Measures of Subjective Memory for People with Epilepsy: A Systematic Review of Measurement Properties.

People with epilepsy frequently express concern about the burden of memory problems in their everyday lives. Self-report memory questionnaires may provide valuable insight into individuals' perceptions of their everyday memory performance and changes over time. Yet, despite their potential utility, the measurement properties of self-report memory questionnaires have not been evaluated in epilepsy. This systematic review aimed to provide a critical appraisal of the measurement properties of self-report memory questionnaires for adults with epilepsy. Following protocol registration (PROSPERO CRD42020210967), a systematic search of PubMed, EMBASE, Web of Science, CINAHL, and PsychInfo from database inception until 27 May 2021 was conducted. Eligible studies were published in English-language peer-reviewed journals, recruited adults with epilepsy, and reported on the development or evaluation of the measurement properties of a self-report memory questionnaire. The COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) methodology was used to evaluate each study of a measurement property, and results were qualitatively synthesised. In total, 80 articles and one test manual were located containing 153 studies of measurement properties pertinent to 23 self-report memory questionnaires. Overall, no scale could be recommended outright for the evaluation of subjective memory symptoms in adults with epilepsy. This was due to the near absence of dedicated content validation studies relevant to this population and shortcomings in the methodology and scientific reporting of available studies of structural validity. Recommendations to support the advancement and psychometric validation of self-report memory questionnaires for people with epilepsy are provided.

Quality of life and neurological disability in children and young people with ataxia telangiectasia.

AIM: To explore neurological factors affecting quality of life (QoL) in children and young people with ataxia-telangiectasia (A-T), from both child and parent perspective. METHOD: 24 children/young people with A-T (mean age 11.2 ± 3.5 years; 13 males) and 20 parents were recruited, and 58% were reassessed after an average interval of 3.4 years. Participants completed the PedsQL QoL assessment. Participants with A-T underwent structured neurological examination. QoL data from 20 healthy controls and their parents was used for comparison. RESULTS: Children/young people with A-T rated their QoL higher than parental ratings across time points, with no longitudinal change. Higher age of the child participant correlated with lower parental (r = -0.43, p = .008) but not child ratings of QoL (r = -0.16, p = .380). Child and parent QoL ratings from the A-T group were lower than respective ratings from controls (ηp2 = 0.44 and ηp2 = 0.75 respectively, both p < .0005, controlled for socioeconomic status). Parental, but not child, ratings of QoL was predicted by a regression model based on neurological scores (R2 = 0.44, p=<.001). INTERPRETATION: Neurological disability does not determine child/young person QoL ratings in A-T. While certain aspects of neurological disability predict parent-proxy ratings, there is no decline in QoL over time. These results may reflect resilience in the face of a complex life-limiting disorder.

Correction: Ceiling effects in the Movement Assessment Battery for Children-2 (MABC-2) suggest that non-parametric scoring methods are required.

[This corrects the article DOI: 10.1371/journal.pone.0198426.].

Improving the Quality of Evaluation Data in Simulation-Based Healthcare Improvement Projects: A Practitioner's Guide to Choosing and Using Published Measurement Tools.

STATEMENT: Simulation is increasingly being used in healthcare improvement projects. The aims of such projects can be extremely diverse. Accordingly, the outcomes or participant attributes that need to be measured can vary dramatically from project-to-project and may include a wide range of nontechnical skills, technical skills, and psychological constructs. Consequently, there is a growing need for simulation practitioners to be able to identify suitable measurement tools and incorporate them into their work. This article provides a practical introduction and guide to the key considerations for practitioners when selecting and using such tools. It also offers a substantial selection of example tools, both to illustrate the key considerations in relation to choosing a measure (including reliability and validity) and to serve as a convenient resource for those planning a study. By making well-informed choices, practitioners can improve the quality of the data they collect, and the likelihood that their projects will succeed.

Multiparametric cerebellar imaging and clinical phenotype in childhood ataxia telangiectasia.

BACKGROUND: Ataxia Telangiectasia (A-T) is an inherited multisystem disorder with cerebellar neurodegeneration. The relationships between imaging metrics of cerebellar health and neurological function across childhood in A-T are unknown, but may be important for determining timing and impact of therapeutic interventions. PURPOSE: To test the hypothesis that abnormalities of cerebellar structure, physiology and cellular health occur in childhood A-T and correlate with neurological disability, we performed multiparametric cerebellar MRI and establish associations with disease status in childhood A-T. METHODS: Prospective cross-sectional observational study. 22 young people (9 females / 13 males, age 6.6-17.8 years) with A-T and 24 matched healthy controls underwent 3-Tesla MRI with volumetric, diffusion and proton spectroscopic acquisitions. Participants with A-T underwent structured neurological assessment, and expression / activity of ataxia-telangiectasia mutated (ATM) kinase were recorded. RESULTS: Ataxia-telangiectasia participants had cerebellar volume loss (fractional total cerebellar volume: 5.3% vs 8.7%, P < 0.0005, fractional 4th ventricular volumes: 0.19% vs 0.13%, P < 0.0005), that progressed with age (fractional cerebellar volumes, r = -0.66, P = 0.001), different from the control group (t = -4.88, P < 0.0005). The relationship between cerebellar volume and age was similar for A-T participants with absent ATM kinase production and those producing non-functioning ATM kinase. Markers of cerebellar white matter injury were elevated in ataxia-telangiectasia vs controls (apparent diffusion coefficient: 0.89 × 10-3 mm2 s-1 vs 0.69 × 10-3 mm2 s-1, p < 0.0005) and correlated (age-corrected) with neurometabolite ratios indicating impaired neuronal viability (N-acetylaspartate:creatine r = -0.70, P < 0.001); gliosis (inositol:creatine r = 0.50, P = 0.018; combined glutamine/glutamate:creatine r = -0.55, P = 0.008) and increased myelin turnover (choline:creatine r = 0.68, P < 0.001). Fractional 4th ventricular volume was the only variable retained in the regression model predicting neurological function (adjusted r2 = 0.29, P = 0.015). CONCLUSIONS: Quantitative MRI demonstrates cerebellar abnormalities in children with A-T, providing non-invasive measures of progressive cerebellar injury and markers reflecting neurological status. These MRI metrics may be of value in determining timing and impact of interventions aimed at altering the natural history of A-T.

Ceiling effects in the Movement Assessment Battery for Children-2 (MABC-2) suggest that non-parametric scoring methods are required.

Initially designed to identify children's movement impairments in clinical settings, the Movement Assessment Battery for Children-2 (MABC-2) is also widely used to evaluate children's movement in research. Standardised scores on the test are calculated using parametric methods under the assumption of normally-distributed data. In a pilot study with thirty five 8-10 year old children (i.e., in Age Band 2 of the MABC-2), we found that maximal performance was often reached. These 'ceiling effects' created distributions of scores that may violate parametric assumptions. Tests of normality, skew, and goodness-of-fit revealed this violation, most clearly on three of the eight sub-tests. A strong deviation from normality was again observed in a sample of 161 children (8-10 years, Experiment 1), however ceiling effects were reduced by modifying the scoring methods, and administering items designed for older children when maximal performance was reached. Experiment 2 (n = 81, 7-10 years) further refined the administration and scoring methods, and again improved the distributions of scores. Despite reducing ceiling effects, scores remained non-parametrically distributed, justifying non-parametric analytic approaches. By randomly and repeatedly resampling from the raw data, we generated non-parametric reference distributions for assigning percentiles to each child's performance, and compared the results with the standardised scores. Distributions of scores obtained with both parametric and non-parametric methods were skewed, and the methods resulted in different rankings of the same data. Overall, we demonstrate that some MABC-2 item scores are not normally-distributed, and violate parametric assumptions. Changes in administering and scoring may partially address these issues. We propose that resampling or other non-parametric methods are required to create new reference distributions to which an individual child's performance can be referred. The modifications we propose are preliminary, but the implication is that a new standardisation is required to deal with the non-parametric data acquired with the MABC-2 performance test.

Evaluation of an internet-based animated preparatory video for children undergoing non-sedated MRI.

OBJECTIVES: We evaluate the value of an internet-based educational animated video designed to prepare children for MRI scans, and whether this video reduces scan-related anxiety in children with a neurological disorder, and healthy controls. METHODS: Participants completed a pre- and post-scan questionnaire evaluating participant online viewing behaviour, understanding of the MRI procedure, anxiety regarding the MRI, impact of animation in preparing the child and whether the child's expectation of the MRI scan matched their experience. RESULTS: 21 children were recruited (12 healthy controls) ranging in age from 6.5 to 11.5 years. The animation was successfully accessed by participants on a range of digital devices and had high levels of approval. Children who viewed the animation had a good understanding of the MRI procedure and low anxiety levels prior to the scan, and reported that their expectations broadly matched the real-life MRI experience. Children reported that the animation positively impacted on their preparation with similar ratings before and after the scan, and the impact on preparation was rated greater by younger children. There were no group differences between healthy children and those with the neurological disorder for ratings of anxiety, impact on preparation and expectation of the experience. CONCLUSION: This evaluation demonstrates accessibility, acceptability and relevance of internet-based educational animation for typically developing children, and children with a neurodisability aged 6 to 11 years, with positive impact on preparation for MRI. Advances in knowledge: The internet-based educational animation provides a widely accessible tool to support preparation of children for non-sedated MRI.

Improvement in children's fine motor skills following a computerized typing intervention.

Children spend a large proportion of their school day engaged in tasks that require manual dexterity. If children experience difficulties with their manual dexterity skills it can have a consequential effect on their academic achievement. The first aim of this paper was to explore whether an online interactive typing intervention could improve children's scores on a standardised measure of manual dexterity. The second aim was to implement a serial reaction time tapping task as an index of children's finger movement learning, and to see whether performance on this task would improve after the intervention. Seventy-eight typically developing children aged between 8 and 10 were tested at their school on the pre-intervention Movement Assessment Battery for Children (2nd edition; MABC-2) and tapping tasks. Twenty-eight of these children volunteered to be randomly allocated to the intervention or control group. Children in the intervention group had a choice of two online games to play at home over a period of four weeks, while the children in the control group were not given these games to play. The intervention and control groups were then re-tested on the MABC-2 manual dexterity and the tapping task. Children in the intervention group significantly improved their manual dexterity scores in the MABC-2 compared to the control group. On average, all children learnt the tapping sequence, however, there were no group differences and no effect of the intervention on the tapping task. These results have important implications for implementing a freely available, easy to administer, fun and interactive intervention to help children improve their manual dexterity skills.

Online Control of Prehension Predicts Performance on a Standardized Motor Assessment Test in 8- to 12-Year-Old Children.

Goal-directed hand movements are guided by sensory information and may be adjusted 'online,' during the movement. If the target of a movement unexpectedly changes position, trajectory corrections can be initiated in as little as 100 ms in adults. This rapid visual online control is impaired in children with developmental coordination disorder (DCD), and potentially in other neurodevelopmental conditions. We investigated the visual control of hand movements in children in a 'center-out' double-step reaching and grasping task, and examined how parameters of this visuomotor control co-vary with performance on standardized motor tests often used with typically and atypically developing children. Two groups of children aged 8-12 years were asked to reach and grasp an illuminated central ball on a vertically oriented board. On a proportion of trials, and at movement onset, the illumination switched unpredictably to one of four other balls in a center-out configuration (left, right, up, or down). When the target moved, all but one of the children were able to correct their movements before reaching the initial target, at least on some trials, but the latencies to initiate these corrections were longer than those typically reported in the adult literature, ranging from 211 to 581 ms. These later corrections may be due to less developed motor skills in children, or to the increased cognitive and biomechanical complexity of switching movements in four directions. In the first group (n = 187), reaching and grasping parameters significantly predicted standardized movement scores on the MABC-2, most strongly for the aiming and catching component. In the second group (n = 85), these same parameters did not significantly predict scores on the DCDQ'07 parent questionnaire. Our reaching and grasping task provides a sensitive and continuous measure of movement skill that predicts scores on standardized movement tasks used to screen for DCD.