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Purpose: Evidence for efficacy of early intervention in infants at high risk of cerebral palsy (CP) is limited. We compared outcome of infants at very high risk of CP after receiving the family centered program COPing with and CAring for infants with special needs (COPCA) or typical infant physiotherapy. Materials and methods: Forty-three infants were randomly assigned before the corrected age of 9 months to 1 year of COPCA ( n  = 23) or typical infant physiotherapy ( n  = 20). Neuromotor development, cognition, and behavior was assessed until 21 months corrected age. Video-recorded physiotherapy sessions were quantitatively analyzed for further process analyses. Outcome was evaluated with nonparametric tests and linear mixed effect models. Results: During and after the interventions, infant outcome in both intervention groups was similar [primary outcome Infant Motor Profile: COPCA 82 (69-94), typical infant physiotherapy 81 (69-89); Hodges Lehman estimate of the difference 0 (confidence interval -5;4)]. Outcome was not associated with contents of intervention. Conclusions: One year of COPCA and 1 year of typical infant physiotherapy in infants at high risk of CP resulted in similar neurodevelopmental outcomes. It is conceivable that combinations of active ingredients from different approaches are needed for effective early intervention.IMPLICATIONS FOR REHABILITATIONFor infants at very high risk of cerebral palsy, 1 year of intervention with the family-centred programme Coping with and Caring for infants with special needs resulted in similar infant outcome as 1 year of typical infant physiotherapy.Infant's neuromotor, cognitive, and behavioural outcome was not associated with specific interventional elements, implying that the various elements may have a similar effect on developmental outcome.We suggest that a specific mix of ingredients of different approaches may work best, resulting in comprehensive care including both infant and family needs.

Purpose: To compare family and functional outcome in infants at very high risk of cerebral palsy, after receiving the family centred programme "Coping with and Caring for infants with special needs (COPCA)" or typical infant physiotherapy. Materials and methods: Forty-three infants at very high risk were included before 9 months corrected age and randomly assigned to one year COPCA ( n  = 23) or typical infant physiotherapy ( n  = 20). Family and infant outcome were assessed before and during the intervention. Physiotherapy intervention sessions were analysed quantitatively for process analysis. Outcome was evaluated with non-parametric tests and linear mixed-effect models. Results: Between-group comparisons revealed no differences in family and infant outcomes. Within-group analysis showed that family's quality of life improved over time in the COPCA-group. Family empowerment was positively associated with intervention elements, including "caregiver coaching." Conclusions: One year of COPCA or typical infant physiotherapy resulted in similar family and functional outcomes. Yet, specific intervention elements, e.g., coaching, may increase empowerment of families of very high risk infants and may influence quality of life, which emphasizes the importance of family centred services.Implications for rehabilitationOne year of the family centred programme "Coping with and a Caring for infants with special needs" compared with typical infant physiotherapy resulted in similar family outcome and similar functional outcome for the infants at very high risk for cerebral palsy.Specific contents of intervention, such as caregiver coaching, are associated with more family empowerment and increased quality of life.Emphasis on family needs is important in early intervention for infants at very high risk for cerebral palsy.

Background: Development of postural problems in Cerebral Palsy (CP) is largely unknown. Postural muscle activity is organized into two levels: 1) direction-specificity; 2) fine-tuning of direction-specific activity., Aim: To study development of postural control until 21 months corrected age in subgroups of infants at very high-risk (VHR) of CP: a) with and without CP at 21 months; b) with and without cystic periventricular leukomalacia (cPVL), the brain lesion with highest risk of CP., Methods and Procedures: Longitudinal electromyography recordings of postural muscles during reaching were made in 38 VHR-infants (severe brain lesion or clear neurological signs) between 4.7 and 22.6 months (18 CP, of which 8 with cPVL). Developmental trajectories were calculated using linear mixed effect models., Outcomes and Results: VHR-infants with and without CP showed virtually similar postural development throughout infancy. The subgroup of VHR-infants with cPVL improved performance in direction-specificity with increasing age, while they performed throughout infancy worse in fine-tuning of postural adjustments than infants without cPVL., Conclusions and Implications: VHR-infants with and without CP have a similar postural development that differs from published trajectories of typically developing infants. Infants with cPVL present from early age onwards dysfunctions in fine-tuning of postural adjustments; they focus on direction-specificity., (Copyright © 2018 Elsevier Ltd. All rights reserved.)

Background: Early identification of infants at risk of cerebral palsy (CP) is desirable in order to provide early intervention. We previously demonstrated differences in knee jerk responses between 3-month-old high risk and typically developing infants., Aims: To improve early identification by investigating whether the presence of tonic responses (continuous muscle activity occurring after the typical phasic response), clonus or contralateral responses to the knee jerk during infancy is associated with CP., Study Design: Longitudinal EMG-study., Subjects: We included 34 high-risk infants (median gestational age 31.9 weeks) who participated in the LEARN2MOVE 0-2 years trial., Outcome Measures: Video-recorded knee jerk EMG-assessments were performed during infancy (1-4 times). Developmental outcome was assessed at 21 months corrected age (CA). Binomial generalized estimating equations models with repeated measurements were fitted using predictor variables., Results: Infants who later were diagnosed with CP (n = 18) showed more often than infants who were not diagnosed with CP i) tonic responses - from 4 months CA onwards, ii) clonus - from 13 months CA onwards, and iii) contralateral responses - from 15 months CA onwards., Limitations: The main limitation is the relatively small sample size., Conclusions: The assessment of tonic responses to the knee jerk using EMG may be a valuable add-on tool to appraise a high risk of CP., (Copyright © 2018 Elsevier B.V. All rights reserved.)

Background: In typical development, postural adjustments during reaching change in the second half of infancy, including increasing rates of direction-specific adjustments. These changes are absent or different in infants at risk of cerebral palsy (CP). To discover whether these changes are related to acquisition of independent walking, we studied postural adjustments during reaching in infants before and after they learned to walk., Methods: Ten typically developing (TD) infants and 11 infants at very high risk (VHR) of CP were assessed before and after they learned to walk. Reaching movements were elicited during supported sitting, while surface electromyography was recorded of arm, neck, and trunk muscles. Percentages of direction-specific adjustments (first level of control), and recruitment patterns and anticipatory activation (second level of control) were calculated., Results: In both groups, postural adjustments during reaching were similar before and after acquisition of independent walking. Direction-specificity increased with age in typically developing infants but not in VHR-infants., Conclusion: Increasing age rather than the transition to independent walking is associated with increasing direction-specificity of TD-infants during reaching while sitting, while infants at very high risk of CP show no increase in direction-specificity, suggesting that they gradually grow into a postural deficit., (Copyright © 2017 Elsevier Inc. All rights reserved.)

Aim: To assess development of reaching and head stability in infants at very high risk (VHR-infants) of cerebral palsy (CP) who did and did not develop CP., Method: This explorative longitudinal study assessed the kinematics of reaching and head sway in sitting in 37 VHR-infants (18 CP) one to four times between 4.7 months and 22.6 months corrected age. Developmental trajectories were calculated using linear mixed effect models. Motor function was evaluated with the Infant Motor Profile (IMP) around 13 months corrected age., Results: Throughout infancy, VHR-infants with CP had a worse reaching quality than infants without CP, reflected for example by more movement units (factor 1.52, 95% CI 1.16-1.99) and smaller transport movement units (factor 1.86, 95% CI 1.20-2.90). Total head sway of infants with and without CP was similar, but infants with CP used more head movement units to achieve stability. The rate of developmental change in infants with and without CP was similar. Around 13 months, head control and reaching quality were interrelated; both were associated with IMP-scores., Interpretation: Infants with CP showed a worse kinematic reaching quality and head stability throughout infancy from early age onwards than VHR-infants without CP, implying that kinematically they do not grow into a deficit, but exhibit deficits from early infancy on., What This Paper Adds: Reaching quality improves throughout infancy in all infants at high risk (VHR-infants). Infants with cerebral palsy (CP) show a worse reaching quality than VHR-infants without CP. Infants with CP achieve head stability differently from infants without CP. Infants with CP exhibit kinematic reaching problems from early age onwards., (© 2017 Mac Keith Press.)

Aim: First, to systematically review the evidence on the effect of intervention applied during the first postnatal year in infants with or at very high risk of cerebral palsy (CP) on child and family outcome. Second, to assess whether type and dosing of intervention modify the effect of intervention., Method: Relevant literature was identified by searching the PubMed, Embase, and CINAHL databases. Selection criteria included infants younger than 12 months corrected age with or at very high risk of CP. Methodological quality including risk of bias was scrutinized., Results: Thirteen papers met the inclusion criteria. Seven studies with moderate to high methodological quality were analysed in detail; they evaluated neurodevelopmental treatment only (n=2), multisensory stimulation (n=1), developmental stimulation (n=2), and multifaceted interventions consisting of a mix of developmental stimulation, support of parent-infant interaction, and neurodevelopmental treatment (n=2). The heterogeneity precluded conclusions. Yet, two suggestions emerged: (1) dosing may be critical for effectiveness; (2) multifaceted intervention may offer best opportunities for child and family., Interpretation: The literature on early intervention in very high-risk infants with sufficient methodological quality is limited, heterogeneous, and provides weak evidence on the effect. More studies are urgently needed. Suggestions for future research are provided., (© 2016 Mac Keith Press.)

Aim: Having observed slow pupillary light responses (PLRs) in infants at high risk of cerebral palsy, we retrospectively evaluated whether these were associated with specific brain lesions or unfavourable outcomes., Methods: We carried out neurological examinations on 30 infants at very high risk of cerebral palsy five times until the corrected age of 21 months, classifying each PLR assessment as normal or slow. The predominant reaction during development was determined for each infant. Neonatal brain scans were classified based on the type of brain lesion. Developmental outcome was evaluated at 21 months of corrected age with a neurological examination, the Bayley Scales of Infant Development Second Edition and the Infant Motor Profile., Results: Of the 30 infants, 16 developed cerebral palsy. Predominantly slow PLRs were observed in eight infants and were associated with periventricular leukomalacia (p = 0.007), cerebral palsy (p = 0.039), bilateral cerebral palsy (p = 0.001), poorer quality of motor behaviour (p < 0.0005) and poorer cognitive outcome (p = 0.045)., Conclusion: This explorative study suggested that predominantly slow PLR in infants at high risk of cerebral palsy were associated with periventricular leukomalacia and poorer developmental outcome. Slow PLR might be an expression of white matter damage, resulting in dysfunction of the complex cortico-subcortical circuitries., (©2016 Foundation Acta Paediatrica. Published by John Wiley & Sons Ltd.)

The aim of this systematic review was to study motor and cognitive outcome in infants with severe early brain lesions and to evaluate effects of side of the lesion, sex, and social economic status on outcome. A literature search was performed using the databases Pubmed and Embase. Included studies involved infants with either cystic periventricular leukomalacia (cPVL), preterm, or term stroke (i.e. parenchymal lesion of the brain). Outcome was expressed as cerebral palsy (CP) and intellectual disability (mental retardation). Median prevalence rates of CP after cPVL, preterm, and term stroke were 86%, 71%, and 29% respectively; of intellectual disability 50%, 27%, and 33%. Most infants with cPVL developed bilateral CP, those with term stroke unilateral CP, whereas after preterm stroke bilateral and unilateral CP occurred equally often. Information on the effects of sex and social economic status on outcome after specific brain lesions was very limited. Our findings show that the risk for CP is high after cPVL, moderate after preterm stroke, and lowest after term stroke. The risk for intellectual disability after an early brain lesion is lower than that for CP. Predicting outcome at individual level remains difficult; new imaging techniques may improve predicting developmental trajectories., (© 2016 The Authors. Developmental Medicine & Child Neurology © 2016 Mac Keith Press.)

Background: Children with cerebral palsy (CP) have impaired postural control. Posture is controlled in two levels: direction-specificity, and fine-tuning of direction-specific adjustments, including recruitment order. Literature suggests that direction-specificity might be a prerequisite for independent sitting., Aim: To study development of postural adjustments in infants at very high risk for CP (VHR-infants) during developing the ability to sit independently., Method: In a longitudinal study surface electromyograms of the neck-, trunk- and arm muscles of 11 VHR-infants and 11 typically developing (TD) infants were recorded during reaching in sitting before and after developing the ability to sit unsupported (median ages: VHR 8.0 and 14.9months; TD 5.7 and 10.4months). Sessions were video-recorded., Results: In VHR- and TD-infants the prevalence of direction-specific adjustments and recruitment order did not change when the infant learned to sit independently. In VHR-infants able to sit independently more successful reaching was associated with a higher frequency of bottom-up recruitment (Spearman's rho=0.828, p=0.006) and a lower frequency of simultaneous recruitment (Spearman's rho=-0.701, p=0.035), but not with more direction-specificity. In TD-infants not able to sit independently, more successful reaching was associated with higher rates of direction-specific adjustments at the neck level (Spearman's rho=0.778, p=0.014), but not with recruitment order., Conclusions: In VHR- and TD-infants postural adjustments during reaching in terms of direction-specificity and recruitment order are not related to development of independent sitting. Postural adjustments are associated with success of reaching, be it in a different way for VHR- and TD-infants., Clinical Trial Registration Number: NTR1428., (Copyright © 2014 Elsevier Ltd. All rights reserved.)

Purpose: To evaluate longitudinal applicability of the gross motor function measure (GMFM) in infants younger than 2 years., Methods: Twelve infants at very high risk for cerebral palsy were enrolled between 1 and 9 months corrected age. The children were assessed 4 times during 1 year with the GMFM-66, GMFM-88, and other neuromotor tests., Results: Longitudinal use of the GMFM in infancy was hampered by age and function-specific limitations. The GMFM-66 differentiated less at lower-ability levels than at higher-ability levels. The GMFM-88 demonstrated flattening of the developmental curve when infants had developed more motor abilities. We formulated adaptations for the longitudinal use of GMFM in infancy., Conclusions: To facilitate use of the GMFM in infancy, an adapted version may be an option. Further research is required to assess reliability and validity, and in particular, the sensitivity to change of the suggested adaptations.

Aim: The aim of this study was to examine the effects of intervention in infants at risk of developmental disorders on motor outcome, as measured by the Infant Motor Profile (IMP) and using the combined approach of a randomized controlled trial and process evaluation., Method: At a corrected age of 3 months, 46 infants (20 males, 26 females) recruited from the neonatal intensive care unit at the University Medical Centre Groningen (median birthweight 1210 g, range 585-4750 g; median gestational age 30 wks, range 25-40 wks) were included on the basis of definitely abnormal general movements. Exclusion criteria were severe congenital disorders and insufficient understanding of the Dutch language. The infants were assigned to either the family-centred COPing with and CAring for Infants with Special Needs (COPCA) intervention group (n=21; 9 males, 12 females) or the traditional infant physiotherapy (TIP) intervention group (n=25; 11 males, 14 females) for a period of 3 months. Three infants assigned to the TIP group (one male, two females) did not receive physiotherapy. IMP scores were measured by blinded assessors at 3, 4, 5, 6, and 18 months. At each age, the infants were neurologically examined. Physiotherapeutic sessions at 4 and 6 months were videotaped. Quantified physiotherapeutic actions were correlated with IMP scores at 6 and 18 months., Results: The IMP scores of both the COPCA and TIP groups before, during, and after the intervention did not differ. Some physiotherapeutic actions were associated with IMP outcomes; the associations differed for infants who developed cerebral palsy (n=10) and those who did not (n=33)., Interpretation: At randomized controlled trial level, the scores of both the TIP and COPCA groups did not differ in effect on motor outcome, as measured with the IMP. The analysis of physiotherapeutic actions revealed associations between these actions and IMP outcomes. However, the small sample size of this study precludes pertinent conclusions., (© The Authors. Journal compilation © Mac Keith Press 2011.)

Background: It is widely accepted that infants at risk for cerebral palsy need paediatric physiotherapy. However, there is little evidence for the efficacy of physiotherapeutic intervention. Recently, a new intervention program, COPCA (Coping with and Caring for infants with special needs - a family centered program), was developed. COPCA has educational and motor goals. A previous study indicated that the COPCA-approach is associated with better developmental outcomes for infants at high risk for developmental disorders. LEARN 2 MOVE 0-2 years evaluates the efficacy and the working mechanisms of the COPCA program in infants at very high risk for cerebral palsy in comparison to the efficacy of traditional infant physiotherapy in a randomized controlled trial. The objective is to evaluate the effects of both intervention programs on motor, cognitive and daily functioning of the child and the family and to get insight in the working elements of early intervention methods., Methods/design: Infants are included at the corrected age of 1 to 9 months and randomized into a group receiving COPCA and a group receiving traditional infant physiotherapy. Both interventions are given once a week during one year. Measurements are performed at baseline, during and after the intervention period and at the corrected age of 21 months. Primary outcome of the study is the Infant Motor Profile, a qualitative evaluation instrument of motor behaviour in infancy. Secondary measurements focus on activities and participation, body functions and structures, family functioning, quality of life and working mechanisms. To cope with the heterogeneity in physiotherapy, physiotherapeutic sessions are video-recorded three times (baseline, after 6 months and at the end of the intervention period). Physiotherapeutic actions will be quantified and related to outcome., Discussion: LEARN 2 MOVE 0-2 years evaluates and explores the effects of COPCA and TIP. Whatever the outcome of the project, it will improve our understanding of early intervention in children with cerebral palsy. Such knowledge is a prerequisite for tailor-made guidance of children with CP and their families., Trial Registration: The trial is registered under NTR1428.