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The Manual Ability Cerebral palsy (CP) is the most common neurodisability in

children. It has been defined as ‘a disorder of movement and


posture due to a defect or lesion of the immature brain’ (Bax
Classification System 1964) and as ‘a group of non-progressive, but often changing
motor impairment syndromes secondary to lesions or anom-
(MACS) for children alies of the brain arising in the early stages of development’
(Mutch et al. 1992). A proposed new definition of CP focuses

with cerebral palsy: on a broader perspective of activity restriction and disability


(Bax et al. 2005). Classification of subtypes of CP has been pro-
posed based on location of the lesion, part of the body involved,
scale development and or degree of impairment (Ingram 1964). However, none of
these descriptions provides information about the child’s func-
evidence of validity tional abilities in daily life. One recent classification with a clear
functional approach is the Gross Motor Function Classification

and reliability System (GMFCS; Palisano et al. 1997). The GMFCS describes
gross motor function in terms of self-initiated movements
with emphasis on function in sitting and walking. This classi-
fication is based on the concept of disability (World Health
Ann-Christin Eliasson* PhD OT; Organization 1980) and of functional limitation (National
Lena Krumlinde-Sundholm PhD OT, Department of Woman Institutes of Health 1993). The GMFCS has been widely adopt-
and Child Health, Karolinska Institute, Stockholm; ed internationally, suggesting that it has filled a gap in func-
Birgit Rösblad PhD PT, Department of Community Medicine tional classification (Morris and Bartlett 2004). No similar
and Rehabilitation, University of Umeå, Umeå; classification of hand function has been available, but one is
Eva Beckung PhD PT, Queen Silvia Children’s Hospital, needed to broaden the functional perspective of CP beyond
Göteborg; gross motor issues.
Marianne Arner PhD MD, Hand Unit, Department of Classification is the process of grouping data, persons, or
Orthopaedics, Lund University Hospital, Lund; objects into classes according to common characteristics,
Ann-Marie Öhrvall MSc OT, Department of Habilitation, thereby reducing the number of data elements. The usefulness
Stockholm County Council, Stockholm, Sweden; of a classification depends on how understandable and clear
Peter Rosenbaum MD FRCP, CanChild Centre for Childhood the descriptions are, and how meaningful different levels are.
Disability Research, McMaster University, Hamilton, The most common way to classify hand function in CP has
Ontario, Canada. been to use terms like ‘mild’, ‘moderate’, and ‘severe’ impair-
ment (Claeys et al. 1983). Although some other classifica-
*Correspondence to first author at Neuropaediatric tions are available (House et al. 1981, Zancolli and Zancolli
Research Unit Q2:07, Astrid Lindgren Children’s Hospital, 1981, Krägeloh-Mann et al. 1993, Beckung and Hagberg 2002),
SE-171 76 Stockholm, Sweden. all of them classify aspects of grasping rather than functional
E-mail: ann-christin.eliasson@kbh.ki.se performance and none has been tested for reliability.
This proposed new classification, the Manual Ability
Classification System (MACS), is designed to classify how chil-
dren with CP use their hands when handling objects in daily
activities. The focus is on manual ability, as defined in the
The Manual Ability Classification System (MACS) has been International Classification of Functioning, Disability and
developed to classify how children with cerebral palsy (CP) use Health (ICF; World Health Organization 2001). It has its start-
their hands when handling objects in daily activities. The ing point in upper limb function but is also influenced by envi-
classification is designed to reflect the child’s typical manual ronmental, personal, and contextual factors. MACS follows
performance, not the child’s maximal capacity. It classifies the Penta and coworkers’ definition of manual ability as ‘the capac-
collaborative use of both hands together. Validation was based ity to manage daily activities that require the use of the upper
on the experience within an expert group, a review of the limbs, whatever the strategies involved, which can be observed
literature, and thorough analysis of children across a spectrum from activity performance in the person’s everyday context’
of function. Discussions continued until consensus was reached, (Penta et al. 2001). MACS is not designed to classify best capaci-
first about the constructs, then about the content of the five ty and does not mean to distinguish different capacities
levels. Parents and therapists were interviewed about the between hands. MACS reports the collaboration of both hands
content and the description of levels. Reliability was tested together: it is not an assessment of each hand separately. It
between pairs of therapists for 168 children (70 females, 98 looks for the children’s manual ability to ‘handle object(s) in
males; with hemiplegia [n=52], diplegia [n=70], tetraplegia daily life’. By that we mean those activities that are relevant and
[n=19], ataxia [n=6], dyskinesia [n=19], and unspecified CP age appropriate for the child including, for example, eating,
[n=2]) between 4 and 18 years and between 25 parents and dressing, playing, writing, etc.; these are distinct from
their children’s therapists. The results demonstrated that MACS advanced skilled activities that require special training for per-
has good validity and reliability. The intraclass correlation formance, such as playing a musical instrument. Neither does it
coefficient between therapists was 0.97 (95% confidence classify activities closely related to academic skills in school.
interval 0.96–0.98), and between parents and therapist was 0.96 The emphasis of MACS is on handling objects in an individual’s
(0.89–0.98), indicating excellent agreement. personal space, the space immediately close to one’s body, as

Developmental Medicine & Child Neurology 2006, 48: 549–554 549


distinct from objects that are not within reach, thus minimizing environments, mainly in their homes or schools. Using those
the potential confounding influence of limitations in gross videos, we discussed and defined the classification levels based
motor function. Distinctions between the levels are based on on the assumption that five levels would be suitable, if possi-
the child’s ability to handle objects, i.e. the quantity and quality ble, to correspond to the structure of GMFCS. To reduce the
of performance and need for assistance or adaptations to per- complexity of the work we agreed to start by developing a
form manual tasks in everyday life. classification suitable for children aged 8 to 12 years. We have
The aims of this report are to describe the development worked with a Swedish and an English language version of
of MACS and provide evidence of its validity and reliability. the classification in parallel.
Efforts were made to follow recommendations about accu- The study was conducted after approval from the Ethics
mulating evidence for scale validity according to guidelines Research Committee at the Karolinska Hospital, Sweden, and
published in Standards for Educational and Psychological the State Government, Victoria, Australia. Informed consent
Testing (American Educational Research Association 1999). was obtained from the parents of the children.

Method External processes for validation


PROCESSES OF DEVELOPMENT AND VALIDATION OF THE When the first version of the classification was outlined an
CLASSIFICATION external validation process was initiated, by involving profes-
The first step in developing MACS was to assemble an expert sionals within paediatric rehabilitation and parents of children
group, comprising six people (two occupational therapists, with CP. To collect opinions from professionals we presented
two physical therapists, one hand surgeon, and one develop- preliminary versions of the classification at national and inter-
mental paediatrician), each with over 20 years of clinical national conferences (Sweden 2002, Canada 2003, Norway
experience and research in paediatric rehabilitation. It was 2003). Interactive poster presentations were used where the
considered important to involve people from different pro- participants were encouraged to classify children from video
fessional backgrounds and experience of research (Fink et al. clips and to comment on the classification. Oral presenta-
1984). The work in the expert group has proceeded through tions were also given after which the audience was invited to
workshops, telephone conferences, and e-mail contacts for discuss and offer feedback. Comments and suggestions col-
more than 3 years, with the construct development of the lected on these occasions were brought back to the expert
classification as an ongoing process. First, an inventory of group and processed. The refinement of the wording and
available tests and classifications of hand function was con- the distinctions between levels was a continuous process
structed. An essential next step was to clarify the conceptual until the reliability study started.
background and to define and formulate the basic underlying An additional step for validity testing was to investigate if
construct. Discussions occurred until consensus was reached this concept was meaningful for parents of children with CP
in the expert group. (Öhrvall 2005). After a brief introduction, 25 parents classi-
It then became important to describe the content of the fied their child according to the MACS leaflet (Appendix I).
different levels of the classification. We video recorded chil- Thereafter, each parent participated in a semi-structured tape-
dren with different subtypes of CP and fields of functioning recorded interview following an interview guide (Kvale 1996).
when performing various manual activities in their natural The questions focused on the parents’ thoughts about the
classification. They were asked if this concept was meaning-
ful for describing their child’s hand function. They were then
asked for their rationale for choosing a particular level instead
Table I: Distribution of children by age and by Gross Motor of others. A third set of questions concerned their judgement
Function Classification System (GMFCS) levels (n=164) of the overall usefulness of the classification. The same pro-
cedure was repeated with the child’s occupational therapist
Age (y) n GMFCS levels or, sometimes, physiotherapist.
I II III IV V

4 8 3 2 – – 3 Data processing of the interviews


5 15 3 4 1 2 5 Textual analysis of the materials from parents and therapists
6 10a 2 2 3 2 – was performed separately (Kvale 1996), based on the three
7 12 1 4 2 3 2 sets of questions, and for each level of MACS. The remarks
8 16 9 1 1 3 2 were analyzed to understand if the distinction between lev-
9 13 4 3 5 1 – els was apparent or not (Polit and Hungler 1995).
10 29 9 2 6 7 5
11 16 4 4 4 1 3 PROCESS OF TESTING THE RELIABILITY OF THE CLASSIFICATION
12 16 5 5 – 5 1
Design
13 3 2 – – 1 –
Interrater reliability was studied by asking pairs of therapists
14 7 1 2 3 1 –
15 10 – 2 2 4 2 to classify the same child, and by asking parents and thera-
16 4 2 1 – – 1 pists to classify the same child. This meant that several thera-
17 3 1 – – – 2 pists classified more than one child whereas each child was
18 6a – – 1 2 – classified only once by each therapist.
Total 168 (164) 47 32 28 32 26 The parents and almost all therapists received a brief intro-
Per cent – 28 20 17 20 16 duction to MACS with the MACS leaflet, which they were
aGMFCS level missing for one child at 6y and three children at 18y.
encouraged to read thoroughly (Appendix I). They were also
invited to discuss uncertainties with the classification and

550 Developmental Medicine & Child Neurology 2006, 48: 549–554


rating procedure with someone from the expert group before seemed to be related to the interpretation of certain words,
actually rating the children. for example ‘independence’. One parent emphasized that
the child was independent in simple tasks, whereas the ther-
Procedure and participants apist thought about activities that were more complex where
As originally planned, we started by classifying children aged the child needed continuous support.
between 8 and 12 years. During the process of developing When discussing the overall usefulness of the classification,
MACS we frequently received comments from parents as well parents suggested that MACS could be used for several purpos-
as therapists that the classification could be valid for children es: e.g., in contact with the social services and local council to
at other ages as well. We decided therefore to include chil- discuss the need for support and personal assistance; as infor-
dren between 4 and 18 years in the reliability testing. In total mation for parents of infants with CP; for newly employed staff
168 children between 4 and 18 years (98 males, 70 females) working with the child; and as information between pro-
were classified by 93 therapists from six cities in Sweden and fessionals working with the same person. The therapists
one city in Australia (Table I). The severity of disability, accord- expressed similar ideas but also emphasized the ability to use
ing to GMFCS, varies, as do the subtypes of CP (Tables I and II). MACS in the clinic for planning of treatment and goal setting.
The Swedish classification of CP subtypes was used (Hagberg
and Hagberg 1993). INTERRATER RELIABILITY BETWEEN THERAPISTS
The classifications of 168 children between 4 and 18 years
Interrater reliability between therapists for children aged were analyzed. ICC was 0.97 (95% confidence interval [CI]
4 to 18 years 0.96–0.98) for the whole group, indicating excellent agree-
The 168 children were classified independently by two thera- ment (Table III). The total agreement was 84.5%. When ICC
pists (Table III). Twenty children from Melbourne, Australia, was calculated for the different ages, there were similar
were analyzed first and separately to control for cultural dif- results (Table IV). A group of 20 children from Australia was
ferences, and were then included in further calculations. included. When this group was analyzed separately no differ-
ences in the understanding of the MACS were found; howev-
Interrater reliability between parents and therapists er, ICC was slightly lower at 0.91 (95% CI 0.77–0.96).
Twenty-five children between 8 and 12 years were classified
independently by their parents and one therapist, in most cases INTERRATER RELIABILITY BETWEEN PARENTS AND THERAPISTS
an occupational therapist. The children were selected to cover ICC between parents and therapists was 0.96 (95% CI 0.89–
what we assumed would be a wide range of functional abili- 0.98), indicating excellent agreement for the 25 children
ties, with the intention of representing all levels of MACS.

Statistics
For analysis of interrater reliability a one-way random effects Table II: Types of cerebral palsy (CP) by Manual Ability
model (referred to as intraclass correlation coefficient [ICC] Classification System (MACS) rating by professionals
1) was used, which, according to Shrout and Fleiss (1979), is
suitable when different raters are assessing the children using Type of CP MACS levels
I II III IV V Total %
average measures. The ICC result is equivalent to a weighted
kappa (Fleiss and Cohen 1973). Rater agreement, the coeffi- Hemiplegia 10 38 4 – – 52 31
cient of total agreement between therapists (number of chil- Diplegia 12 23 18 15 2 70 42
dren with total agreement divided by total number of children; Tetraplegia/quadriplegia – – 1 9 9 19 12
Polit and Hungler 1994) was calculated. For descriptive analy- Ataxia 1 2 2 1 – 6 4
sis, the result from the first rater of MACS was used. For cor- Dyskinetic CP – – 3 4 12 19 12
relation between MACS and GMFCS, Spearman’s rank corre- Total 23 63 28 29 23 166a –
lation was used. % 14 38 17 17 14 – –
aCP type was unavailable for two children.
Results
VALIDATION OF THE CLASSIFICATION
All parents and therapists responded positively to the ques-
tion about whether MACS was built on a useful construct for Table III: Numbers of children 4 to 18 years in each Manual
describing the child’s hand function. They also found the dif- Ability Classification System level as rated by two therapistsa
ferences between the five levels meaningful and easy to under-
Therapist 2
stand. Parents emphasized the advantage of describing what
I II III IV V Total
the children actually can do instead of focusing on their limi-
tations. The distinction between capacity and performance Therapist 1
did not cause confusion for the families. Several therapists I 19 4 – – – 23
also emphasized that MACS was giving a structure to their II 2 56 6 – – 64
thinking, and that it was important to have one classification III – 5 22 1 – 28
for all kinds of CP. IV – 1 3 22 3 29
V – – – 1 23 24
The rationale for choosing a particular level instead of
Total 21 66 31 24 26 168
others was most often clear for both parents and therapists.
Although parents and therapists mainly agreed about the lev- aIntraclass correlation coefficient=0.97 (95% CI 0.96–0.98).

els there were some disagreements (reported below). These Numbers in bold represent total agreement.

Manual Ability Classification System for Children with CP Ann-Christin Eliasson et al. 551
between 8 and 12 years. The total agreement was 72%. There DISTRIBUTION OF MACS LEVELS AMONG SUBTYPES OF CP
was disagreement about one level for seven children, five of Children with all different subtypes of CP were included in
whom were rated on a higher level by the parents and two by the study (Table II, Fig. 1). The children with hemiplegia
the therapists. The disagreement was distributed across all were mainly seen in Level II but also in Levels I and III.
levels of MACS. Children with diplegia were found in Levels I to IV, whereas
children with dyskinetic CP and tetra- or quadriplegia were
apparent in Levels IV to V.
Table IV: Distribution of intraclass correlation coefficient
RELATION BETWEEN MACS AND GMFCS
(ICC) for different age groups
The correlation between GMFCS and MACS was 0.79 (p<0.05;
Age (y) n ICC (95% CI) Table V), with complete agreement in 49%. A large variation
occured at MACS Level II, where 40% of the children were
4–7 45 0.98 (0.97–0.99)
scored and where the agreement between GMFCS and MACS
8–12 90 0.96 (0.94–0.98)
was just 35%. Differences were also observed in the other levels
13–18 33 0.98 (0.96–0.99)
All: 4–18 168 0.97 (0.96–0.98) (Fig. 2).

CI, confidence interval. Discussion


MACS provides a new perspective for classifying manual ability
in children and adolescents with CP. As judged by parents of
Table V: Correlation between Manual Ability Classification children with CP and health professionals, MACS was based on
System (MACS) and Gross Motor Function Classification a valid construct. The classification looks at activities and gives
System (GMFCS) level ratingsa a single ‘level’ for the collaborative use of both hands when
MACS levels
handling objects in daily life. Parents and therapists perceived
I II III IV V Total the classification as a meaningful description of variations in
manual ability and felt confident when using MACS. Interrater
GMFCS levels reliability between the parent and therapist as well as between
I 15 29 2 – – 46 therapists was excellent; interrater reliability for different age
II 3 22 7 – – 32 groups was equally good. The results of testing with the
III 5 10 9 4 – 28 English version in Australia, although involving only a small
IV – 2 9 17 4 32
group of children, did not differ from the Swedish version,
V – – – 6 20 26
Totalb 23 63 27 27 24 164
indicating that linguistic or cultural differences do not affect
the outcome of MACS.
aSpearman’s correlation coefficient r=0.79. bGMFCS missing for During the process of constructing MACS several problems
four children. Numbers in bold represent total agreement. were identified. Commonly parents reported that the child

100 100

90 90

80 80

70 70

60 60
Per cent

Per cent

50 50

40 40

30 30

20 20

10 10

0 0
I II III IV V I II III IV V
MACS levels MACS levels

Diplegia Ataxia Dys CP GMFCS II GMFCS IV GMFCS V


Hemiplegia Tetra/quadri GMFCS I GMFCS III

Figure 1: Distribution of children (per cent of collected Figure 2: Distribution of children between levels of Manual
material) according to subtypes of cerebral paresis (CP). Ability Classification System (MACS) and Gross Motor
MACS, Manual Ability Classification System; dys, dystonia; Function Classification System (GMFCS).
tetra, tetraplegia; quadri, quadriplegia.

552 Developmental Medicine & Child Neurology 2006, 48: 549–554


could achieve a lot when they were motivated. Other com- some had just read the leaflet. The selection of children was
ments were that some children required frequent support also based on convenience, but it seems to reflect the pattern
from adults to initiate activity and to find strategies and solu- of children with CP described in epidemiological studies. The
tions to problems. This highlights the complexity of manual distribution of subtypes of CP agreed fairly well with earlier
ability during task performance. There is a motor–cognitive reports (Hagberg and Hagberg 1993). This heterogeneous
skill dimension in almost all tasks, with the cognitive part sample of raters and children demonstrates the applicability
involving ‘knowing what to do’ and the motor part involving of MACS and strengthens the generalizability of the results so
‘doing it correctly’ (Exner and Henderson 1995). Note, how- far as they reflect community realities.
ever, that when scoring MACS the reason for limited perfor- CP affects the whole body to a variable extent (Beckung and
mance (not handling objects) is not the investigated goal; rather Hagberg 2002). Limitation of activity associated with gross
MACS should be scored based on what the children actually motor function can be classified by GMFCS, whereas a reliable
do. This means, for example, when classifying children with and valid classification for limitation of activity related to manu-
hemiplegia, it is their ability to perform the task, indepen- al ability has not been available until now. Accordingly, the rela-
dent of whether they use one or two hands, independent of tion between gross and fine motor function is unclear. The
poor or good sensibility, that is classified. Gross motor func- high but not perfect correlation between MACS and GMFCS in
tion is also likely to influence the ability to handle objects, the present study indicates that MACS is built on a different
especially in situations that require the child to move to construct to GMFCS. In only half of the children was there
another location in the room or to bend to retrieve the exact agreement between MACS and GMFCS levels. This sug-
object. To minimize the influence of gross motor function on gests that gross and fine motor function in children with CP do
the scoring on MACS we limited the classification to situa- not neatly run in parallel, and can and should be independent-
tions in which the objects to be handled were within reach of ly classified. Hand function classified by Bimanual Fine Motor
the child. Function and GMFCS has earlier shown different development
A third problem identified and addressed was that of age. (Beckung and Hagberg 2002). The distribution of children
The MACS takes into consideration that children’s ability to belonging to different levels of GMFCS was fairly even (18–22%
handle objects increases with age. Hence, older children are for all levels) whereas 40% of the children belonged to Level II
expected to achieve more difficult tasks. However, this was in MACS. Noteworthy also was that the lowest proportion of
mainly discussed as a problem in the expert group; it did not children were classified in Level I. This distribution of MACS
apparently affect the classifications made by families and levels may highlight hidden problems of manual activity limita-
therapists. Data collection started in the 8 to 12 year age tion in children with CP and needs further investigation. MACS
group, when most of the basic ‘normal development’ would did not correspond to subtypes of CP; instead the levels of
usually have occurred. When starting to use MACS for other MACS were distributed across diagnoses in about the same way
ages the term ‘age-related’ was incorporated into the classifi- as the GMFCS (Gorter et al. 2004).
cation. Obviously, children handle different objects at 4 years Interestingly, several parents mentioned the importance of
compared with adolescent age (e.g. fragile or sharp objects), highlighting the children’s ability to use their hands. We also
but this seemed obvious to people when deciding about believe that MACS will enhance the communication among
MACS levels. Likewise the word ‘independence’ needs to be professionals and families in the same way as the GMFCS, i.e. to
used in an age-related perspective, because a young child determine the child’s needs, make management decisions, and
needs more help and supervision than an older one. compare and generalize results of intervention (Palisano et al.
So far we have not investigated the stability of the classifica- 1997). Our intention is that this first report on MACS will be fol-
tion over time, but our expectation is that most children will lowed by a series of studies designed to deepen the knowledge
stay at the same level. However, it is important to note that we of the construct of MACS as well as the knowledge on hand
expect that as children get older they will develop, learn to han- function in children with CP.
dle additional objects, and perform new age-related activities,
even if their relative functional characteristics are such that they DOI: 10.1017/S0012162206001162
handle objects as described in that MACS level. There will prob-
ably be some instability of levels in the youngest age groups as Accepted for publication 30th January 2006.
described earlier for the GMFCS (Wood and Rosenbaum
2000), and the interrater reliability may be lower for these The data in this paper won the Gayle G Arnold award for best
scientific paper at the 2005 AACPDM Annual Meeting.
young children (Palisano et al. 1997). Knowledge of typical
development of hand function in children with CP would have Acknowledgements
been useful when creating MACS but that has not, to our We acknowledge the parents and therapists without whose
knowledge, been described in the literature. A study collecting contribution this study would not have been accomplished. We also
thank those parents who participated in the validating process, and
longitudinal data with MACS is urgently needed. Christinne Imms and the Australian group of therapists for data
MACS is reliable, both between therapists and between collection of the English version. Financial support was granted
parents and therapists. Disagreements were distributed across from the Sven Jerring Fond, Josef och Linnea Karlssons Memorial
all levels, indicating that none of the levels was more difficult Foundation, and Stockholm Habilitation Service’s research unit.
to judge than others. In all but one case, differences between
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Appendix I: The Manual Ability Classfication System (MACS) leaflet (which can be downloaded in several languages from
www.macs.nu)

554 Developmental Medicine & Child Neurology 2006, 48: 549–554

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