Over the past 4 years the team from the Cerebral Palsy Alliance Research Institute, University of Notre Dame in Australia have published a series of articles setting out the case for a principled approach to intervention for infants who are at high risk for cerebral palsy.
As a first step they developed an approach to intervention on best available evidence of interventions that work in older children and that aim to harness the neuroplasticity mechanisms at work in the developing brain. (Morgan 2015). The intervention is based on the principles of active motor learning, family centred care, parent coaching and environmental enrichment. Intervention is customised to parent goals and enrichment style and the child’s motor ability.
They named the approach GAME (Goals, Activity and Motor Enrichment) (Morgan et al., 2014).
The Three Components of the GAME approach
Goal oriented intensive motor training
Parent-identified goal areas are targeted for practice during the therapy session and and in a home program (HP).
During practice of motor task, the therapist or parent scaffolds all motor tasks, so that the infant could always actively complete at least a part of the task. As performance improved, the challenge is increased by altering the task or environment to a new and appropriate level of difficulty.
Manual assistance is provided by the therapist and parent only when necessary for safety or to give the infant the “idea” of the movement. Manual assistance was reduced or withdrawn as soon as the infant demonstrated self-initiated progress with the task; ensuring self-generated motor activity was the focus of all practice.
Once a motor skill is learned, variability of practice is introduced to increase the complexity and generalizability of the skill.
Early weightbearing and sit to stand from the parents’ lap are part of each HP even if standing was not identified as a specific goal.
Rehabilitation research in older children and adults with brain injuries suggest that functional weight bearing exercises can both improve motor control and provide strength training . Given that the expected impairments of CP include weakness and reduced selective motor control, early activation of muscles of the lower limb using both concentric and eccentric exercise could enhance the development of upright mobility.
Similarly, practice of reaching and grasping a variety of objects is a standard part of motor training for all infants in order to expose the infants who are expected to be delayed, to a variety of objects to advance grasp and reach behaviours .
Environmental enrichment (EE) is broadly defined as an environment that enhances and promotes social, motor and sensory stimulation. EE has been proven to enhance neuroplasticity and promote memory and motor function in animal studies, but the effect in humans is less well understood. (van Praag H et al, 2001, Nithianantharajah J, et al 2006)
Parents are encouraged and assisted to set up motor enriched play environments to promote child self-generated movements, exploration and task success.
This includes instruction in careful toy selection “matched” to the desired motor task, plus physical set up of areas for practicing and repeating activities related to the identified goal areas, weightbearing, and reaching and grasping tasks. Conventional baby equipment (e.g. highchairs, toys) already purchased by the family was used wherever possible.
The whole environment for motor learning is taken into account and therefore intervention also includes:
- Evidence-based early learning stimulation and role modeling to enhance cognitive and language development (e.g. reading books to children, limiting passive television watching);
- Optimising sleep hygiene, for example assisting with implementing sleep routines;
- Feeding interventions (e.g. anti-reflux medications) to ensure adequate caloric nutrition and pain-free backdrops for learning.
- The importance of variable daily experiences for infants was deliberately addressed and support given when parents articulated difficulty leaving the house.
- Siblings and extended family members were also actively encouraged to take part in the HP and therapy sessions to promote: family knowledge; family acceptance; family wellbeing; repetition of learning opportunities; and provide a natural source of varied social interaction for the infant.(Morgan et al 2015)
Parents play an active role in selecting goals and carrying out a home program.
Parents are provided with the knowledge and skills to promote and support their baby's self initiated and generated actions, exploration and engagement with motor tasks.
- Parents are coached to identify their child’s voluntary attempts to move and self-regulate, plus understand the usual trajectory of emergent motor skills and how to stimulate progress.
- Parents are trained in simple motor task analysis and coached in appropriate strategies to enhance their child’s development both at a specific goal level and in general early learning and development principles.
- Parents are taught to optimize the best use of their infants’ awake time and the naturally occurring opportunities for learning. Learning optimization included both parent-directed and structured practice of desired motor tasks, where the parent role was integral to the child’s learning (e.g. creating repetitions) and constructing opportunities for independent play (e.g. playing alone with motor enriching toys set up for the child).
- Parents are encouraged to both observe the therapist eliciting a motor behaviour from the baby and to attempt it themselves. Specific feedback was given to parents to enable them to tease out why some attempts were successful for the baby and others weren’t.
- As new motor skills emerged parents are coached in strategies to increase the challenge of the task; for example remove support or introduce a more complex toy.
- The importance of allowing trial and error during practice is discussed and parents are encouraged to devise their own activities to enhance goal attainment.
The GAME RCT
The effectiveness of the GAME approach for infants at high risk of cerebral has now been tested in a single blind randomized controlled trial infants at high risk of cerebral. (Morgan et al 2016).
The GAME intervention was compared to standard care. Primary outcome was motor skills on the Peabody Developmental Motor Scales-2 (PDMS-2). Secondary outcomes included Canadian Occupational Performance Measure (COPM), Bayley Scales of Infant and Toddler Development (BSID-III) and Gross Motor Function Measure-66 (GMFM-66). Outcome assessors were masked to group allocation and data analyzed with multiple regression.
All n=30 infants enrolled received the assigned intervention until 16 weeks post enrollment. The GAME intervention was offered via home visits at least fortnightly until the infants first birthday (CA). All SC providers used a combination of centre based visits and home programs. Some infants in SC received home visits as well as clinic based appointments. Intervention approaches varied and was typically “eclectic” borrowing from a variety of therapy intervention paradigms including neurodevelopmental therapy and sensory integration.
At 12 months of age, n=26 completed assessments. Significant between group differences were found in raw scores on the PDMS-2 in favour of GAME (B=20.71, 95%CI 1.66-39.76, p=0. 03) and at 12 months on the total motor quotient (B=8.29, 95%CI 0.13-16.45,p =0.05). Significant between group differences favored GAME participants at 12 months on the cognitive scale of the BSID-III and satisfaction scores on the COPM.
The authors conclude that the study suggests that 6‐9 months of GAME, a clinically feasible intervention, is more effective than standard care to advance the motor function of infants at high risk of CP. As expected, infants with milder brain injury responded better to intervention than those with severe brain injury.
This is evidenced both in the norm referenced measure (PDMS-2) as well as the criterion referenced GMFM-66.
Dose of intervention
One aspect of the outcomes is worth highlighting: dose of intervention has been highlighted as an important component affecting change in function over time.
"Previous CP trials (Gordon, 2011; Sakzewski, Ziviani, & Boyd, 2013) in older children have shown that high-dose motor-learning based therapy leads to better results than low-dose motor-learning therapy, causing experts to hypothesise that many therapy interventions studied to date might be under-dosed. Interestingly, in older children, when two effective motor-learning interventions are compared head-to-head at the same high-dose, similar patient outcomes result. Recent systematic reviews have therefore identified that in addition to type of therapy mattering (effective versus ineffective) also the intensity of the therapy is important for treatment success (Myrhaug, Østensjø, Larun, Odgaard-Jensen, & Jahnsen, 2014).
In our GAME study, the difference in dose of intervention was not statistically significant between the groups, due to the large variation within each group, however the median values clearly demonstrate that most GAME participants received a higher number of therapy sessions and most GAME parents engaged in more home practice.
Correlations between dose and PDMS-2 scores at the primary end-point were low, but this may also be an artefact of small sample sizes, which was further exacerbated by the volume of missing logbook data. It is therefore still possible that both the higher dose of intervention as well as characteristics of GAME contributed to the gains achieved in the GAME group but more research is needed." (Morgan et al 2016)
A.M. Gordon (2011) To constrain or not to constrain: and other stories of intensive upper extremity training for children with unilateral cerebral palsy. Developmental Medicine & Child Neurology, 53 (s4) 56–61
Morgan C, Novak I, Dale RC, Guzzetta A, Badawi N. Singl doi: 10.1016/j.ridd.2016.04.005.e blind randomised controlled trial of GAME (Goals - Activity - Motor Enrichment) in infants at high risk of cerebral palsy. Res Dev Disabil. 2016 Aug;55:256-67. https://www.ncbi.nlm.nih.gov/pubmed/27164480
Morgan C, Novak I, Dale RC, Guzzetta A, Badawi N. GAME (Goals - Activity - Motor Enrichment): protocol of a single blind randomised controlled trial of motor training, parent education and environmental enrichment for infants at high risk of cerebral palsy. BMC Neurol. 2014 Oct 7;14:203. https://www.ncbi.nlm.nih.gov/pubmed/27027732
Morgan C, Novak I, Dale RC, Badawi N. Optimising motor learning in infants at high risk of cerebral palsy: a pilot study. BMC Pediatr. 2015 Apr 1;15:30. http://bmcpediatr.biomedcentral.com/articles/10.1186/s12887-015-0347-2
Morgan C, Novak I, Badawi N. Enriched environments and motor outcomes in cerebral palsy: systematic review and meta-analysis. Pediatrics. 2013 Sep;132(3):e735-46. doi: 10.1542/peds.2012-3985 .http://pediatrics.aappublications.org/content/132/3/e735.long
Sakzewski L, Ziviani J, Boyd RN. Efficacy of upper limb therapies for unilateral cerebral palsy: a meta-analysis. Pediatrics. 2014 Jan;133(1):e175-204. doi: 10.1542/peds.2013-0675. Epub 2013 Dec 23. Review. PubMed PMID: 24366991.
Tinderholt Myrhaug, H., Østensjø, S., Larun, L., Odgaard-Jensen, J., & Jahnsen, R. (2014). Intensive training of motor function and functional skills among young children with cerebral palsy: a systematic review and meta-analysis. BMC Pediatrics, 14, 292. http://doi.org/10.1186/s12887-014-0292-5
Nithianantharajah J, Hannan AJ. Enriched environments, experience-dependent plasticity and disorders of the nervous system. Nat Rev Neurosci. 2006;7(9):697–709pmid:16924259
van Praag H, Kempermann G, Gage FH . Neural consequences of environmental enrichment. Nat Rev Neurosci. 2000;1(3):191–198 pmid:11257907