Assessing the Psychometric Properties of the Illness Management and Recovery Scale: A Systematic Review Using the Consensus-Based Standards for the Selection of Health Measurement Instruments (COSMIN).

The Illness Management and Recovery Scale (IMR-S) is based on the IMR program, developed to assess the recovery process for people with severe mental disorders by considering the perceptions of clients and clinicians involved in it. The aim of this study was to analyze the psychometric properties of the IMR-S so as to determine the reliability and suitability of its scores for evaluating recovery. Two coders searched five databases for studies, published between January 2004 and May 2023, that describe the psychometric assessment of the IMR-S. Studies were assessed using the COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) Risk of Bias checklist. Finally, 46 papers were included. Methodological quality was very good for most of the studies that provide information on internal validity, and limited for those that report on responsiveness. Measurement properties were positive for convergent validity and measurement error. The quality of evidence was high for structural validity studies. Although this study only includes research published in English and may have overlooked certain psychometric properties evaluated in studies published in other languages, our findings suggest that the IMR-S is a valid and reliable instrument, demonstrating its potential to offer guidance for clinical practice.

A randomized controlled trial of a home-based computerized executive function intervention for children with cerebral palsy.

Children with cerebral palsy (CP) often show executive function (EF) impairments that are key to quality of life. The aim of this study was to assess whether a home-based computerized intervention program improves executive functions (EFs) compared to usual care. Sixty participants (30 females) with CP (8-12 years old) were paired by age, sex, motor ability, and intelligence quotient score and then randomized to intervention and waitlist control groups. The intervention group received a 12-week home-based computerized EF intervention (5 days/week, 30 min/day, total dose 30 h). Core and higher-order EFs were assessed before, immediately after, and 9 months after completing the intervention. The intervention group performed better than the waitlist control group in the three core EFs (immediately and 9 months after the intervention): inhibitory control (F = 7.58, p = 0.13 and F = 7.85, p = 0.12), working memory (F = 8.34, p = 0.14 and F = 7.55, p = 0.13), and cognitive flexibility (F = 4.87, p = 0.09 and F = 4.19, p = 0.08). No differences were found between the groups in higher-order EFs or EF manifestations in daily life. CONCLUSIONS: A home-based computerized EF intervention improved core EFs in children with CP, but further research is needed to identify strategies that allow the transfer of these improvements to everyday life. TRIAL REGISTRATION: NCT04025749 retrospectively registered on 19 July 2019. WHAT IS KNOWN: • One in two children with cerebral palsy has an intellectual impairment. Visual perception and executive functions are the most reported specific cognitive deficits. • The majority of interventions for cerebral palsy focus on motor impairments, but only a few randomized controlled trials have explored the effect of interventions on executive functions. WHAT IS NEW: • A home-based computerized cognitive intervention can improve the core executive functions of children with cerebral palsy. • Short- and long-term effects on core executive functions have been found.

Factors Related to Quality of Life in Children With Cerebral Palsy.

BACKGROUND: We investigated the influence of relevant demographic, clinical, neuropsychological, and psychosocial variables on the proxy-reported quality of life (QOL) of children with cerebral palsy (CP). METHODS: The proxy-reported Cerebral Palsy Quality of Life-Child questionnaire (CP QOL-Child) was completed by 58 children with CP (mean age 10.22 years, SD 1.67). Relationships between QOL scores and demographic, clinical, neuropsychological, and psychosocial variables were assessed. CP QOL scores and other variables that correlated significantly were introduced into a multiple linear regression model. RESULTS: Executive functioning and motor functional status were explanatory variables for the CP QOL total score. Executive functions explained three specific QOL domains: Social Wellbeing and Acceptance, Feelings about Functioning, and Emotional Wellbeing and Self-esteem. Parental stress also explained Social Wellbeing and Acceptance. Motor functional status and visual perception were explanatory variables for the Access to Services domain. Finally, autism spectrum disorder (ASD) traits were an explanatory variable for the Participation and Physical Health domain. CONCLUSION: Executive functioning and motor functional status importantly influence QOL of children with CP. Visual perception, ASD symptoms, and parental stress variables are related with specific QOL domains. These findings demonstrate that interventions targeting cognitive functions in children with CP may positively influence QOL.

Interventions with an Impact on Cognitive Functions in Cerebral Palsy: a Systematic Review.

This systematic review aimed at investigating those interventions that impact on cognitive functioning in children and adults with cerebral palsy (CP). A systematic database search was conducted and twenty-eight studies suitable for inclusion were identified, of which only nine were randomized controlled trials (RCTs). Among all the studies included, ten were multi-modal (cognitive and physical tasks), eleven physical, five cognitive, and two alternative and augmentative communication interventions. The evidence suggests that multi-modal and physical interventions improve general cognitive functioning. Multi-modal and cognitive interventions have an impact on visual perception. Both interventions, together with physical interventions have an effect on a specific executive function domain (inhibitory control), and only cognitive interventions improved other executive function domains such as working memory. However, no RCT assessed the effects of all executive function domains. Few studies have looked at interventions to improve memory and language, and there is a scarcity of long-term research. Future RCTs must be of higher quality and better account for age and sex differences, as well as the clinical heterogeneity of CP. To date, there is evidence that multi-modal, cognitive or physical interventions have an impact on general cognitive functioning, visual perception and executive functions in children with CP, which may support their cognitive development.The protocol was registered with the International Prospective Register of Systematic Reviews (PROSPERO): CRD42020152616.

Study protocol of a randomized controlled trial of home-based computerized executive function training for children with cerebral palsy.

BACKGROUND: Cerebral palsy (CP) is frequently associated with specific cognitive impairments, such as executive dysfunction which are related to participation and quality of life (QOL). The proposed study will examine whether a computerized executive function (EF) training programme could provide superior benefits for executive functioning, participation, QOL and brain plasticity, as compared to usual care. METHODS: A single-blind randomized controlled trial (RCT) design will be performed. Thirty children with CP aged 8 to 12 years will participate in a home-based computerized multi-modal executive training programme (12 weeks, 5 days a week, 30 min a day training, total dose = 30 h). Thirty children with CP matched by age, sex, motor and intelligence quotient (IQ) will compose the waitlist group. Cognitive, behavioural, emotional, participation and QOL measures will be obtained at three time points: before, immediately after and 9 months after completing the training. Additionally, structural and functional (resting state) magnetic resonance images (MRI) will be obtained in a subsample of 15 children from each group. Outcomes between groups will be compared following standard principles for RCTs. DISCUSSION: The study will test whether the cognitive training programme exerts a positive effect not only on neuropsychological and daily functioning of children with CP but also on other measures such as participation and QOL. We will also use brain MRI to test brain functional and structural changes after the intervention. If this on-line and home-based training programme proves effective, it could be a cost-effective intervention with short- and long-term effects on EF, participation or QOL in CP. TRIAL REGISTRATION: ClinicalTrials.gov: NCT04025749. Registered 19 July 2019. Retrospectively registered.

Executive function and general intellectual functioning in dyskinetic cerebral palsy: Comparison with spastic cerebral palsy and typically developing controls.

AIM: To comprehensively describe intellectual and executive functioning (EF) in people with dyskinetic cerebral palsy (DCP), by comparing their performance with that of: 1) age- and sex-matched typically developing controls (TDC); and 2) participants with spastic cerebral palsy (SCP) matched for age, sex, term/preterm and gross motor function classification system (GMFCS). METHOD: This cross-sectional study was conducted by the University of Barcelona in collaboration with five institutions. Participants were people with DCP (n = 52; 24 females, median age 20.5 y: 5mo, interquartile range [IQR] = 13.75 y: 7mo; GMFCS I-V). As comparison groups, participants with SCP (n = 20; 10 females, median age = 20.5 y: 5.5mo, IQR = 13.75 y 9mo; GMFCS I-V) and TDC (n = 52; 24 females, median age = 20 y: 4mo, IQR = 12 y 7mo) were included. Intelligence and EF were assessed using common tests in all participants. RESULTS: Both CP groups had lower intelligence than TDC and performed poorer in almost all EF tasks. Intelligence was higher in DCP than SCP (z = -2.51, p = 0.01). Participants with DCP also performed significantly better in goal-setting tasks (z = 2.27, p = 0.03) and information processing (z = -2.54, p = 0.01) than those with SCP. CONCLUSION: People with DCP present lower general intellectual functioning and poorer EF across multiple domains than typically developing controls. People with DCP have higher general intellectual functioning and better EF than people with SCP when levels of motor severity are similar.

Brain lesion scores obtained using a simple semi-quantitative scale from MR imaging are associated with motor function, communication and cognition in dyskinetic cerebral palsy.

Purpose: To characterise brain lesions in dyskinetic cerebral palsy (DCP) using the semi-quantitative scale for structural MRI (sqMRI) and to investigate their relationship with motor, communication and cognitive function. Materials and methods: Thirty-nine participants (19 females, median age 21y) with DCP were assessed in terms of motor function, communication and a variety of cognitive domains. Whole-head magnetic resonance imaging (MRI) was performed including T1-MPRAGE, T2 turbo spin echo (axial plane), and fluid attenuated inversion recovery images (FLAIR). A child neurologist visually assessed images for brain lesions and scored these using the sqMRI. Ordinal, Poisson and binomial negative regression models identified which brain lesions accounted for clinical outcomes. Results: Brain lesions were most frequently located in the ventral posterior lateral thalamus and the frontal lobe. Gross (B = 0.180, p < .001; B = 0.658, p < .001) and fine (B = 0.136, p = .003; B = 0.540, p < .001) motor function were associated with global sqMRI score and parietal involvement. Communication functioning was associated with putamen involvement (B = 0.747, p < .028). Intellectual functioning was associated with global sqMRI score and posterior thalamus involvement (B = -0.018, p < .001; B = -0.192, p < .001). Selective attention was associated with global sqMRI score (B = -0.035, p < .001), parietal (B = -0.063, p = .023), and corpus callosum involvement (B = -0.448, p < .001). Visuospatial and visuoperceptive abilities were associated with global sqMRI score (B = -0.078, p = .007) and medial dorsal thalamus involvement (B = -0.139, p < .012), respectively. Conclusions: Key clinical outcomes in DCP are associated with specific observable brain lesions as indexed by a simple lesion scoring system that relies only on standard clinical MRI.

Cognitive functioning in dyskinetic cerebral palsy: Its relation to motor function, communication and epilepsy.

BACKGROUND: Cerebral palsy (CP) is a disorder of motor function often accompanied by cognitive impairment. There is a paucity of research focused on cognition in dyskinetic CP and on the potential effect of related factors. AIM: To describe the cognitive profile in dyskinetic CP and to assess its relationship with motor function and associated impairments. METHOD: Fifty-two subjects with dyskinetic CP (28 males, mean age 24 y 10 mo, SD 13 y) and 52 typically-developing controls (age- and gender-matched) completed a comprehensive neuropsychological assessment. Gross Motor Function Classification System (GMFCS), Communication Function Classification System (CFCS) and epilepsy were recorded. Cognitive performance was compared between control and CP groups, also according different levels of GMFCS. The relationship between cognition, CFCS and epilepsy was examined through partial correlation coefficients, controlling for GMFCS. RESULTS: Dyskinetic CP participants performed worse than controls on all cognitive functions except for verbal memory. Milder cases (GMFCS I) only showed impairment in attention, visuoperception and visual memory. Participants with GMFCS II-III also showed impairment in language-related functions. Severe cases (GMFCS IV-V) showed impairment in intelligence and all specific cognitive functions but verbal memory. CFCS was associated with performance in receptive language functions. Epilepsy was related to performance in intelligence, visuospatial abilities, visual memory, grammar comprehension and learning. CONCLUSION: Cognitive performance in dyskinetic CP varies with the different levels of motor impairment, with more cognitive functions impaired as motor severity increases. This study also demonstrates the relationship between communication and epilepsy and cognitive functioning, even controlling for the effect of motor severity.

White matter integrity in dyskinetic cerebral palsy: Relationship with intelligence quotient and executive function.

BACKGROUND: Dyskinetic cerebral palsy (CP) is one of the most disabling motor types of CP and has been classically associated with injury to the basal ganglia and thalamus. Although cognitive dysfunction is common in CP, there is a paucity of published quantitative analyses investigating the relationship between white matter (WM) microstructure and cognition in this CP type. AIMS: This study aims (1) to compare brain WM microstructure between people with dyskinetic CP and healthy controls, (2) to identify brain regions where WM microstructure is related to intelligence and (3) to identify brain regions where WM microstructure is related to executive function in people with dyskinetic CP and (4) to identify brain regions where the correlations are different between controls and people with CP in IQ and executive functions. PATIENTS AND METHODS: Thirty-three participants with dyskinetic CP (mean ± SD age: 24.42 ± 12.61, 15 female) were age and sex matched with 33 controls. Participants underwent a comprehensive neuropsychological battery to assess intelligence quotient (IQ) and four executive function domains (attentional control, cognitive flexibility, goal setting and information processing). Diffusion weighted MRI scans were acquired at 3T. Voxel-based whole brain groupwise analyses were used to compare fractional anisotropy (FA) and of the CP group to the matched controls using a general lineal model. Further general linear models were used to identify regions where white matter FA correlated with IQ and each of the executive function domains. RESULTS: White matter FA was significantly reduced in the CP group in all cerebral lobes, predominantly in regions connected with the parietal and to a lesser extent the temporal lobes. There was no significant correlation between IQ or any of the four executive function domains and WM microstructure in the control group. In participants with CP, lower IQ was associated with lower FA in all cerebral lobes, predominantly in locations that also showed reduced FA compared to controls. Attentional control, goal setting and information processing did not correlate with WM microstructure in the CP group. Cognitive flexibility was associated with FA in regions known to contain connections with the frontal lobe (such as the superior longitudinal fasciculus and cingulum) as well as regions not known to contain tracts directly connected with the frontal lobe (such as the posterior corona radiata, posterior thalamic radiation, retrolenticular part of internal capsule, tapetum, body and splenium of corpus callosum). CONCLUSION: The widespread loss in the integrity of WM tissue is mainly located in the parietal lobe and related to IQ in dyskinetic CP. Unexpectedly, executive functions are only related with WM microstructure in regions containing fronto-cortical and posterior cortico-subcortical pathways, and not being specifically related to the state of fronto-striatal pathways which might be due to brain reorganization. Further studies of this nature may improve our understanding of the neurobiological bases of cognitive impairments after early brain insult.

Whole-brain structural connectivity in dyskinetic cerebral palsy and its association with motor and cognitive function.

Dyskinetic cerebral palsy (CP) has long been associated with basal ganglia and thalamus lesions. Recent evidence further points at white matter (WM) damage. This study aims to identify altered WM pathways in dyskinetic CP from a standardized, connectome-based approach, and to assess structure-function relationship in WM pathways for clinical outcomes. Individual connectome maps of 25 subjects with dyskinetic CP and 24 healthy controls were obtained combining a structural parcellation scheme with whole-brain deterministic tractography. Graph theoretical metrics and the network-based statistic were applied to compare groups and to correlate WM state with motor and cognitive performance. Results showed a widespread reduction of WM volume in CP subjects compared to controls and a more localized decrease in degree (number of links per node) and fractional anisotropy (FA), comprising parieto-occipital regions and the hippocampus. However, supramarginal gyrus showed a significantly higher degree. At the network level, CP subjects showed a bilateral pathway with reduced FA, comprising sensorimotor, intraparietal and fronto-parietal connections. Gross and fine motor functions correlated with FA in a pathway comprising the sensorimotor system, but gross motor also correlated with prefrontal, temporal and occipital connections. Intelligence correlated with FA in a network with fronto-striatal and parieto-frontal connections, and visuoperception was related to right occipital connections. These findings demonstrate a disruption in structural brain connectivity in dyskinetic CP, revealing general involvement of posterior brain regions with relative preservation of prefrontal areas. We identified pathways in which WM integrity is related to clinical features, including but not limited to the sensorimotor system. Hum Brain Mapp 38:4594-4612, 2017. © 2017 Wiley Periodicals, Inc.

Measuring intellectual ability in cerebral palsy: The comparison of three tests and their neuroimaging correlates.

Standard intelligence scales require both verbal and manipulative responses, making it difficult to use in cerebral palsy and leading to underestimate their actual performance. This study aims to compare three intelligence tests suitable for the heterogeneity of cerebral palsy in order to identify which one(s) could be more appropriate to use. Forty-four subjects with bilateral dyskinetic cerebral palsy (26 male, mean age 23 years) conducted the Raven's Coloured Progressive Matrices (RCPM), the Peabody Picture Vocabulary Test-3rd (PPVT-III) and the Wechsler Nonverbal Scale of Ability (WNV). Furthermore, a comprehensive neuropsychological battery and magnetic resonance imaging were assessed. The results show that PPVT-III gives limited information on cognitive performance and brain correlates, getting lower intelligence quotient scores. The WNV provides similar outcomes as RCPM, but cases with severe motor impairment were unable to perform it. Finally, the RCPM gives more comprehensive information on cognitive performance, comprising not only visual but also verbal functions. It is also sensitive to the structural state of the brain, being related to basal ganglia, thalamus and white matter areas such as superior longitudinal fasciculus. So, the RCPM may be considered a standardized easy-to-administer tool with great potential in both clinical and research fields of bilateral cerebral palsy.

Does verbal and gestural expression ability predict comprehension ability in cerebral palsy?

Some people with cerebral palsy have motor and associated impairments that may hinder verbal and gestural expression to various extents. This study explores whether the ability to produce verbal or gestural expressions may be related to the comprehension of verbal communications and gestures. The influence of severity of motor impairment, general cognitive performance, and age on comprehension ability was also explored. Forty people with cerebral palsy were assigned to different groups according to their verbal and gestural expression abilities. A neuropsychological assessment of comprehension abilities and general cognitive performance was carried out. Multiple linear regression analysis was applied to identify the possible influence of expression abilities on comprehension abilities and also to detect the possible contribution of severity of motor impairment, general cognitive performance, and age. Results indicate that verbal and gestural comprehension was mainly predicted by general cognitive performance. Severity of motor impairment and age did not contribute to predicting comprehension abilities. Only verbal grammar comprehension was significantly predicted by verbal expression ability. Verbal expression ability may be an important marker for cerebral palsy therapies. In non-ambulant patients with bilateral cerebral palsy, impaired gestural expression should not be taken as an indicator of impaired gestural comprehension.