Implementation of COPCA
Akhbari-Ziegler, Schirin
DOI:
10.33612/diss.132156787
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Publication date: 2020
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Akhbari-Ziegler, S. (2020). Implementation of COPCA: A family-centred early intervention programme in infant physiotherapy. University of Groningen. https://doi.org/10.33612/diss.132156787
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General discussion
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The aim of this thesis was to provide insight in the application and effect of physiotherapy for infants with an elevated risk for developmental motor disorders in Switzerland. In particular, the focus was on the implementation process of the family-centred early intervention programme “COPing with and CAring for infants with special needs” (COPCA). To this end, I first examined the psychometric properties of the second version of the Groningen Observer Protocol (GOP 2.0) (chapter 2). The next step was to analyse the contents of typical infant physiotherapy (TIP) in Switzerland. Neuro-Developmental Treatment (NDT) is the most widely used approach in physiotherapeutic early intervention. To get an idea to what extent actions belonging to NDT are incorporated into infant physiotherapy in Switzerland or whether more recent approaches are also applied we conducted an observational study (chapter 3). From this study we learned, that educational actions towards the caregivers in TIP in Switzerland were present in only about one fifth of the sessions’ treatment time. When present, they consisted of caregiver training. This largely differs from COPCA in which the main strategy regarding educational actions towards caregivers consists of coaching. Due to this difference we became aware that it was important to clarify what coaching in COPCA means. We described the theoretical background of coaching in COPCA and the practical consequences of this background for coaching of families with an infant with a developmental motor disorder (chapter 4). During the literature study on the perspective of coaching in rehabilitation, we were confronted with the manifold ambiguities on the concept of coaching. This makes the concept of coaching difficult to understand. Hence, we wrote a narrative review to point out the ambiguities and the challenges in the incorporation of coaching in daily practice. We concluded with the provision of suggestions for successful implementation (chapter 5). To implement COPCA in Switzerland, where COPCA until recently was largely unknown, it was necessary to educate paediatric physiotherapists in COPCA. From the study described in chapter 3 we learned that becoming a COPCA coach demands from conventionally trained paediatric physiotherapist changes in their professional behaviour. Therefore, we evaluated how well the physiotherapists were able to change their physiotherapeutic actions and whether this was similar for all therapeutic actions (chapter 6). The shift from child-centred to family-centred practice and from instruction of families to the application of coaching demands also changes of the role of the family members. They have to give up the expectation that the physiotherapist is treating the infant and they have to play a part as active partner in the intervention process. We did not know, how caregivers cope with the role given to them in COPCA, as caregivers’ experiences with COPCA had not been examined until then. Through a qualitative study, we obtained insight in caregivers’ experiences with COPCA, which assists the implementation of COPCA in daily practice (chapter 7). To explore the effects of COPCA and TIP on motor,
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201 cognitive and family outcome in infants born preterm and their familes, we conducted a pilot randomized controlled trial (RCT) combined with process evaluation. The study suggested that COPCA and especially caregiver coaching in COPCA were associated with better motor outcome in very preterm infants than TIP (chapter 8).
In the following sections, I first address assessment tools used in the studies. Next I discuss the main findings of this thesis, the methodological considerations, the clinical implications and suggestions for further research. The discussion closes with concluding remarks, summarising the main implications of the thesis.
REFLECTION ABOUT ASSESSMENT TOOLS USED IN
THE THESIS
Groningen Observation Protocol version 2.0 (GOP 2.0): A tool to
quantify the contents of infant physiotherapy
Our study on the psychometric properties of the second version of GOP 2.0 demonstrated that it is a reliable tool to quantify contents of infant physiotherapy in Switzerland. The completeness of the instrument indicated that virtually all actions used by physiotherapists in Switzerland in the study could be classified. These results are in line with the results of a study of Hielkema et al.,1 which demonstrated that the
GOP 2.0 is reliable to assess the contents of infant physiotherapy in the Netherlands. The applicability of GOP 2.0 in both countries suggests that it can be used also in other health care settings. There are various reasons why an instrument like the GOP 2.0 is important and necessary. The quality of descriptions of the intervention elements in studies is notably poor.2 A detailed description of the intervention is a requirement
for a reliable implementation of that intervention and for the replication of research findings.2 Guidelines for reporting parallel group randomised trials like the Consolidated
Standards of Reporting Trials (CONSORT 2020)3 demand the reporting of details of
intervention elements and a clear comparison between the intervention elements of the parallel groups. Second, the quantification of the contents of interventions allows an in-depth understanding of the characteristics of an intervention. The black box that complex intervention programmes represent gets at least opened.4 Elements
of the intervention become distinguishable and quantifiable. This allows for process evaluation, the evaluation of associations between particular intervention elements and defined outcomes.5 Knowledge on the effectiveness and ineffectiveness of specific
intervention elements is essential for evidence-based practice. In this thesis, GOP 2.0 was used as follows: (1) to quantify contents of infant physiotherapy (chapter 3), (2) to
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evaluate changes of contents over time (chapter 6), to compare different intervention programmes (chapter 8), and to conduct a process evaluation (chapter 8).
The Infant Motor Profile (IMP)
In the pilot RCT (chapter 8) the Infant Motor Profile (IMP) was the primary outcome instrument.6 Before addressing the psychometric properties of the IMP, I first summarise
the IMP’s construct.
The IMP is a modern video-based instrument to assess spontaneous motor behaviour in infants aged 3 to 18 months. The IMP has five domains: variation, adaptability, symmetry, fluency and performance.6,7 Variation and adaptability are novel
domains based on the neuronal group selection theory (NGST).8,9 According to the
NGST, motor development has two phases of variability, the primary and the secondary variability. In the phase of primary variability, infants have manifold movement variations at their disposal, but the movements are adapted to the environment only to a marginal extent.8 In the phase of secondary variability, the infant gradually develops
the ability to adapt his/her movements to the environment, in a task specific manner. The start of the secondary phase is at function-specific ages, for instance, it occurs earlier in reaching and grasping than in walking. The ability to select from the given movement repertoire the best fitting strategy for a specific task (adaptability) develops through active trial-and-error experiences and the associated sensory information. The motor behaviour in the second phase is characterised by variation and adaptability. In infants with a brain lesion, variation as well as adaptability are limited. The lesion reduces the motor repertoire of the infant and hampers the ability to select the best fitting strategy from the given movement repertoire. The limited adaptability is caused by deficits in self-produced sensorimotor experiences as a consequence of the limited repertoire and by impairments in using sensory information.9 Limited variation is a
strong indicator for a structural brain lesion. Limited adaptability may be an expression of a lesion of the brain but more often it is a sign of a minor dysfunction of the brain due to changes in the monoaminergic systems.9 The three other domains of the IMP
represent well-known phenomena of motor development. The domain symmetry may be regarded as a specific variant of the domain variation; it examines the occurrence of asymmetric movements. Stereotyped asymmetric movements are an indicator for a dysfunction of the brain and may represent the first sign of a unilateral cerebral palsy (CP). The domain fluency evaluates the presence or absence of fluent movements. Non-fluent movements, like stiff, jerky or tremulous movements, are an expression of a limited capacity to modulate acceleration and deceleration subtly. Non-fluent movements may arise in humans of any age and are a sign of a minimal dysfunction of the nervous system.9 The first four domains address the quality of motor behaviour.
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203 The fifth domain, the performance domain, assesses motor behaviour according to the achievement of motor milestones and represents an important aspect of the infant’s activities of daily living. The IMP consists of 80 items (25 variation, 15 adaptability, 10 symmetry, 7 fluency and 23 performance), which are examined in different positions (supine, prone, sitting, standing and walking) and during reaching, grasping and manipulation.6 At each age - due to age dependent changes in motor behaviour - a
specific selection of items is examined, dependent on the functional level of motor development. For example, in an infant of 3 months, the majority of items in sitting and standing do not require specific testing; they get a predefined score. In an infant of 18 months, the items in supine are not assessed as they are not appropriate anymore.6 The
resulting score for each IMP domain is a percentage, expressing the relation between the score of the infant and the maximum reachable score of 100%. The total IMP score is calculated on the basis of the domain scores. In infants older than 6 month the scores of the five domains are summed up, in younger infants the domain adaptability is not included due to the limited validity of this subscale in these infants.
The psychometric properties of the IMP are good. Multiple studies showed that the IMP is a reliable instrument.6,7,10 These studies also showed the good construct-
and concurrent validity. Low IMP scores are associated with perinatal risk factors like preterm birth and lesions of the brain. For clinical practise, it is even more important that the IMP has a promising predictive validity for CP. Low total IMP scores and low scores in the domain variation and performance are associated with later diagnosis of CP and a lower IQ score at 4 years.11,12 Moreover, studies showed that the IMP is a
responsive instrument to evaluate the effectiveness of early intervention and is more responsive than the Alberta Infant Motor Scales, a commonly used instrument in paediatric physiotherapy.13,14 The responsiveness of the IMP was the reason to select
the IMP as our primary outcome parameter in the pilot RCT.
Family related outcomes
Family-centred practices, which have become practice-of-choice in early intervention,15
indicate among others things, that the focus of intervention is the entire family. The family is regarded as a unit. The intervention does not only target the child, but all family members are included as active partners in the intervention process. COPCA, as a family-centred approach, aims to encourage the family’s capacities to stimulate the infant’s development during daily care and to support the family in the process of decision making regarding functional activity and participation in daily living.16 This
indicates that in order to evaluate the effect of a family-centred physiotherapeutic early intervention approach not only child-related, but also family-related outcomes have to be evaluated. To meet this requirement, we applied in our pilot RCT (chapter 8)
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the Family subscale of the Family Empowerment Scale (FES)17 and the German version
of the Measurement of Processes of Care (MPOC),18,19 a parental questionnaire to
quantify the extent to which the caregivers experience family-centeredness in the care of their child. We hypothesised to find positive effects of COPCA with these outcome measures, as COPCA is family-centred and aims to empower the family. Unfortunately, we only gradually understood that the family related measurements applied in this study – though they were the best available - were not really meaningful. The FES scores were similar and high at all measurement points in both groups, and did not change during the course of the study. This indicates that in both groups no beneficial effect on empowerment of families could be demonstrated with the FES. This result contrasts with the findings in the qualitative study about caregivers’ experiences with COPCA (chapter 7). The mothers participating in the latter study reported that they learned to support the infant autonomously and gained confidence during the intervention process. According to the description of King et al.20, an empowered
mind-set includes confidence, self-efficacy and self-determination. This is what the mothers in our qualitative study reported: they experienced that they learned to challenge the infant adequately and to create an enriched environment at home, to set reachable goals and that they gained certainty in handling the infant and situation better, which allows for the conclusion that their empowerment grew during the course of 6 months of COPCA intervention. An explanation for the inconsistency between the qualitative study and the pilot RCT may be that the FES measures empowerment as a current state not related to an early intervention process and that the FES was originally developed for families with children with emotional disabilities, which represents a quite different context. It may also imply that the questions of the FES do not easily catch the feelings of empowerment in families of young infants in need of intervention. This indicates, that a sensitive instrument measuring family empowerment related to changes in context of early intervention is lacking.
We applied the MPOC, because it was the only outcome measure available evaluating caregivers’ experience with family-centred approaches. However, the MPOC could not be analysed in our study due to many missing data. Two therapists (one of each group) reported that one family that they had guided could not answer the MPOC-questions at all. The mothers of these families had a low educational level. This may indicate that a certain educational level is required to fill out the MPOC. Another observation was that many caregivers, including those with a higher educational level, responded to a high proportion of questions ‘not applicable’. This could be explained by the design of the MPOC, as it was developed to measure experiences with health care in families of children with already diagnosed disabilities, receiving care in a multidisciplinary rehabilitation treatment center. This implies that MPOC’s
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205 intended context differs from the one in our pilot RCT. The participants in our study were caregivers of preterm infants at moderate to high risk of developmental disorders without a developmental diagnosis at the beginning of the intervention process. In such a situation, caregivers usually sail between hope and anxiety. The preterm infants and their families were allocated to paediatric physiotherapy by a neonatologist shortly after discharge from the hospital. All families received individual face-to-face interventions and had no other therapies. This means, they did not receive services from a multidisciplinary rehabilitation treatment center. As a consequence, all questions related to a rehabilitation center context or a manifest disability (e.g. provision of written information, support to cope with disability, multidisciplinary agreements) were often answered with ‘not applicable’. This includes almost all questions of the scale scores ‘Providing General Information’ and ‘Providing Specific Information about the child’, six of the 16 questions of the scale score ‘Enabling & Partnership’ (8,12,16,17,22,25), seven of the 17 questions of the scale score ‘Coordinated and Comprehensive Care’ (4,5,6,7,21,37,45) and one of the nine questions of the scale score ‘Respectful & Supportive Care’ (47). A similar experience was reported by Blauw-Hospers.21 Our
findings may correspond to the recent meta-analysis on the MPOC,22 that concluded
that in research the MPOC is generally used with success, that is, in children of two years and older after a diagnosis has been established. This indicates, that a reliable, applicable and sensitive instrument measuring perception of family-centeredness in families with children younger than two years, who receive exclusively a face-to-face intervention, is missing and is urgently needed.
For the development of instruments that are capable to measure meaningful family outcomes, I suggest the following. Including parents as active partners in the intervention process means strengthening their capacity to support their child’s development and increasing their engagement. Therefore a measurement of the parents’ capacity and engagement could be a meaningful outcome in early intervention. This idea is supported by the systematic review of Ward et al.,23 which highlighted
the importance to measure changes in parent capacity and self-efficacy as a result of the intervention process. However, the review also concluded that such an outcome measure is currently lacking. King et al.20 investigated family experiences of
Solution-Focused Coaching, a family-centred coaching approach in paediatric rehabilitation,24
in a qualitative study. The study summarised themes concerning family experiences in a conceptual framework. This framework illustrated associations between Solution-Focused Coaching and enhanced family capacity and participation, the amount of engagement and an empowered mind-set. Therefore, the study of King et al.20 supported
the notion that besides empowerment, also capacity and engagement are meaningful aspects of family outcome. In the perspective paper on coaching in rehabilitation
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(chapter 4), we described that COPCA coaches support the entire family in such a way that participation and quality of life is maintained and/or promoted. For this reason, meaningful family outcomes should include also these two aspects. The outcome of the RCT of Hielkema et al.25 which compared family’s quality of life of families with an infant
at very high risk of CP after receiving COPCA or TIP, supports this notion. The family’s quality of life in the COPCA group increased over time, whereas it remained similar in the TIP group. Also, the narrative review and the qualitative study (chapters 5 and 7) generated suggestions for meaningful family outcomes. They could include the quality of parent-infant interaction (e.g. responsiveness towards the infant, confidence in the infants’ development), the degree to which the family was addressed as an entire unit in the intervention process, and the degree to which the intervention was adapted to daily requirements. These ideas may form the basis for the development of meaningful family-related outcome instruments for family-centred intervention approaches in early intervention and paediatric rehabilitation.
INFANT PHYSIOTHERAPY IN SWITZERLAND: COPCA
AND TIP
Contents of typical infant physiotherapy in Switzerland
The contents of TIP in Switzerland, monitored between 2013 and 2014 (chapter 3), were very heterogeneous and primarily provided in a therapeutic or clinical setting. NDT-related actions like hands-on facilitation techniques were the most applied neuromotor actions. Educational actions towards the caregivers were applied only in around one fifth of the treatment time and consisted of caregiver training. Caregiver coaching was not applied. Specific communications with the caregiver like information exchange, instructions or provision of feedback were also only applied in around one fifth of the treatment time. Most communication time was spent on communication with the infant, a typical characteristic of child-centred approaches. The contents of infant physiotherapy did not differ for younger or older infants except for positions. Older infants were more often treated in so-called higher positions like sitting or standing. The study indicated, that that early physiotherapy intervention as practiced by the physiotherapists in Switzerland in 2013 and 2014 followed evidence-based recommendations only to a limited extent. Intervention components supported by current evidence of effectiveness, including family-centred practice, 15, 26,27 family
coaching and challenging the infant to self-produced motor behaviour and provision of trial-and-error experiences,4,13 were infrequently applied.
This study provided insight in the application of infant physiotherapy before the beginning of the implementation process of COPCA in Switzerland and the circumstance
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207 that recent evidence-based approaches were applied only to a limited extent. Early physiotherapy intervention as practiced in 2013 and 2014 represents the result of the continuously changing process of the NDT approach over time, resulting in heterogeneity and elective application. The data showed that therapists often use specific handling with the idea that it allows the infant to learn new motor actions.
This knowledge helped us to become aware of what physiotherapists in Switzerland would need to become a COPCA coach, e.g. knowledge on evidence-based recommmendations in infant physiotherapy including family-centred practice with parental involvement, family coaching and neuromotor actions like offering the infant ample opportunities to explore own activities and to learn from trial-and-error.
Theoretical background of coaching in COPCA and challenges using
coaching approaches
In the perspective paper on coaching in rehabilitation (chapter 4), we presented and critically discussed the theoretical background and the resulting practical consequences of COPCA’s specific coaching approach. The perspective consists of four sections: (1) the meta-model of COPCA’s coaching including our concept of the human being, our ethical attitude and our ideals, (2) theories underlying COPCA: family-centred practice,15, 26,27 the neuronal group selection theory,9,28,29 the transactional model of
development,30 coaching theories (the goal-oriented coaching approach),31 family
system theory,32 communication theory33 and humanistic psychology,34,35 (3) translation
into practice consisting of aims of coaching in COPCA, the setting, understanding of intervention, focus of attention, communication, relationship and role allocation, and (4) the praxeology section, describing the required knowledge of a COPCA coach and coaching skills. The paper concluded that COPCA’s coaching approach is demanding for the paediatric physiotherapist as well as for the family members. In order to apply the coaching skills, the conventionally trained physiotherapist has to acquire new knowledge and skills and to change attitudes and habits. In addition, family members need to learn to cope with an active role in the intervention process.
Coaching is applied increasingly often in early childhood intervention. However, a variety of coaching approaches with different underlying theories, definitions and coaching strategies exist.31 In addition, the role of a coach can be understood in manifold
ways. Many studies using coaching as intervention strategy do not define coaching, and a theoretical underpinning of the coaching approach is generally missing.23,36,37 A definition
of the coaching approach in COPCA, the aims of coaching in COPCA and some coaching strategies were already defined in the studies of Dirks et al.16 and Blauw-Hospers et al.5
Yet, the perspective paper summarised the COPCA approach systematically. It started
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with a comprehensive review of the theoretical framework, which formed the basis for COPCA’s practical application in the families. Through the study described in chapter 3, we became aware that it was important to clarify what coaching in COPCA means. This was deemed essential for a successful implementation of COPCA in Switzerland, as therapists in Switzerland were not used to apply caregiver coaching. The perspective paper is also a useful instrument for candidate COPCA coaches to acquire knowledge on coaching in COPCA. Moreover, the ideas of the perspective functioned as a reference to describe the intervention of the COPCA group in the RCT (chapter 8).
In the narrative review (chapter 5) we especially discussed the inconsistencies in the terminology of coaching used in the literature of early intervention programmes using coaching. This inconsistency represents an ambiguity hampering the incorporation of coaching in relationship-directed forms of interventions based on principles of family-centred practice (RD-FCI).38-42 We described the key components of ‘Parent training’
and ‘Parent coaching’, which are both labelled as ‘coaching’ in the literature. However, literature also suggests that ‘Parent training’ and ‘Parent coaching’ are two different approaches, with different attitudes and goals.43,44 Therefore we suggested to use the
term ‘coaching’ exclusively for coaching provided in RD-FCI. We identified three main challenges relating to the implementation of coaching approaches in practice: (1) to change the professional role from the child’s therapist to coach of the family,44,45 (2) to
require new knowledge in particular on coaching skills and adult learning,40,41,45,46 and
(3) to translate knowledge and beliefs into practice.42 To cope with these challenges
professional education and ongoing support in the practical implementation and ample time are indispensable.39,40,42,46,47 The review provides suggestions about the
duration, the contents and educational methods of professional education provided by the literature39,40, 41,42, 46,47 to promote successful implementation of coaching in
early childhood interventions. The implementation of coaching in RD-FCI may also by hindered by barriers, e.g. beliefs and attitudes that contrast with those in RD-FCI, and strong habits. Providing paediatric physiotherapists with opportunities to understand their own beliefs and attitudes, and how these beliefs and attitudes relate to those required in RD-FCI, may support the adoption of beliefs and attitudes needed to become a coach in RD-FCI.39 It is very challenging to change strong habits. The opportunity for
ample practice of a new behaviour in a supportive environment is a good way to acquire new and long lasting behaviours.48 The insight we gained through the process of writing
of the narrative review made us critically aware of the challenges and barriers associated with the implementation of COPCA - a family-centred approach using coaching. As a result, we better understand how we have to arrange courses on COPCA.
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Evaluation of changes in physiotherapeutic actions during a COPCA
course
The COPCA course during which we monitored changes of physiotherapeutic actions with GOP 2.0 (chapter 6) consisted of three parts of contact days (1 x 2 days and 2 x 1 day) and two intervals. The entire course lasted six months. In parallel, physiotherapits continued treating children. Already after the first part of contact days, the participating conventionally trained physiotherapists changed their actions considerably. In the category ‘neuromotor actions’, more self-produced motor behaviour and trial-and-error experiences and less hands-on guidance was offered to the infants. In addition, in the category ‘educational actions towards the caregivers’, caregiver coaching was applied almost all the time. This indicated a shift from a child-centred to a family-centred form of early physiotherapeutic intervention and an increased application of the neuromotor principles of the NGST. However, during the entire course, the neuromotor actions changed less, less extensively and less sustainably than the educational actions; we even noticed a minor drop back to previous actions after the third part of contact days in the category of ‘neuromotor actions’. With course progression, the caregivers’ active involvement in the intervention process increased. In the intervention session recorded prior to the course only one third of the caregivers were actively involved, in the session after the course all caregivers were part of the treatments.
This indicated that the paediatric physiotherapist as well as the family members had accomplished a successful change of role. The amount of observed changes in behaviour is in accordance with the “behaviour change wheel” of Michie et al:48
the changes depend on physiological and psychological capability, reflective and automatic motivation and social and physical opportunity. Capability is among other things dependent on knowledge and skills. It may be assumed that the COPCA course offered the candidate COPCA coaches enough opportunities for the acquisition of new knowledge and skills related to coaching, and that the physiotherapists offered these opportunities to the family members through coaching. The paediatric physiotherapist as well as the caregivers participated in this study on a voluntary, self-chosen basis, indicating that both parties were intrinsically motivated to apply COPCA in the intervention process. They had carried out a reflective process and made the decision to participate. We assume that the intervention process over six months in the home environment of the family - indicating an environmental restructuring - offered all participants the opportunity for the role change.
The changes in neuromotor actions were less robust. The application of hands-on techniques represents a strhands-ong habit. In this chands-ontext, also the automatic part of motivation plays an important role. Changing strong habits requires besides capacity,
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reflective motivation and opportunities, also a supportive environment, opportunities for modelling of the new behaviour and enablement, e.g. enough repetition and practice.47 Obviously the COPCA course did not include these elements sufficiently. On
the basis of the findings of this study the curriculum of the COPCA course has been adapted. We increased the number of course days (3 x 2), which offers the opportunity to spend more time to discuss the application of the neuromotor NGST principles in the family context, to give more feedback and to show more videos of the desired neuromotor actions.
This study also confirmed the usefulness of GOP 2.0. Moreover we realised, how important behaviour change theories are for the understanding of the behaviour of the course participants and to understand how challenges and barriers encountered in behaviour change can be mastered.
The view of the caregivers
COPCA does not only demand behaviour changes from the physiotherapists but also from the family members. The study described in the previous section offered the opportunity to evaluate the caregivers’ experiences with COPCA in a qualitative study (chapter 7). The study revealed that the responding mothers evaluated COPCA as a very suitable form of intervention for the whole family. They described benefits of COPCA for the infants as well as for the caregivers. The mothers appreciated especially the home-based intervention, the support from the COPCA coaches, the attainment of new competences, and being involved in the intervention process as active and equal partners. The learning process during the six months of COPCA intervention had enabled them to support the infant at home autonomously, to recognise the competences of the infant and therewith to gain confidence in the infant’s development.
This study indicated that mothers appreciated both components of COPCA, the family component and the neurodevelopmental component. They were involved in the intervention process as active an equal partner and became autonomous in supporting motor development of their infant. In doing so they felt supported by the COPCA coach, indicating that the COPCA coach met their needs. Through challenging the infant to self-produced motor behaviour, they recognised the capacities of the infant and their own competences, which brought them confidence. The ecological orientation of COPCA, which means that the intervention took place in the real life environment of the family during daily care giving activities, was crucial for all mothers. The results of the study suggest that the two components of COPCA are not only a theoretical framework for COPCA coaches, but also translated meaningfully into daily practice for the mothers. Our findings are in line with the review of Kruijsen-Terpstra et al. (2014),49 which showed
that parents of children with CP preferred intervention in the home environment
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211 instead of in a clinical setting. The findings also correspond to those of the study of Hinojosa (1990)50 which highlighted the importance of appropriate family support as
a critical factor for the amount of active involvement of caregivers in the intervention process. Offering the infant opportunities for self-produced motor behaviour integrated in recurring daily life activities, presumably results in high dosing of variable activities, which is a critical factor in respect to the effectiveness of early intervention.51
However, physiotherapeutic early intervention is not frequently offered in the home environment. Of the nine studies included in the systematic review of Spittle et al. (2015)52 evaluating the effect of physiotherapeutic early intervention, only two
studies were conducted in the home environment of the families. Offering intervention at home is also in Switzerland a difficult topic, as the insurances, who usually pay the intervention, are mostly not willing to pay for the extra costs of home intervention, like travel expenses and travel time. This condition represents a barrier to provide home-based intervention in form of an external restriction. For this reason it is becoming clear that also contextual factors have to be considered in the implementation process of COPCA. The financial barrier in the home-aspect of the COPCA intervention may be reduced by means of provision of information on COPCA, including the findings of recent research, to those financially responsible, political instances, and the associations of paediatricians.
The effect of COPCA on motor development in very preterm infants
In an randomized controlled pilot trial combined with process evaluation including infants born very preterm and their familes, we addresed the questions whether (1) child- and family- related outcomes after six months of intervention, differed between the COPCA (n=8) and TIP (n=8) groups, and (2) if specific physiotherapy actions were associated with the infants’ motor outcome (chapter 8). The application of GOP 2.0 allowed us to show that the two interventions differed significanly with respect to neuromotor and educational actions and to the degree to which caregivers were actively involved in the intervention. None of the infants who patricipated in the study was diagnosed with CP at the age of two years. This indicates that the study sample represented a group of infants with only mild to moderately increased risks of motor impairment. The study demonstrated, that COPCA in comparison to TIP resulted in better results at 18 months of corrected age in the IMP domains variation and performance. Furthermore, we found a time-group interaction in these two IMP domains, indicating that the COPCA group improved significantly more between basline and follow-up than the TIP group, especially between the end of the intervention and follow-up. The process evaluation showed, that two actions characteristic of TIP, namely caregiver training and hands-on techniques, were negatively associated with scores on9
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the IMP domain variation. In contrast the typical COPCA action caregiver coaching was positively associated with the IMP domain performance.
Our results, better IMP variation and performance in the COPCA group, correspond to those of Sgandurra et al.14,53 Both studies showed that it is possible to increase
variation (the size of motor repertoire) in infants at mildly to moderately increased risk of developmental motor disorders through early intervention. The studies suggested that the increased motor repertoire is a result of enhanced explorative activity of the infant and an enriched environment. In the study of Sgandurra et al.,14,53 a highly
instrumentalised playpen with preprogrammed activities (‘scenarios’) (Care Toy) was applied to enhance the infant’s activity and to create an enriched environment. In COPCA the enhancement of activities is offered primarily through caregiver-infant interaction during daily routines like playing, which aims to challenge the infant not only in motor development but also in general development, including cognition, language and social development. The other result of the RCT that caregiver coaching was associated with better performance is in line with the study of Blauw-Hospers et al.,5 who also demonstrated that COPCA’s caregiver coaching had a beneficial effect on
motor outcome at 18 months.
The study concluded that caregiver coaching is a promising and sustainable intervention element. It enables parents to challenge the infants’ motor development long-term and in an autonomous way in their real life environment. In turn, this challenge allows the infant to increase his/her motor repertoire. Hands-on techniques in infants with no or only a mild brain lesion seem to interfere with the infants’ explorative activity and therewith may reduce the opportunities for enhancing the motor repertoire.
METHODOLOGICAL CONSIDERATIONS
Providing insight in the application and effect of infant physiotherapy and the implementation of a new programme, which is associated with a change of paradigm is a methodological challenge.
A helpful tool in the quantitative studies of this thesis (chapters 3, 6, 9) was GOP 2.0, which allowed us to objectively and reliably quantify the contents of infant physiotherapy, in order to evaluate the changes of contents over time, to compare different intervention programmes, and to conduct a process evaluation. These aspects represent strengths of the individual studies. Therefore a main strength for the whole thesis was that we started with the assessment of the psychometric properties of GOP 2.0 in Switzerland, a prerequisite for its application. Another strength of the series of studies was that we quantified the contents of TIP in Switzerland before beginning the implementation process of COPCA. This allowed us to anticipate challenges, hurdles and barriers that we might encounter in the process of implementation (e.g.
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213 impart knowledge on evidence-based recommmendations in early physiotherapy interventions), in order to be as successful as possible. The writing of the perspective paper on coaching in rehabilitation and the narrative review may also be regarded as strengths in respect to the whole process, as the perspective paper clarified the comprehensive framework for candidate and certificated COPCA coaches. The writing of the narrative review enhanced our awareness of the challenges and barriers associated with the implementation process and resulted in a better understanding about how we have to conduct the COPCA courses. The specific strength of the study described in chapter 6 was the inclusion and application of behaviour change theories in order to understand the behaviour of the course participants and to become aware how challenges and barriers could be mastered. The specific strengths of the RCT of chapter 8 are the two-fold approach of investigating effectiveness and processes as well as its longitudinal design. The process evaluation allowed for gaining important information about the underlying working mechanism of COPCA, whereas the longitudinal design allowed for the application of a linear mixed model analysis, which enabled us to uncover the sustainable effect of coaching.
Naturally, the complex processes underlying this thesis are also associated with limitations. A limitation in all the studies including human beings (chapters 2, 3, 6, 7, 8) is a potential selection bias, as participation was voluntary. This implies that the study samples (therapist and families) were not necessarily representative samples. Therefore, the results should be reproduced before generalisation. Another limitation of all studies that used video-recordings of therapeutic sessions (chapters 2, 3, 6, 8) was that the presence of a video camera and an operator might have influenced the behaviour of the therapist and family members. A specific limitation of the study described in chapter 6 is that we monitored changes of physiotherapeutic actions only until two weeks after the course. This precluded the evaluation of long-term changes in therapeutic actions. The qualitative study about the caregivers’ experiences (chapter 7) has two specific limitations. First, no fathers participated in the study, and second we did not explicitly ask about unhelpful or disagreeable aspects of COPCA. As a consequence, the study shows only the experiences of the mothers. We do not know whether the results would have been different if fathers also had participated. This might be the case, as King et al.54 described differences in the evaluation of care between mothers
and fathers. Second, we do not know, if some parents also see harm in COPCA. The last limitations I want to point out consider the RCT (chapter 8). Its major limitation is the small sample size, resulting in underpowering. Despite the underpowering, we found significant differences between the two intervention groups. This may be a chance finding, but it may also mean that the effect of well-implemented COPCA is larger than we expected. The small sample size of the study may be attributed to the following:
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(1) the recruitment had to be conducted by neonatologists whose overloaded working hours left little mind-space for a post discharge study on early physiotherapeutic intervention, (2) families with very preterm born infants have already quite a large burden, which sometime makes them hesitant to participate in research, and (3) due to the slow recruitment, enrollment had to be stopped before the planned number of infants had been included, as the project’s resources ran out. Another limitation of the RCT was that long-term outcome data of the participating infants were lacking, as the last follow-up took place at the corrected age of two years. Outcome assessed in this stage of development should be interpreted with caution, ascognitive and motor delays may become obvious later in life, when demands in cognition and motor skills increase.55
The final limitation that I would like to mention is that were not able to demonstrate potentially possible family related differences between the two interventions.
FUTURE PERSPECTIVES
The GOP 2.0 allowed us to objectively and reliably quantify the contents of infant physiotherapy in order to evaluate changes of contents over time, to compare different intervention programmes, and to conduct a process evaluation. However, the GOP 2.0 was developed, before we wrote the perspective in rehabilitation and the narrative review (chapter 4 and 5). Through the discussions about the contents of these papers and the related literature, we got aware, that the GOP 2.0 does not represent the different coaching skills described by Rush et al.29 to a sufficiently differentiated extent.
A further adaptation of the GOP 2.0 in this direction would offer the possibility to quantitatively evaluate the different coaching skills by paediatric physiotherapists in general and COPCA coaches in particular. Moreover, it would offer the possibility to perform process evaluation, that is, the evaluation of potential associations between individual coaching skills and defined family related outcomes. This could be a valuable step on the way to determine which particular coaching skills promote well-being of families and which coaching components are mainly responsible for the reported positive results of coaching.36 This is an important question, which is not answered yet.
Other possibilities to bring light in this situation are qualitative studies examining caregivers’ experiences with coaching approaches. Our study about caregivers’ experiences furnished some insight in caregivers’ experiences with COPCA. However, further studies, which include also fathers, that address also the potentially negative aspects of COPCA, and that pay attention to the value of specific coaching skills, may deepen our insight. A phenomenological approach using semi-structured interviews offers a good possibility to reach this goal. Another open question, that warrants further research, is whether coaching is an effective approach for all families or whether some families would prefer and benefit more from conventional caregiver training.
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215 Currently, reliable, applicable and sensitive family related outcomes, measuring perception of family-centeredness in families with infants and very young children, family engagement, parents’ confidence, competence, and capacity are missing. I urge that such instruments are developed in the near future.
Finally, I recommend future studies that evaluate the effect of COPCA in a large group of infants with mildly to moderately increased risks of motor developmental disorders (e.g. preterm born infants), with adequate power and long-term follow-up, to validate the promising results of our explorative RCT.
CONCLUDING REMARKS
The main findings of this thesis result in the following conclusions:
• The GOP 2.0 is a valuable tool with adequate psychometric properties to quantify contents of paediatric physiotherapy interventions. It allows for process evaluation, which in turn may provide insight in the underlying mechanisms of potential influences of intervention on outcome. In other words process evaluation provides knowledge on the effectiveness of specific intervention elements, which is essential for the promotion of evidence-based practice.
• Increased awareness on the implementation of evidence-based recommmendations in daily practice of physiotherapeutic early intervention is warranted and mandatory.
• Feedback on the basis of video recordings of interventions is a powerful means to improve the awareness of actions and interactions during the intervention sessions. This awareness is a premise for the implementation of evidence-based practice. Paediatric physiotherapists should be invited to provide and receive reciprocal peer-feedback on video recordings of intervention sessions. • The application of coaching approaches demands the acquisition of new
knowledge and skills, a change of attitudes and habits, as well as the establishment of a common sense between the physiotherapist and the family members. Thus, a successful implementation of coaching approaches requires role adaptations. To become aware of the challenges and barriers encountered on the path of role transition, and to develop strategies to master the challenges and barriers, behaviour change theories are essential. Coaches and family members should be aware, that the processes involved in change of roles require ample time.
• Caregivers highly appreciate the support from coaches, because coaching enables them to autonomously promote their infant’s development by
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integrating therapeutic activities in daily routines. Caregiver coaching was associated with an increased motor repertoire of the infant and with better family empowerment and quality of life. Therefore coaching is a promising intervention element, which may sustainably improve infants’ and family outcomes.
Disclousure statement
The author declares no conflict of interest. COPCA courses are provided by the Zurich University of Applied Sciences. The author has no financial profit.
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REFERENCES
1. Hielkema T, Toonen RF, Hooijsma SJ, et al. Changes in the Content of Pediatric Physical Therapy for Infants: A Quantitative, Observational Study. Phys Occup
Ther Pediatr. December 2017:1-32. doi:10.1080/01942638.2017.1405863
2. Moher D, Hopewell S, Schulz KF, et al. CONSORT 2010 Explanation and Elaboration: updated guidelines for reporting parallel group randomised trials.
BMJ. 2010;340. doi:10.1136/bmj.c869
3. Hoffmann TC, Glasziou PP, Boutron I, et al. Better reporting of interventions: template for intervention description and replication (TIDieR) checklist and guide. BMJ. 2014;348:g1687. doi:10.1136/bmj.g1687
4. Blauw-Hospers CH, Dirks T, Hulshof LJ, Hadders-Algra M. Development of a quantitative tool to assess the content of physical therapy for infants. Pediatr
Phys Ther. 2010;22(2):189-197. doi:10.1097/PEP.0b013e3181dbd5f1
5. Blauw-Hospers CH, Dirks T, Hulshof J, Bos AF, Hadders-Algra M. Pediatric physical therapy in infancy: from nightmare to dream? A two-arm randomized trial. Phys Ther. 2011; 91: 132-138. doi: 10.2522/ptj.20100205.
6. Heineman KR, Bos AF, Hadders-Algra M. The Infant Motor Profile: a standardized and qualitative method to assess motor behaviour in infancy. Dev
Med Child Neurol. 2008;50(4):275-282. doi:10.1111/j.1469-8749.2008.02035.x
7. Heineman KR, La Bastide-Van Gemert S, Fidler V, Middelburg KJ, Bos AF, Hadders-Algra M. Construct validity of the Infant Motor Profile: relation with prenatal, perinatal, and neonatal risk factors. Dev Med Child Neurol. 2010;52(9):e209-215. doi:10.1111/j.1469-8749.2010.03667.x
8. Hadders-Algra M. The neuronal group selection theory: a framework to explain variation in normal development. Dev Med Child Neurol. 2000; 42: 566–572. doi: 10.1017/s0012162200001067
9. Hadders-Algra M. Variation and variability: key words in human motor development. Phys Ther. 2010;90(12):1823-1837. doi:10.2522/ptj.20100006 10. Hecker E, Baer GD, Stark C, Herkenrath P, Hadders-Algra M. Inter- and
Intrarater Reliability of the Infant Motor Profile in 3- to 18-Month-Old Infants.
Pediatr Phys Ther. 2016;28(2):217-222. doi:10.1097/PEP.0000000000000244
11. Heineman KR, Bos AF, Hadders-Algra M. Infant Motor Profile and cerebral palsy: promising associations. Dev Med Child Neurol. 2011;53 Suppl 4:40-45. doi:10.1111/j.1469-8749.2011.04063.x
12. Heineman KR, Schendelaar P, Van den Heuvel ER, Hadders-Algra M. Motor development in infancy is related to cognitive function at 4 years of age. Dev
Med Child Neurol. 2018;60(11):1149-1155. doi:10.1111/dmcn.13761
13. Hielkema T, Blauw-Hospers CH, Dirks T, Drijver-Messelink M, Bos AF, Hadders-Algra M. Does physiotherapeutic intervention affect motor outcome in high-risk infants? An approach combining a randomized controlled trial and process evaluation. Dev Med Child Neurol. 2011;53(3):e8-15. doi:10.1111/j.1469-8749.2010.03876.x
9
Schirin_Thesis.indd 217
218
14. Sgandurra G, Lorentzen J, Inguaggiato E, et al. A randomized clinical trial in preterm infants on the effects of a home-based early intervention with the “CareToy System.” PLoS One. 2017;12(3). doi:10.1371/journal.pone.0173521 15. Dunst Carl J., Trivette Carol M., Hamby Deborah W. Meta-analysis of family-centered helpgiving practices research. Ment Retard Dev Disabil Res Rev. 2007;13(4):370-378. doi:10.1002/mrdd.20176
16. Dirks T, Blauw-Hospers CH, Hulshof LJ, Hadders-Algra M. Differences between the family-centered “COPCA” program and traditional infant physical therapy based on neurodevelopmental treatment principles. Phys Ther. 2011; 91:1303-22. doi: 10.2522/ptj.20100207.
17. Koren PE, DeChillo N, Friesen BJ. Measuring empowerment in families whose children have emotional disabilities: A brief questionnaire. Rehabilitation
Psychology. 1992;37(4):305-321. doi:10.1037/h0079106
18. King S, Rosenbaum P, King G. The Measure of Processes of Care: A Means to
Assess Family-Centred Behaviours of Health Care Providers. Hamiltion, ON:
McMaster University, Neurodevelopmental Clinical Research Unit; 1995. 19. Iten N, Kraus de Camargo O. The Measure of Processes of Care (MPOC-56 (R)
D.04). ã 1995 King, Rosenbaum and King. CanChild for Childhood Disability
Research, McMaster University
20. King G, Schwellnus H, Servais M, Baldwin P. Solution-Focused Coaching in Pediatric Rehabilitation: Investigating Transformative Experiences and Outcomes for Families. Phys Occup Ther Pediatr. 2019; 39:16-32. doi: 10.1080/01942638.2017.1379457
21. Blauw-Hospers CH. Early intervention in infants at high risk for developmental disorders [dissertation]. Groningen; NL: University of Groningen; 2010. 22. Almasri NA, An M, Palisano RJ. Parents’ Perception of Receiving
Family-Centered Care for Their Children with Physical Disabilities: A Meta-Analysis.
Phys Occup Ther Pediatr. July 2017:1-17. doi:10.1080/01942638.2017.1337664
23. Ward R, Reynolds JE, Pieterse B, Elliott C, Boyd R, Miller L. Utilisation of coaching practices in early interventions in children at risk of developmental disability/delay: a systematic review. Disabil Rehabil. April 2019:1-22. doi:10. 1080/09638288.2019.1581846
24. Baldwin P, King G, Evans J, McDougall S, Tucker MA, Servais M. Solution-focused coaching in pediatric rehabilitation: an integrated model of practice. Phys
Occup Ther Pediatr. 2013;33:467-483. doi: 10.3109/01942638.2013.784718
25. Hielkema T, Boxum AG, Hamer EG, et al. LEARN2MOVE 0-2 years, a randomized early intervention trial for infants at very high risk of cerebral palsy: family outcome and infant’s functional outcome. Disabil Rehabil. May 2019:1-9. do i:10.1080/09638288.2019.1610509
26. Bamm EL, Rosenbaum P. Family-centered theory: origins, development, barriers, and supports to implementation in rehabilitation medicine. Arch
Phys Med Rehabil. 2008;89(8):1618-1624. doi:10.1016/j.apmr.2007.12.034
Schirin_Thesis.indd 218
219 27. Rosenbaum P, King S, Law M, King G, Evans J. Family-Centred Service. Phys
Occup Ther Pediatr. 1998;18(1):1-20. doi:10.1080/J006v18n01_01
28. Hadders-Algra M. Early human motor development: From variation to the ability to vary and adapt. Neurosci Biobehav Rev. 2018;90:411-427. doi:10.1016/j.neubiorev.2018.05.009
29. Edelman GM. Neural Darwinism: The Theory of Neural Group Selection. Oxford (UK): University Press; 1989.
30. Sameroff AJ, ed. The transactional model of development: How children
and contexts shape each other. Washington, DC: American Psychological
Association; 2009.
31. Ives Y. What is ‘Coaching’? An Exploration of Conflicting Paradigms. Int J Evid
Based Coach Mentor. 2008;6:100-113.
32. Goldberg H, Goldberg I. Family therapy: an overview. Belmont, CA: Thomson Brooks; 2008.
33. Watzlawick P, Beavin-Bavelas J, Jackson D. Some tentative axioms of communication. In: Pragmatics of Human Communication - A Study of Interactional
Patterns, Pathologies and Paradoxes. New York, NY: WW Norton; 1967.
34. Rogers CR. A theory of therapy personality and interpersonal relationships. In: Kochm SW, ed. Psychology: A study of science. New York, NY: McGraw-Hill; 1959. 35. Rogers CR. Client-centered therapy: Its current practice, implications, and
theory. Boston, MS: Houghton Mifflin; 1951.
36. Kemp P, Turnbull AP. Coaching With Parents in Early Intervention: An Interdisciplinary Research Synthesis. Infants & Young Children. 2014;27(4):305. doi:10.1097/IYC.0000000000000018
37. Schwellnus H, King G, Thompson L. Client-centred coaching in the paediatric health professions: a critical scoping review. Disabil Rehabil. 2015;37(15):1305-1315. doi:10.3109/09638288.2014.962105
38. Campbell PH, Sawyer LB. Supporting Learning Opportunities in Natural Settings Through Participation-Based Services. J Early Intervention. 2007;29(4):287-305. doi:10.1177/105381510702900402
39. Campbell PH, Sawyer LB. Changing Early Intervention Providers’ Home Visiting Skills Through Participation in Professional Development. Topic Early Child
Spec Educ. 2009;28(4):219-234. doi:10.1177/0271121408328481
40. Salisbury CL, Woods J, Copeland C. Provider Perspectives on Adopting and Using Collaborative Consultation in Natural Environments. Topic Early Child
Spec Educ. 2010;30(3):132-147. doi:10.1177/0271121409349769
41. Campbell PH, Coletti CE. Early Intervention Provider Use of Child Caregiver-Teaching Strategies. Infant Young Child. 2013;26(3):235-248. doi:10.1097/ IYC.0b013e318299918f
42. Meadan H, Douglas SN, Kammes R, Schraml-Block K. “I’m a Different Coach with Every Family”: Early Interventionists’ Beliefs and Practices. Infant Young
Child. 2018;31(3):200-214. doi:10.1097/IYC.0000000000000118
9
Schirin_Thesis.indd 219
220
43. Kemp P, Turnbull AP. Coaching With Parents in Early Intervention: An Interdisciplinary Research Synthesis. Infant Young Child. 2014;27(4):305. doi:10.1097/IYC.0000000000000018
44. Rush D, Shelden M, Dunn W. The Early Childhood Coaching Handbook. Tion. Baltimore, Md: BROOKES PUB; 2011.
45. Graham F, Boland P, Ziviani J, Rodger S. Occupational therapists’ and physiotherapists’ perceptions of implementing Occupational Performance Coaching. Disabil Rehabil. 2018;40(12):1386-1392. doi:10.1080/09638288.2 017.1295474
46. Friedman M, Woods J, Salisbury C. Caregiver Coaching Strategies for Early Intervention Providers: Moving Toward Operational Definitions. Infant Young
Child. 2012;25(1):62. doi:10.1097/IYC.0b013e31823d8f12
47. Salisbury C, Woods J, Snyder P, et al. Caregiver and Provider Experiences With Coaching and Embedded Intervention. Topic Early Child Spec Educ. 2018;38(1):17-29. doi:10.1177/0271121417708036
48. Michie S, van Stralen MM, West R. The behaviour change wheel: A new method for characterising and designing behaviour change interventions.
Implement Sci. 2011;6:42. doi:10.1186/1748-5908-6-42
49. Kruijsen-Terpstra AJA, Ketelaar M, Boeije H, et al. Parents’ experiences with physical and occupational therapy for their young child with cerebral palsy: a mixed studies review. Child Care Health Dev. 2014;40(6):787-796. doi:10.1111/ cch.12097
50. Hinojosa J. How Mothers of Preschool Children with Cerebral Palsy Perceive Occupational and Physical Therapists and Their Influence on Family Life. The
Occup Ther J Res. 1990;10(3):144-162. doi:10.1177/153944929001000302
51. Hadders-Algra M, Boxum AG, Hielkema T, Hamer EG. Effect of early intervention in infants at very high risk of cerebral palsy: a systematic review.
Dev Med Child Neurol. 2017;59(3):246-258. doi:10.1111/dmcn.13331
52. Spittle A, Orton J, Anderson PJ, Boyd R, Doyle LW. Early developmental intervention programmes provided post hospital discharge to prevent motor and cognitive impairments in preterm infants (Review). Cochrane Database
Syst Rev. 2015 Nov 24; 11:CD005495.
53. Sgandurra G, Bartalena L, Cecchi F, et al. A pilot study on early home-based intervention through an intelligent baby gym (CareToy) in preterm infants.
Res Dev Disabil. 2016;53-54:32-42. doi:10.1016/j.ridd.2016.01.013
54. King Gillian A, King Susanne M, Rosenbaum Peter L. How mothers and fathers view professional caregmng for children with disabilities. Dev Med Child
Neurol. 2008;38(5):397-407. doi:10.1111/j.1469-8749.1996.tb15098.x
55. de Kieviet JF, Piek JP, Aarnoudse-Moens CS, Oosterlaan J. Motor development in very preterm and very low-birth-weight children from birth to adolescence: a meta-analysis. JAMA. 2009;302(20):2235-2242. doi:10.1001/jama.2009.1708
Schirin_Thesis.indd 220
221
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