 0.880.99 (mean 0.97;

Jongjit et al., 2006)
ICC = 0.991.0 (Wong et al.,
2002)
ICC = 0.820.94 (Ziviani et al.,
2001)
ICC = 0.730.98 (Ottenbacher
et al., 1997)

Interrater

TestRetest

No difference between modes of

administration (Sperle et al.,
1997; Ottenbacher et al., 1996)

Equivalence

Spearman rank coefficient >

0.9 (Engelbert et al., 1997)
ICC > 0.8 for all domain
summary scores
ICC = 0.630.98 for content
areas (Nichols &
Case-Smith 1996)

ICC = 0.97 (2-week interval)

High concordance between child and
parent scores; ICC = 0.96
(Young et al., 1995)

ICC = 0.910.97
(Wassenberg-Severijnen et al.,
2003)

ICC = 0.850.98 (Ottenbacher ICC = 0.730.94 (different rater long ICC = 0.98 (for both telephone and
et al., 1997)
interval; Ottenbacher et al., 1997)
in-person; Ottenbacher et al.,
ICC = 0.850.98 (same rater long
1997)
interval; Ottenbacher et al., 1997)
ICC = 0.97 and 0.98 (Ottenbacher et
al., 1996)
ICC 0.99 (Berg et al., 2004)

ICC = 0.90.99 (Jongjit et al.,

2006)

Intrarater

TABLE 3. Reliability of the WeeFim, PEDI, and ASK for Children with Osteochondrodysplasia

Downloaded by [dessy rahma] at 01:56 13 November 2015

Self-Care Measures in Osteochondrodysplasia

91

2001), although these findings are not specific to children with osteochondrodysplasia (Table 4).

Downloaded by [dessy rahma] at 01:56 13 November 2015

Content Validity
Forty-four percent of the items on the WeeFIM, 40% of the items on the PEDI functional skill component, 37% of the items on the PEDI caregiver assistance component, and 26% of the items on the ASK were judged to address ICF d5 self-care domain categories (Table 1). Items in the WeeFIM were adapted for children from the
original adult version, the Functional Independence Measure (FIM). Rasch analysis of the WeeFIM ratings identified that items in the motor dimension formed
a unidimensional construct with acceptable measurement properties (Chen et al.,
2005). The PEDI was developed using Rasch analysis scales by a panel of experts in
pediatric rehabilitation. The ASK was developed from first principles rather than
being a modification of existing tools or items and had clear confirmation of unidimensionality with a hierarchical structure among the items (Young et al., 2000;
Table 4).
Responsiveness to Change
Little research exists on responsiveness to change for the three measures, and no
studies were found for children with osteochondrodysplasia. The PEDI was reported by Haley et al. (1992) to be very successful in detecting recovery of function in 23 children with mild to moderate traumatic injuries assessed at 1 and
6 months after discharge from hospital. However, when a group of 23 children
with multiple, significant disabilities were assessed 8 months apart, only the mobility domain showed positive change (Haley et al., 1992). The authors concluded
that the PEDI is selectively responsive to change in certain clinical cohorts and
recommended further studies to examine the responsiveness of the PEDI in other
populations of children. Ottenbacher et al. (2000) examined responsiveness for the
WeeFIM using five statistical procedures (Reliability Change Index, Proportional
Change Index, effect size, standardized response means, and paired t-tests). This
study found that all five indexes indicated that the WeeFIM was able to reliably
document change in functional abilities over a 1-year period for a group of 174 children with developmental disabilities, receiving early intervention or school therapy. Responsiveness of the ASK was assessed in 34 children who demonstrated
what was described as a clinically important change over a 6-month period. The
performance version of the ASK was found to be more responsive to change than
the capability version, and the ASK performance was found to be slightly more responsive to change than the Childhood Health Assessment Questionnaire (CHAQ;
Young et al., 2000).
Clinical Utility
All three measures provide clear instructions for administration and guidelines for
scoring (Table 1). No specific equipment is required for completion. Administration time is considerably less for the ASK (59 min) and the WeeFIM (1520 min)
compared with the PEDI (4560 min). The WeeFIM and the ASK are more versatile than the PEDI when considering administration methods. Unlike the PEDI,
the ASK also can be completed through postal survey and the WeeFIM through

92

Content Validity

Item hierarchy shown

(Young et al., 2000)

Examined using a panel of

31 experts in pediatric
rehabilitation, child
development, occupational
therapy, physical therapy,
and speech pathology
(Haley et al., 1992)
Developed from first
principles

Items developed with

Rasch RSA (Haley et al.,
1992)

High correlation (Pearson)

between CHAQ and ASK
performance (0.81) and ASK
capability (0.82; Young et al.,
2000)
Moderate correlation
(Spearman) with HUI for similar
constructs (0.43) and low for
dissimilar constructs (0.03,
Young et al., 2000)

PEDI is able to discriminate

between disabled and
nondisabled children (Feldman
et al., 1990)

Moderate correlation with CHAQ

(0.49; Young et al., 2000)
High correlation with PODCI
(0.78; Pencharz et al., 2001)

ASK capability less responsive than the ASK performance

(Young et al., 2000)

Moderate to high correlation

(Pearson) with PDMS (0.640.95;
Nichols & Case-Smith 1996)
High correlation (Spearman) for
PEDI self-care, mobility, and
communication items (Ziviani et
al., 2001)
ASK performance is as responsive or slightly more
High correlation (Pearson)
between parent and child reports responsive than CHAQ in 34 children with musculoskeletal
issues over 6-month period (Young et al., 2000)
(0.92; Young et al., 1995)

Significant changes in all domains for 23 children with mild

to moderate traumatic injuries assessed within 6 months of
leaving hospital. Only mobility domain showed positive
change for 23 children with multiple disabilities assessed 8
months apart.
Selectively responsive to change in certain clinical samples
only (Haley et al., 1992)

Reliability Change Index, Proportional Change Index,

effect size, standardized response means and paired
t-tests applied to results from 174 children. Able to
indicate significantly reliable changes in functional
performance over 3 time points in a 1-year period in
children with chronic disabilities (Ottenbacher et al., 2000)

Measures similar skill levels as

BDIST (0.830.94) & VABS
(0.720.91; Ottenbacher et al.,
1999)
Spearman correlation coefficients
> 0.88 for self-care, mobility, and
communication (PEDI; Ziviani et
al., 2001)
Moderate to high correlation
(Pearson) with BDIST (0.700.80;
Feldman et al., 1990)

Rasch analysis showed

differences in item difficulty
across ages (Chen et al., 2005)

Developmental differences
among children with disabilities
mimics that of children without
disabilities (Chen et al., 2005)
Children in the disabled group
scored significantly lower than
the nondisabled group
(Feldman et al., 1990)

Responsiveness to Change

Criterion Validity

Construct Validity

Note: BDIST: Battelle Development Inventory Screening Test; VABS: Vineland Adaptive Behavior Scales; PDMS: Peabody Developmental Motor Scales; CHAQ: Childhood Health Assessment
Questionnaire; HUI: Health Utilities Index; PODCI: Pediatric Outcomes Data Collection Instrument.

ASK

PEDI

WeeFIM Rasch analysis

demonstrated distinct
motor and cognitive
domains (Chen et al., 2005)

Tool

TABLE 4. Validity of the WeeFIM, PEDI, and ASK for Children with Osteochondrodysplasia

Downloaded by [dessy rahma] at 01:56 13 November 2015

Self-Care Measures in Osteochondrodysplasia

93

telephone interview. Training is recommended but not essential for administration

of the WeeFIM and the PEDI. No training is required for administration of the
ASK.

Downloaded by [dessy rahma] at 01:56 13 November 2015

DISCUSSION
A systematic review identified the WeeFIM, PEDI, and ASK as having adequate
reliability, validity, and clinical utility for use with children with osteochondrodysplasia. None of the three measures were designed specifically for children with osteochondrodysplasia, and consequently, clinimetric information for children with
osteochondrodysplasia is limited. All three measures, however, have been validated
for use with children with physical impairments and psychometric properties most
likely generalize to children with osteochondrodysplasia. We recommend the following considerations when deciding on which of the three measures to use: (a)
the ICF domain function of interest (capacity, capability, or performance), (b) the
purpose for assessment (clinical or research, screening or treatment planning), (c)
the perspective required (parent, child, or clinician), and (iv) the time and method
available for completion (face-to-face, telephone, or mail).
All three assessments provide useful information on self-care performance (what
the child actually does do in his/her natural environment, i.e., usual performance).
The PEDI and ASK also provide information on capability (what the child can
do in his/her natural environment, i.e., best performance). As a consequence, both
measures can be used to assess the childs performance in real life settings and also
provide information on environmental modifications that might improve performance.
Time for completion varies among the three assessments and is a consideration
for utility. All three assessments include items for the self-care domain categories of
washing oneself (d510), caring for body parts (d520), toileting (d530), and dressing
(d540). Items for eating (d550) and drinking (d560) are included on the PEDI and
WeeFIM. The PEDI provides the most comprehensive information (217 items);
however, it takes the most time to complete. The ASK (30 items) and WeeFIM (18
items) have fewer items and can be completed in person or by phone interview. The
versatility and time for completion of the ASK and WeeFIM are advantageous in a
research setting when multiple measures or remote data collection are required. If
time and resources permit, the PEDI is a more comprehensive measure, particularly
for planning treatment or discharge to another setting.
Identification of environmental and personal factors that restrict participation
and the need for caregiver assistance is advantageous for children with osteochondrodysplasia. Both the WeeFIM and the PEDI consider the childs performance
from a caregivers perspective, giving an indication of any additional burden of
care issues and equipment required. This provides significant additional detail
that gives the examiner the ability to quantify and discuss options for potential
modifications to the environment that may increase the childs ability to be more
independent. The ASK presents a unique opportunity to evaluate the impact of
musculoskeletal impairment on self-care skills from the childs own perspective
that could assist therapists to identify environmental and personal factors that
may be influencing performance and therefore to identify broader treatment goals

94

Ireland and Johnston

Downloaded by [dessy rahma] at 01:56 13 November 2015

within a client-, family-, and community-centered context. The ASK and the PEDI
are appropriate only for children, while the WeeFIM has the added bonus of being
able to document performance over the lifespan via the FIM designed for older
children, adolescents, and adults.
The main limitation of this systematic review was the small number of studies
that have examined the psychometric properties of measures in children with osteochondrodysplasia. A second limitation may be that inclusion of only self-care
measures published in English has excluded individual assessment tools or specific
items with relevance to children with osteochondrodysplasia. Finally, the International Working Group on Constitutional Diseases of Bone continues to identify
new subtypes that were previously labeled as genetic or congenital abnormalities.
It is possible that future reviews may include articles that we have excluded.
CONCLUSIONS
This systematic review identified three measures, the WeeFIM, PEDI, and ASK,
that are used to evaluate self-care performance in children with a functional or
physical disability and have properties rendering them appropriate for children
with osteochondrodysplasia. Additional research is required to further examine the
psychometric properties of each assessment when applied with children with osteochondrodysplasia. Each measure has advantages and disadvantages. Differences in
purpose and method of administration provide therapists the opportunity to select
a measure to match the desired ICF self-care domain of interest.
Declaration of interest: The authors report no conflict of interest. The authors
alone are responsible for the content and writing of this paper.
ABOUT THE AUTHORS
Penelope Ireland, BPhty (Honors), is a senior physiotherapist at the Queensland Paediatric Rehabilitation Service, The Royal Childrens Hospital, Brisbane,
Australia. Leanne M. Johnson, BPhty (Honors), PhD, is an adjunct senior lecturer
in the Division of Physiotherapy, School of Health and Rehabilitation Sciences, The
University of Queensland, Brisbane, Australia.
REFERENCES
Amako, M., Fassier, F., Hamdy, R. C., Aarabi, M., Montpetit, K., & Glorieux, F. H. (2004). Functional analysis of upper limb deformities in osteogenesis imperfecta. Journal of Pediatric Orthopaedics, 24(6), 689694.
Apajasalo, M., Sintonen, H., Rautonen, J., & Kaitila, I. (1998). Health-related quality of life of
patients with genetic skeletal dysplasias. European Journal of Pediatrics, 157(2), 114121.
Baitner, A. C., Maurer, S. G., Gruen, M. B., & Di Cesare, P. E. (2000). The genetic basis of the
osteochondrodysplasias. Journal of Pediatric Orthopaedics, 20(5), 594605.
Berg, M., Jahnsen, R., Froslie, K. F., & Hussain, A. (2004). Reliability of the Pediatric Evaluation
of Disability Inventory (PEDI). Physical and Occupational Therapy in Pediatrics, 24(3), 6177.
Braun, S., Msall, M. E., & Granger, C. V. (1991). Manual for the Functional Independence Measure for Children (WeeFIM). Version 1.4. Buffalo, NY: Centre for Functional Assessment Research, Uniform Data System for Medical Rehabilitation, State University of New York.

Downloaded by [dessy rahma] at 01:56 13 November 2015

Self-Care Measures in Osteochondrodysplasia

95

Chen, C. C., Bode, R. K., Granger, C. V., & Heinemann, A. W. (2005). Psychometric properties
and developmental differences in childrens ADL item hierarchy: A study of the WeeFIM
instrument. American Journal of Physical Medicine & Rehabilitation, 84(9), 671679.
Collins, M. T., Kushner, H., Reynolds, J. C., Chebli, C., Kelly, M. H., Gupta, A., et al. (2005). An
instrument to measure skeletal burden and predict functional outcome in fibrous dysplasia of
bone. Journal of Bone and Mineral Research, 20(2), 219226.
Damignani, R., Young, N. L., Cole, W. G., Anthony, A. M., & Badley, E. M. (2004). Impairment
and activity limitation associated with epiphyseal dysplasia in children. Archives of Physical
Medicine and Rehabilitation, 85(10), 16471652.
Dawson, B., & Trapp, R. (2004). Basic and clinical biostatistics (4th ed.). New York: Macraw Hills.
Engelbert, R. H., Uiterwaal, C. S., Gerver, W. J., van der Net, J. J., Pruijs, H. E., & Helders, P. J.
(2004). Osteogenesis imperfecta in childhood: Impairment and disability. A prospective study
with 4-year follow-up. Archives of Physical Medicine and Rehabilitation, 85(5), 772778.
Engelbert, R. H. H., Custers, J. W. H., van der Net, J., van der Graff, Y., Beemer, F. A., & Helders,
P. J. M. (1997a). Functional outcomes in osteogenesis imperfecta: Disability profiles using the
PEDI. Pediatric Physical Therapy, 9, 1822.
Engelbert, R. H., van der Graaf, Y., van Empelen, R., Beemer, F. A., & Helders, P. J. (1997b).
Osteogenesis imperfecta in childhood: Impairment and disability. Pediatrics, 99(2), E3.
Feldman, A. B., Haley, S. M., & Coryell, J. (1990). Concurrent and construct validity of the Pediatric Evaluation of Disability Inventory. Physical Therapy, 70(10), 602610.
Furlong, W., Barr, R. D., Feeny, D., & Yandow, S. (2005). Patient-focussed measures of functional
health care status and health-related quality of life in pediatric orthopedics: A case study in
measurement selection. Health and Quality of Life Outcomes, 3(3), 115.
Guyatt, G., Walter, S., & Norman, G. (1987). Measuring change over time: Assessing the usefulness of evaluative instruments. Journal of Chronic Diseases, 40(2), 171178.
Haley, S. M., Costner, W. J., Ludlow, L. H., Haltiwanger, J. T., & Andrellos, P. J. (1992). Pediatric
Evaluation of Disability Inventory (PEDI). Development, Standardisation and Administration
Manual (version 1). Boston: New England Medical Center Hospitals.
Harvey, A., Robin, J., Morris, M. E., Graham, H. K., & Baker, R. (2008). A systematic review of
measures of activity limitation for children with cerebral palsy. Developmental Medicine and
Child Neurology, 50(3), 190198.
Holsbeeke, L., Ketelaar, M., Schoemaker, M. M., & Gorter, J. W. (2009). Capacity, capability,
and performance: Different constructs or three of a kind? Archives of Physical Medicine and
Rehabilitation, 90(5), 849855.
Horton, W. A., Hall, J. G., & Hecht, J. T. (2007). Achondroplasia. Lancet, 370(9582), 162172.
Huang, R. P., Ambrose, C. G., Sullivan, E., & Haynes, R. J. (2006). Functional significance of
bone density measurements in children with osteogenesis imperfecta. Journal of Bone and
Joint SurgeryAmerican Volume, 88(6), 13241330.
Jessen, E. C., Colver, A. F., Mackie, P. C., & Jarvis, S. N. (2003). Development and validation of a
tool to measure the impact of childhood disabilities on the lives of children and their families.
Child Care, Health and Development, 29(1), 2134.
Jongjit, J., Komsopapong, L., Saikaew, T., Wanich, U., Chewapanich, S., Udomsubpayakul, U.,
et al. (2006). Reliability of the functional independence measure for children in normal Thai
children. Pediatrics International, 48(2), 132137.
Kelly, M. H., Brillante, B., Kushner, H., Gehron Robey, P., & Collins, M. T. (2005). Physical
function is impaired but quality of life preserved in patients with fibrous dysplasia of bone.
Bone, 37(3), 388394.
Land, C., Rauch, F., Montpetit, K., Ruck-Gibis, J., & Glorieux, F. H. (2006). Effect of intravenous
pamidronate therapy on functional abilities and level of ambulation in children with osteogenesis imperfecta. Journal of Pediatric, 148(4), 456460.
Law, M. (2004). Outcome measures rating form. Ontario, Canada: Canchild Centre for Disability
Research.
Leet, A. I., Wientroub, S., Kushner, H., Brillante, B., Kelly, M. H., Robey, P. G., et al. (2006). The
correlation of specific orthopaedic features of polyostotic fibrous dysplasia with functional
outcome scores in children. Journal of Bone and Joint SurgeryAmerican Volume, 88(4),
818823.

Downloaded by [dessy rahma] at 01:56 13 November 2015

96

Ireland and Johnston

Mokkink, L. B., Terwee, C. B., Patrick, D. L., Alonso, J., Stratford, P. W., & Knol, D. L. (2009).
The COSMIN checklist manual. Retrieved 30 October 2010, from http://www.cosmin.nl
Mokkink, L. B., Terwee, C. B., Patrick, D. L., Alonso, J., Stratford, P. W., Knol, D. L., et al. (2010).
The COSMIN checklist for assessing the methodological quality of studies on measurement
properties of health status measurement instruments: An international Delphi study. Quality
of Life Research, 19(4), 539549.
Nichols, D. S., & Case-Smith, J. (1996). Reliability and validity of the Paediatric Evaluation of
Disability Inventory. Pediatric Physical Therapy, 8, 1524.
Ottenbacher, K. J., Msall, M. E., Lyon, N., Duffy, L. C., Granger, C. V., & Braun, S. (1999). Measuring developmental and functional status in children with disabilities. Developmental Medicine
and Child Neurology, 41(3), 186194.
Ottenbacher, K. J., Msall, M. E., Lyon, N., Duffy, L. C., Ziviani, J., Granger, C. V., et al. (2000).
The WeeFIM instrument: Its utility in detecting change in children with developmental disabilities. Archives of Physical Medicine and Rehabilitation, 81(10), 13171326.
Ottenbacher, K. J., Msall, M. E., Lyon, N. R., Duffy, L. C., Granger, C. V., & Braun, S. (1997).
Interrater agreement and stability of the Functional Independence Measure for Children
(WeeFIM): Use in children with developmental disabilities. Archives of Physical Medicine
and Rehabilitation, 78(12), 13091315.
Ottenbacher, K. J., Taylor, E. T., Msall, M. E., Braun, S., Lane, S. J., Granger, C. V., et al. (1996).
The stability and equivalence reliability of the functional independence measure for children
(WeeFIM). Developmental Medicine and Child Neurology, 38(10), 907916.
Pencharz, J., Young, N. L., Owen, J. L., & Wright, J. G. (2001). Comparison of three outcomes
instruments in children. Journal of Pediatric Orthopaedics, 21(4), 425432.
Sakkers, R., Kok, D., Engelbert, R., van Dongen, A., Jansen, M., Pruijs, H., et al. (2004). Skeletal
effects and functional outcome with olpadronate in children with osteogenesis imperfecta: A
2-year randomised placebo-controlled study. Lancet, 363(9419), 14271431.
Sakzewski, L., Boyd, R., & Ziviani, J. (2007). Clinimetric properties of participation measures for
5- to 13-year-old children with cerebral palsy: A systematic review. Developmental Medicine
and Child Neurology, 49(3), 232240.
Seikaly, M. G., Kopanati, S., Salhab, N., Waber, P., Patterson, D., Browne, R., et al. (2005). Impact
of alendronate on quality of life in children with osteogenesis imperfecta. Journal of Pediatric
Orthopaedics, 25(6), 786791.
Sperle, P. A., Ottenbacher, K. J., Braun, S. L., Lane, S. J., & Nochajski, S. (1997). Equivalence reliability of the functional independence measure for children (WeeFIM) administration methods.
American Journal of Occupational Therapy, 51(1), 3541.
Tuysuz, B. (2004). A new concept of skeletal dysplasias. Turkish Journal of Pediatrics, 46(3),
197203.
Uniform Data System for Medical Rehabilitation. (2006). The WeeFIM II clinical guide, version
6.0. Buffalo, NY: Author.
Wassenberg-Severijnen, J. E., Custers, J. W., Hox, J. J., Vermeer, A., & Helders, P. J. (2003). Reliability of the Dutch Pediatric Evaluation of Disability Inventory (PEDI). Clinical Rehabilitation, 17(4), 457462.
Wong, V., Wong, S., Chan, K., & Wong, W. (2002). Functional Independence Measure (WeeFIM)
for Chinese children: Hong Kong cohort. Pediatrics, 109(2), E36.
World Health Organization. (2007). International classification of functioning, disability and
healthChildren and youth version. Switzerland: Geneva.
Young, N. L., Williams, J. I., Yoshida, K. K., & Wright, J. G. (2000). Measurement properties of
the activities scale for kids. Journal of Clinical Epidemiology, 53(2), 125137.
Young, N. L., Yoshida, K. K., Williams, J. I., Bombardier, C., & Wright, J. G. (1995). The role of
children in reporting their physical disability. Archives of Physical Medicine and Rehabilitation, 76(10), 913918.
Ziviani, J., Ottenbacher, K. J., Shephard, K., Foreman, S., Astbury, W., & Ireland, P. (2001). Concurrent validity of the functional independence measure for children (WeeFIM) and the pediatric evaluation of disabilities inventory in children with developmental disabilities and acquired brain injuries. Physical and Occupational Therapy in Pediatrics, 21(23), 91101.