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Background: The effects of unilateral cerebral palsy (UCP) are largely observed in the upper limb (UL), which represents the main focus of rehabilitation for this disorder. Thanks to an increment in home training and progress in technology innovative systems have been created. The Tele-UPCAT (Tele-monitored UPper Limb Children Action Observation Training) platform is dedicated to the delivery at home of a program for UL rehabilitation, based on action observation therapy (AOT)., Aim: This study aimed to investigate the immediate effectiveness of Tele-UPCAT for promoting UL skills in children with UCP and to determine if immediate effects were retained in the medium and long term., Design: Tele-UPCAT was conducted on an intention-to-treat basis and was proposed as a randomized, allocation concealed (waitlist controlled) and evaluator-blinded clinical trial with two investigative arms: intensive in-home AOT program and standard care (SC)., Setting: This is a home-based AOT program delivered with a customized ICT platform., Population: Thirty children (mean age 11.61±3.55 years) with confirmed diagnosis of spastic UCP with predominant UL involvement and cognitive level within or at normal limits were enrolled in this study. Orthopedic surgery or an intramuscular botulinum toxin A injection in the UL within 6 months prior to enrolment represented an exclusion criteria., Methods: Participants were randomized using concealed random allocation. They were assessed according to the study design with the Assisting Hand Assessment (AHA), the Box and Block Test (BBT) and the Melbourne Assessment 2 (MA2). Linear mixed models were used for statistical analysis., Results: A significant difference between the AOT and SC groups was identified immediately after the training on the AHA (6.406 [2.73] P=0.021) with an effect size (ES) of 1.99, and for the BBT of the less affected hand (9.826 [4.535] P=0.032) with an ES of 1.44. These effects were sustained at medium and long term., Conclusions: This study supports the effectiveness of AOT home training in promoting UL skills in children with UCP, with immediate effects lasting for 6 months., Clinical Rehabilitation Impact: This should encourage the use of technology for rehabilitative purposes and further applications of the AOT paradigm.

Aim: To evaluate the sensitivity, specificity, and predictive value of the Hand Assessment for Infants (HAI) in identifying infants at risk of being diagnosed with unilateral cerebral palsy (CP), and to determine cut-off values for this purpose., Method: A convenience sample of 203 infants (106 females, 97 males) was assessed by the HAI at 3, 6, 9, and 12 months. Sensitivity, specificity, predictive values, and likelihood ratios were calculated using receiver operating characteristic curve analysis. Cut-off values were derived for different ages. The clinical outcome (unilateral CP yes/no) at 24 months or more served as an external criterion to investigate the predictive validity of HAI., Results: Half of the infants developed unilateral CP. The area under the curve ranged from 0.77 (95% CI [confidence interval] 0.63-0.91) to 0.95 (95% CI 0.90-1.00) across HAI scales and age intervals. Likewise, sensitivity ranged from 63% to 93%, specificity from 62% to 91%, and accuracy from 73% to 94%., Interpretation: HAI scores demonstrated overall accuracy that ranged from very good to excellent in predicting unilateral CP in infants at risk aged between 3.5 and 12 months. This accuracy increased with age at assessment and the earliest possible prediction was at 3.5 months of age, when appropriate HAI cut-off values for different ages were applied. What this paper adds The Hand Assessment for Infants (HAI) predicts unilateral cerebral palsy (CP) with high accuracy. HAI cut-off values can guide clinical practice for early identification and diagnosis of unilateral CP., (© 2020 The Authors. Developmental Medicine & Child Neurology published by John Wiley & Sons Ltd on behalf of Mac Keith Press.)

Pre-term spastic diplegia (pSD) due to periventricular leukomalacia is a form of cerebral palsy in which weaknesses in executive functions are reported beyond the core visuo-spatial deficits. The study aimed at improving executive functioning and visuo-spatial skills with an evidence-based training focused on working memory in children with pSD. The intervention study followed a stepped wedge design. 19 children with pSD (11 female and 8 male; age range: 4;1-13;1 years), mild to moderate upper limb impairment and Verbal Intelligence Quotient (VIQ) >80 participated to the study. The children were trained with a home-based adaptive working memory training (CogMed®) over a 5-week period. The primary outcome measure was the CogMed Improvement index; pre- and post-test explorative neuropsychological assessment was conducted with a subset of tests from the NEPSY-II battery. Working memory training in children with pSD significantly improved trained working memory abilities (CogMed indices) as well as non-trained skills, such as visuo-spatial skills, inhibition of automatic responses and phonological processing. The results suggest that standard rehabilitation schedules for children with pSD should be integrated with trainings on executive functions., Competing Interests: The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest., (Copyright © 2021 Di Lieto, Pecini, Brovedani, Sgandurra, Dell'Omo, Chilosi, Guzzetta, Perazza, Sicola and Cioni.)

New rehabilitation programs based on action observation therapy (AOT) are effective in improving motor function in children with congenital hemiplegia. In this pilot study we tested the potential effects of AOT on the reorganization of the motor system by functional magnetic resonance imaging (fMRI). As part of a randomized trial, eight subjects (age range: 6.2-14.5 years) with congenital hemiplegia were randomly assigned to an experimental (EG) or control (CG) group. All children underwent a clinical and neurophysiological assessment with Assisting Hand Assessment (AHA), MRI, and fMRI at baseline (T0), 1(T1), and 8(T2) weeks after the end of 3-week treatment. For the EG, AOT consisted in the observation of uni/bimanual goal-directed actions followed by their execution. CG watched same-duration computer games and then performed the same actions in the same order used in the EG. fMRI study was carried out using two different paradigms, for exploring sensory-motor network (SMN) localization and action observation network (AON). The pattern of brain activation was generally similar between T0 and T1 for both groups, while it was more widespread at T2, compared to T0 and T1, in the EG. This enlargement was coupled with functional improvement at AHA. Single-subject analysis shows a reduction of lateralization indexes both for the AON and the SMN. This pilot study, despite the small sample, showed the fMRI feasibility for providing relevant biomarkers of brain plasticity for monitoring the AOT response in children with congenital hemiplegia. The study was registered at http://www.clinicaltrials.gov (identifier NCT01016496)., (© 2020 Wiley Periodicals, LLC.)

Background: To systematically review and analyse the effects of Action Observation Training on adults and children with brain damage., Methods: Seven electronic databases (Cochrane, EBSCO, Embase, Eric, PubMed, Scopus and Web of Science) were searched up to 16 September 2018 to select Randomized Controlled Trials focused on adults and children with brain damage that included AOT training on upper and/or lower limb carried out for at least 1 week. Identification of studies and data extraction was conducted with two reviewers working independently. Oxford Centre for Evidence-based Medicine (March2009) - Levels of Evidence and Physiotherapy Evidence Database scale were used to grade studies. The data collected from the articles were analysed using software R, version 3.4.3. Hedge's g values were calculated and effect size estimates were pooled across studies. Separate meta-analyses were carried out for each ICF domain (i.e. body function and activity) for upper and lower limb., Results: Out of the 210 records identified after removing duplicates, 22 were selected for systematic review and 19 were included in the meta-analysis. Thirteen studies included in the meta-analysis focused on upper limb rehabilitation (4 in children and 9 in adults) and 6 on lower limb rehabilitation (only studies in adults). A total of 626 patients were included in the meta-analysis. An overall statistically significant effect size was found for upper limb body function (0.44, 95% CI: [0.24, 0.64], p < 0.001) and upper limb activity domain (0.47, 95% CI: [0.30, 0.64], p < 0.001). For lower limb, only the activity domain was analysed, revealing a statistically significant overall effect size (0.56, 95% CI: [0.28, 0.84], p < 0.001)., Conclusions: Action Observation Training (AOT) is an innovative rehabilitation tool for individuals with brain damage, which shows promising results in improving the activity domain for upper and lower limbs, and also the body function domain for the upper limb. However, the examined studies lack uniformity and further well-designed, larger controlled trials are necessary to determine the most suitable type of AOT particularly in children., Systematic Review Registration: CRD42019119600.

Aim: To create normative reference values for unilateral and bilateral use of the hands, using the Hand Assessment for Infants (HAI), a newly developed criterion-referenced assessment measuring hand use in infants aged 3 months to 12 months at risk of cerebral palsy (CP)., Method: In total, 489 HAI assessments of typically developing infants (243 females, 246 males), aged 3 months to 10 months (mean 6mo 14d [SD 2mo 5d]), were collected in Italy and Sweden. Normative growth curves based on mean and SDs were created, as well as skill acquisition curves for each test item. Correlation to age and differences between groups based on sex and nationality, as well as differences between the right and the left hand, were investigated., Results: The growth curves showed a steady increase in mean value and a decrease in SD over age. There were no differences between groups based on sex or nationality. There was a negligible mean difference (0.1 raw score) between the right and left hands., Interpretation: HAI normative reference values are now available, which can assist in identifying deviating hand use for each month of age, as well as a side difference between hands in infants at risk of CP., What This Paper Adds: A Hand Assessment for Infants (HAI) result greater than 2SD below the mean indicates atypical hand use. Skill acquisition curves describe the age at which typically developing infants master the HAI items. Most typically developing infants do not demonstrate asymmetry in hand use., (© 2019 Mac Keith Press.)

Aim: To identify developmental trajectories of hand function in infants aged 3 months to 12 months with unilateral cerebral palsy (CP)., Method: Infants at high risk of unilateral CP were recruited from 3 months of age from follow-up programmes and clinics in Sweden, the Netherlands, Italy, and Australia. Measurements on the Hand Assessment for Infants (HAI) were completed until 12 months of age. Group-based trajectory modelling was used to identify subgroups of infants with similar trajectories of development. Multinomial logistic regression determined associations between demographic variables and trajectory membership., Results: Ninety-seven infants (52 males, 45 females; median gestational age 38wks [interquartile range 30-40wks]) were included. Infants were assessed between two and seven times (mean 4, SD 1.2) with a total of 387 observations. A three-group trajectory model identified a 'low-functioning group' (n=45: 46%), 'moderate-functioning group' (n=30: 31%), and 'high-functioning group' (n=22: 23%). Mean posterior probabilities (0.91-0.96) and odds of correct classification (26.3-33.2) indicated good model fit. Type of brain lesion, sex, side of hemiplegia, country, gestational age, and access to intensive intervention were not associated with group membership., Interpretation: Three trajectories of hand function development for infants with unilateral CP were identified and indicate some greater distinctions between groups with increasing age. The HAI is a valuable measure, capturing development of hand function of infants with unilateral CP over time., What This Paper Adds: Three distinct developmental trajectories of hand function in infants with unilateral cerebral palsy were identified. A low-functioning group made little progress in development of hand function in the first year of life. The degree of impairment on the impaired hand at 6 months of age is highly associated with trajectory membership. Infants with all types of brain lesion were represented across each trajectory group., (© 2018 Mac Keith Press.)

Background: Detecting differences in upper limb use in children with unilateral cerebral palsy (UCP) is challenging and highly dependent on examiner experience. The recent introduction of technologies in the clinical environment, and in particular the use of wearable sensors, can provide quantitative measurement to overcome this issue. This study aims to evaluate ActiGraph GT3X+ as a tool for measuring asymmetry in the use of the two upper limbs (ULs) during the assessment with a standardized clinical tool, the Assisting Hand Assessment (AHA) in UCP patients aged 3-25 years compared to age-matched typically developing (TD) subjects., Methods: Fifty children with UCP and 50 TD subjects were assessed with AHA while wearing ActiGraphs GT3X+ on both wrists. The mean activity of each hand (dominant and non-dominant, MA DH and MA NDH , respectively) and the asymmetry index (AI) were calculated. Two linear mixed model analyses were carried out to evaluate how dependent actigraphic variables (i.e. MA NDH and AI) varied by group (TD vs UCP) and among levels of manual ability based on Manual Ability Classification System (MACS). In both models age, sex, side of hemiplegia, presence/absence of mirror movements were specified as random effects., Results: The MA NDH was significantly lower in UCP compared to TD, while the AI was significantly higher in UCP compared to TD. Moreover, in UCP group there were significant differences related to MACS levels, both for MA NDH and AI. None of the random variables (i.e. age, sex, side, presence/absence of mirror movements) showed significant interaction with MA NDH and AI., Conclusions: These results confirm that actigraphy could provide, in a standardized setting, a quantitative description of differences between upper limbs activity., Trial Registration: ClincalTrials.gov, NCT03054441 . Registered 15 February 2017.

Little is known about the action observation network (AON) in children with unilateral cerebral palsy (UCP). Using fMRI, we aimed to explore AON and sensory-motor network (SMN) in UCP children and compare them to typically developed (TD) children and analyse the relationship between AON (re-)organization and several neurophysiological and clinical measures. Twelve UCP children were assessed with clinical scales and transcranial magnetic stimulation (TMS). For the fMRI study, they underwent a paradigm based on observation of complex and simple object-manipulation tasks executed by dominant and nondominant hand. Moreover, UCP and TD children carried out a further fMRI session to explore SMN in both an active motor and passive sensory task. AON in the UCP group showed higher lateralization, negatively related to performances on clinical scales, and had greater activation of unaffected hemisphere as compared to the bilateral representation in the TD group. In addition, a good congruence was found between bilateral or contralateral activation of AON and activation of SMN and TMS data. These findings indicate that our paradigm might be useful in exploring AON and the response to therapy in UCP subjects.

Introduction: A new rehabilitative approach, called UPper Limb Children Action Observation Training (UPCAT), based on the principles of action observation training (AOT), has provided promising results for upper limb rehabilitation in children with unilateral cerebral palsy (UCP). This study will investigate if a new information and communication technology platform, named Tele-UPCAT, is able to deliver AOT in a home setting and will test its efficacy on children and young people with UCP., Methods and Analysis: A randomised, allocation concealed (waitlist control) and evaluator-blinded clinical trial with two investigative arms will be carried out. The experimental group will perform AOT at home for 3 weeks using a customised Tele-UPCAT system where they will watch video sequences of goal-directed actions and then complete the motor training of the same actions. The control group will receive usual care for 3 weeks, which may include upper limb training. They will be offered AOT at home after 3 weeks. Twenty-four children with UCP will be recruited for 12 participants per group. The primary outcome will be measured using Assisting Hand Assessment. The Melbourne Assessment 2, ABILHAND, Participation and Environment Measure-Children and Youth and Cerebral Palsy Quality of Life Questionnaire will be included as secondary measures. Quantitative measures from sensorised objects and participants worn Actigraphs GXT3+ will be analysed. The assessment points will be the week before (T0) and after (T1) the period of AOT/standard care. Further assessments will be at T1 plus, the week after the AOT period for the waitlist group and at 8 weeks (T2) and 24 weeks (T3) after AOT training., Ethics and Dissemination: The trial has been approved by the Tuscany Paediatric Ethics Committee (169/2016). Publication of all outcomes will be in peer-reviewed journals and conference presentations., Trial Registration: NCT03094455., Competing Interests: Competing interests: None declared., (© Article author(s) (or their employer(s) unless otherwise stated in the text of the article) 2018. All rights reserved. No commercial use is permitted unless otherwise expressly granted.)

Aim: The aim of this study was to develop a descriptive and evaluative assessment of upper limb function for infants aged 3 to 12 months and to investigate its internal scale validity for use with infants at risk of unilateral cerebral palsy., Method: The concepts of the test items and scoring criteria were developed. Internal scale validity and aspects of reliability were investigated on the basis of 156 assessments of infants at 3 to 12 months corrected age (mean 7.2mo, SD 2.5) with signs of asymmetric hand use. Rasch measurement model analysis and non-parametric statistics were used., Results: The new test, the Hand Assessment for Infants (HAI), consists of 12 unimanual and five bimanual items, each scored on a 3-point rating scale. It demonstrated a unidimensional construct and good fit to the Rasch model requirements. The excellent person reliability enabled person separation to six significant ability strata. The HAI produced an interval-level measure of bilateral hand use as well as unimanual scores of each hand, allowing a quantification of possible asymmetry expressed as an asymmetry index., Interpretation: The HAI can be considered a valid assessment tool for measuring bilateral hand use and quantifying side difference between hands among infants at risk of developing unilateral cerebral palsy., What This Paper Adds: The Hand Assessment for Infants (HAI) measures the use of both hands and quantifies a possible asymmetry of hand use. HAI is valid for infants at 3 to 12 months corrected age at risk of unilateral cerebral palsy., (© 2017 Mac Keith Press.)

Background: The neuropsychological literature on preterm-born children with spastic diplegia due to periventricular leukomalacia is convergent in reporting deficits in non-verbal intelligence and in visuo-spatial abilities. Nevertheless, other cognitive functions have found to be impaired, but data are scant and not correlated with neuroimaging findings., Aims: This study analyzes the neuropsychological strengths and weaknesses in preterm-born children with spastic diplegia (pSD) and their relationships with neuroanatomical findings, investigated by a novel scale for MRI classification., Methods and Procedures: Nineteen children with pSD, mild to moderate upper limb impairment and Verbal IQ>80, and 38 normal controls were evaluated with a comprehensive neuropsychological battery (NEPSY-II), assessing Attention/Executive Functioning, Language, Memory, Sensorimotor, Social Perception and Visuospatial Processing domains. The MRIs were quantitatively scored for lesion severity., Outcomes and Results: The results showed that, beyond core visuo-spatial and sensory-motor deficits, impairments in attention and executive functions were present in more than half of the sample, particularly in children with damage to the anterior corpus callosum., Conclusions and Implications: The findings are discussed in terms of clinical and rehabilitative implications tailored for pSD subgroups diversified for neuropsychological and neuroanatomical characteristics., (Copyright © 2016 Elsevier Ltd. All rights reserved.)

Background: Preterm infants are at risk for neurodevelopmental disorders, including motor, cognitive or behavioural problems, which may potentially be modified by early intervention. The EU CareToy Project Consortium (http://www.caretoy.eu) has developed a new modular system for intensive, individualized, home-based and family-centred early intervention, managed remotely by rehabilitation staff. A randomised controlled trial (RCT) has been designed to evaluate the efficacy of CareToy training in a first sample of low-risk preterm infants., Methods/design: The trial, randomised, multi-center, evaluator-blinded, parallel group controlled, is designed according to CONSORT Statement. Eligible subjects are infants born preterm without major complications, aged 3-9 months of corrected age with specific gross-motor abilities defined by Ages & Stages Questionnaire scores. Recruited infants, whose parents will sign a written informed consent for participation, will be randomized in CareToy training and control groups at baseline (T0). CareToy group will perform four weeks of personalized activities with the CareToy system, customized by the rehabilitation staff. The control group will continue standard care. Infant Motor Profile Scale is the primary outcome measure and a total sample size of 40 infants has been established. Bayley-Cognitive subscale, Alberta Infants Motor Scale and Teller Acuity Cards are secondary outcome measures. All measurements will be performed at T0 and at the end of training/control period (T1). For ethical reasons, after this first phase infants enrolled in the control group will perform the CareToy training, while the training group will continue standard care. At the end of open phase (T2) all infants will be assessed as at T1. Further assessment will be performed at 18 months corrected age (T3) to evaluate the long-term effects on neurodevelopmental outcome. Caregivers and rehabilitation staff will not be blinded whereas all the clinical assessments will be performed, videotaped and scored by blind assessors. The trial is ongoing and it is expected to be completed by April 2015., Discussion: This paper describes RCT methodology to evaluate CareToy as a new tool for early intervention in preterm infants, first contribution to test this new type of system. It presents background, hypotheses, outcome measures and trial methodology., Trial Registration: ClinicalTrials.gov: NCT01990183. EU grant ICT-2011.5.1-287932.

The main goal of this study was to investigate the efficacy of Botulinum Toxin A (BoNT-A), combined with an individualized intensive physiotherapy/orthoses treatment, in improving upper limb activity and competence in daily activity in children with hemiplegia, and to compare its effectiveness with that of non-pharmacological instruments. It was a Randomized Clinical Trial of 27 children with spastic hemiplegic cerebral palsy, outpatients of two high speciality Centres for child rehabilitation. Each child was assigned by simple randomization to experimental group (BoNT-A) or control group (placebo). Assisting Hand Assessment (AHA) was chosen as primary outcome measure; other measures were selected according to ICF dimensions. Participants were assessed at baseline (T0), at T1, T2, T3 (1-3-6 months after injection, respectively). Every patient was given a specific physiotherapeutic treatment, consisting of individualized goal directed exercises, task oriented activities, daily stretching manoeuvres, functional and/or static orthoses. BoNT-A group showed a significant increase of AHA raw scores at T2, compared to control group (T2-T0: p=.025) and functional goals achievement (GAS) was also slightly better in the same group (p=.033). Other measures indicated some improvement in both groups, without significant intergroup differences. Children with intermediate severity of hand function at House scale for upper limb impairment seem to have a better benefit from BoNT-A protocol. BoNT-A was effective in improving manipulation in the activity domain, in association with individualized goal-directed physiotherapy and orthoses; the combined treatment is recommended. The study brings more evidence for the efficacy of a combined treatment botulinum toxin injection-physiotherapy-orthoses, and it gives some suggestions for candidate selection and individualized treatment., (Copyright © 2014 Elsevier Ltd. All rights reserved.)