Critically Appraised Paper
Weak evidence supports intensive, task-oriented, early
intervention with parent support for infants with, or at high
risk of, cerebral palsy
E. G. (2017). Effect of early intervention in infants at very
high risk of cerebral palsy: A systematic review. Develop-
mental Medicine and Child Neurology, 59(3), 246–258.
Objective: Review the evidence for early intervention with
infants with, or at risk of, cerebral palsy.
Design: Systematic review
Methods: PubMed, CINAHL, Embase, reference lists,
review articles and Developmental Medicine and Child Neurol-
ogy were searched to January 2016. Inclusion criteria: peer-
reviewed research of moderate to high methodological
quality; written in English; a prospective group design
with comparison group or period; mean age of the infants
at study entry less than 12 months corrected age and indi-
vidual infants younger than 18 months; and intervention
intended to inﬂuence neurodevelopmental outcome. Exclu-
sion was severe comorbidity unrelated to cerebral palsy.
Outcomes of interest were motor, cognitive and family and
parental wellbeing, and their relationship to brain lesion
and type and dose of intervention. Three reviewers inde-
pendently extracted data, completed quality appraisal and
reached consensus through discussion.
Main ﬁndings: Seven randomised trials (high (n=1) and
moderate (n=6) methodological quality) of 299 infants
(groups sizes 6–54) with brain lesion, “neurological
deviancy”, “high suspicion” of cerebral palsy or abnormal
general movements were included. Heterogeneity of inter-
vention type and intensity precluded pooling of results
and drawing ﬁrm conclusions. Weak evidence suggests
that developmental stimulation may improve cognitive
outcomes (two of three studies showed a positive effect)
and parent support improves family wellbeing (three out
of three studies). No convincing evidence that neurodevel-
opmental therapy (one of two studies suggested positive
effect) or developmental stimulation (one of four studies)
improves motor outcomes. Insufﬁcient evidence to draw
conclusions about the impact of sensory input (one study).
Insufﬁcient information was available to identify a relation-
ship between type of brain lesion and intervention effec-
tiveness. Several ﬂaws in the evidence base were noted
including small sample size, ill-deﬁned brain lesions, short
follow-up, inadequate comparison group description, and
adherence to intervention ﬁdelity not addressed.
Authors’ conclusions: Weak evidence in the included stud-
ies leads to the suggestion that intensive early intervention
and multifaceted intervention may be required to optimise
outcomes for infants with, or at risk of, cerebral palsy. Sub-
stantial high-quality research is required to advance the
Contact details of original author:MHadders-Algra,
CAPs Advisory Board Member
Spittle, A., Johnson, R. & Fetters, L. (2016). Effectiveness
of motor interventions in infants with cerebral palsy: A
systematic review. Developmental Medicine and Child
Neurology, 58(9), 900–909. doi:10.1111/dmcn.13105
Objective: To review the evidence for effectiveness of inter-
ventions to improve motor outcomes for children with
cerebral palsy aged 0–2years.
Design: Systematic review
Methods: Systematic searches of six databases (PubMed,
Embase, CINAHL, Cochrane, Web of Knowledge, PEDro)
were undertaken in 2014 and updated in 2015. Inclusion
criteria: all research designs with participants aged birth-
2 years diagnosed with, or at high risk of, cerebral palsy;
motor intervention implemented and motor outcomes
assessed. Motor intervention deﬁned as “therapeutic inter-
vention with motor development or skills as one primary
outcome” (p. 901). Studies were excluded if not written in
English or if the intervention was medical, pharmaceutical
or surgical. Reviewers independently selected studies for
inclusion. Interventions were classiﬁed by reviewer con-
sensus using the International Classiﬁcation of Functioning
Disability and Health framework. Risk of bias was assessed
©2017 Occupational Therapy Australia
Australian Occupational Therapy Journal (2017) 64, 423–425 doi: 10.1111/1440-1630.12426
using American Academy of Cerebral Palsy and Develop-
mental Medicine guidelines. Effect sizes assessed according
to Cohen’s dand quality and strength of evidence ranked
using the Grading of Recommendations Assessment,
Development and Evaluation system. Meta-analysis was
not possible due to heterogeneity.
Main ﬁndings: Thirty-six included papers reported 34 stud-
ies published 1984–2015; 10 randomised controlled trials
(RCT), 4 cohort, 10 single-subject design and 10 case stud-
ies or case series designs. Methodological quality collec-
tively assessed as low. The median sample size of the
RCTs and the cohort study with a control group was 26.
Conﬁrmed diagnosis at study exit ranged from 22% to
77%. Activity level outcomes were most commonly mea-
sured. Only 4 (of 10) RCTs reported statistically signiﬁcant
ﬁndings with “effect sizes ranging from 0.14 (small) to 0.75
(moderate-high)” (p. 906). The single-subject and case ser-
ies designs all showed positive ﬁndings. Duration and
intensity of intervention varied from 6-days/week for
6 weeks to monthly for 12 months. Interventions included
neurodevelopmental therapy, Vojta (passive application of
reciprocal limb movements), environmental enrichment
and constraint-induced movement therapy. Interventions
were complex; the most common component was parent
education. The commonly included intervention elements
in the two RCTs with largest effect sizes were child-
initiated movement, task speciﬁc training and environmen-
Authors’ conclusions: Low quality evidence was ‘weakly
intensive motor intervention with infants with, or at high
risk of, cerebral palsy consistent with contemporary prac-
tice around neuroplasticity and motor learning principles,
and positive outcomes identiﬁed in studies of older chil-
dren –that is, interventions which elicit active movement
in task-oriented activities in a high intensity program.
Contact details of original author: Catherine Morgan,
CAPs Advisory Board Member
Premature birth, brain lesions and abnormalities place
infants at high risk of cerebral palsy (CP). Recently, the
increased use of magnetic resonance imaging (MRI), com-
bined with prognostic tools such as the General Move-
ments Assessment (GMs) and the Hammersmith Infant
Neurological Examination (HINE), has made detection of
infants with, or at risk of, CP possible during the ﬁrst half-
year of life (McIntyre, Morgan, Walker & Novak, 2011;
Romeo, Ricci, Brogna & Mercuri, 2016). Early detection is
crucial, as animal studies show that the timing of interven-
tion is the most critical factor for maximising treatment
outcomes (Friel, Chakrabarty, Kuo & Martin, 2012). Further
to this, is evidence that most gross motor and bimanual
performance potential in children with CP is experienced
in the ﬁrst 2 years of development (Nordstrand, Eliasson &
Holmefur, 2016; Rosenbaum et al., 2002). Early detection
should trigger immediate referral to infant speciﬁc inter-
vention programs. But what is the evidence for these pro-
grams? Two recent papers aimed to systematically review
the evidence for early intervention for infants with, or at
high risk of CP (Hadders-Algra, Boxum, Hielkema &
Hamer, 2017; Morgan et al., 2016).
These two reviews ﬁll an important knowledge gap by
focussing speciﬁcally on children younger than 2 years of
age when intervention can drive neural circuit develop-
ment during the most dynamic phase of plasticity. Had-
ders-Algra et al. (2017) made special effort to highlight
differences in methodology compared to the earlier review
by Morgan et al. (2016). These include a focus on children
<1 years old, methods used for rating the quality of
included studies, inclusion of family outcomes, the effect of
disease state on intervention response and the dose and
type of intervention provided. These differences led to
inclusion of signiﬁcantly fewer studies in Hadders-Algra
et al. (n=7) compared with Morgan et al. (2016) (n=34).
Despite this, outcomes for each review are similar. Evi-
dence for intervention remains weak. Studies are mostly
small with low methodological quality. There is consider-
able heterogeneity across studies including participants,
interventions and outcome measures. As a result, meta-
analysis of data for pooled effect, the ability to make deﬁni-
tive conclusions on the effect of disease state on treatment
response, or on the dose and type of intervention provided,
are not possible. Both reviews concluded that trends in the
data support intervention models based on motor learning
theory (e.g., child-initiated movement), environmental
enrichment and parent education underpinned by ecologi-
cal and family-centred frameworks.
One point of difference between the reviews relates to
neurodevelopmental therapy (NDT). Hadders-Algra et al.
(2017) suggested that minimal hands on postural support
techniques may be useful for infants with CP. Morgan et al.
(2016) suggested that trends in data do not support neuro-
maturational approaches such as NDT. Despite these dif-
ferences, we agree with comments by Morgan et al. that
whatever the intervention type, a comprehensive descrip-
tion of the key ingredients is essential, especially where
there is a diversity of interpretation (Mayston, 2016).
While evidence remains weak, the consistent outcomes
and emerging evidence described by both reviews, have
important implications for occupational therapists. Work-
ing with infants with CP means focussing on occupation,
rather than using neuro-maturational approaches that seek
to normalise movement. The core philosophies of our occu-
pation-based profession are entirely consistent with the
key ingredients of evidence-based models of early inter-
vention proposed in these reviews including child-initiated
movement, task speciﬁcity, environmental modiﬁcation
and parent support and education.
©2017 Occupational Therapy Australia
424 CRITICALLY APPRAISED PAPER
Paediatric occupational therapists should abandon gen-
eralised, low intensity models of intervention. Alongside a
speciﬁc, targeted, therapist-guided program, interventions
should include a goal-directed home program as a prag-
matic solution to achieving optimal intensity and to ensure
generalisation of skills (Novak, Cusick & Lannin, 2009).
Evidence-based occupational therapy with infants at high
risk of CP includes collaborating with medical profession-
als in the early detection of CP by using diagnostic tools
such as the GMs and HINE; conﬁdent application of evi-
dence-based early intervention models including COPing
with and CAring for Infants with Special Needs (COPCA)
(Dirks, Blauw-Hospers, Hulshof & Hadders-Algra, 2011),
Goals, Activity and Motor Enrichment (GAME) (Morgan,
Novak, Dale, Guzzetta & Badawi, 2014), baby constraint-
induced movement therapy (Eliasson, Sjostrand, Ek, Krum-
linde-Sundholm & Tedroff, 2014) and bimanual therapy
(Hoare & Greaves, 2017). New measures such as the Hand
Assessment for Infants (HAI) (Krumlinde-Sundholm et al.,
2015) and the Mini-Assisting Hand Assessment (Greaves,
Imms, Dodd & Krumlinde-Sundholm, 2013) are available
and enable targeting of upper limb interventions to the just
right challenge, and objective evaluation of outcomes of
In Australia, the introduction of the National Disability
Insurance Scheme provides great opportunity for families
of children with CP to access occupational therapy. For
children older than 2 years of age with CP, there are effec-
tive interventions supported by strong evidence (Novak
et al., 2013). Evidence-based interventions implemented
with strong treatment ﬁdelity in clinical practice, including
for younger infants, are required. Current evidence-based
interventions are not transdisciplinary approaches, but are
highly speciﬁc and targeted interventions provided by
skilled therapists who have gained experience in, and
knowledge about, working with very young infants.
Organisations need to ensure specialist training opportuni-
ties for staff, adapt services based on the evolving evi-
dence, and foster and recognise clinical expertise.
Department of Paediatrics Monash University and School of
La Trobe University,
Melbourne, Victoria, Australia
Occupational Therapy Department, The Royal Children’s
Melbourne, Victoria, Australia
Dirks, T., Blauw-Hospers, C. H., Hulshof, L. J. & Hadders-Algra, M.
(2011). Differences between the family-centered “COPCA” program
and traditional infant physical therapy based on neurodevelopmen-
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behavior during an early critical period to restore skilled limb move-
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Effect of early intervention in infants at very high risk of cerebral
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©2017 Occupational Therapy Australia
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