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Background: Angelman syndrome (AS) is a rare neurodevelopmental disorder characterized by severe intellectual disability, little to no expressive speech, visual and motor problems, emotional/behavioral challenges, and a tendency towards hyperphagia and weight gain. The characteristics of AS make it difficult to measure these children's functioning with standard clinical tests. Feasible outcome measures are needed to measure current functioning and change over time, in clinical practice and clinical trials. Aim: Our first aim is to assess the feasibility of several functional tests. We target domains of neurocognitive functioning and physical growth using the following measurement methods: eye-tracking, functional Near-Infrared Spectroscopy (fNIRS), indirect calorimetry, bio-impedance analysis (BIA), and BOD POD (air-displacement plethysmography). Our second aim is to explore the results of the above measures, in order to better understand the AS phenotype. Methods: The study sample consisted of 28 children with AS aged 2–18 years. We defined an outcome measure as feasible when (1) at least 70% of participants successfully finished the measurement and (2) at least 60% of those participants had acceptable data quality. Adaptations to the test procedure and reasons for early termination were noted. Parents rated acceptability and importance and were invited to make recommendations to increase feasibility. The results of the measures were explored. Results: Outcome measures obtained with eye-tracking and BOD POD met the definition of feasibility, while fNIRS, indirect calorimetry, and BIA did not. The most important reasons for early termination of measurements were showing signs of protest, inability to sit still and poor/no calibration (eye-tracking specific). Post-calibration was often applied to obtain valid eye-tracking results. Parents rated the BOD POD als most acceptable and fNIRS as least acceptable for their child. All outcome measures were rated to be important. Exploratory results indicated longer reaction times to high salient visual stimuli (eye-tracking) as well as high body fat percentage (BOD POD). Conclusions: Eye-tracking and BOD POD are feasible measurement methods for children with AS. Eye-tracking was successfully used to assess visual orienting functions in the current study and (with some practical adaptations) can potentially be used to assess other outcomes as well. BOD POD was successfully used to examine body composition. Trial registration: Registered d.d. 23-04-2020 under number 'NL8550' in the Dutch Trial Register: https://onderzoekmetmensen.nl/en/trial/23075 [ABSTRACT FROM AUTHOR]

This study aims to investigate distractibility quantified by recording and analyzing eye movements during task-irrelevant distraction in children with and without ADHD and in children with and without neurological disorders. Gaze behavior data and press latencies of 141 participants aged 6–17 that were collected during a computerized distraction paradigm with task-irrelevant stimuli (IDistrack) were analyzed. Children using attention-regulating medication were excluded from participation. Data were analyzed for subgroups that were formed based on the presence of neurological disorders and the presence of ADHD separately. Participants with ADHD and participants with neurological disorders spent less time fixating on the target stimuli compared to their peers without ADHD (p = 0.025) or their peers without neurological disorders (p < 0.001). Participants with and without ADHD had equal press latencies (p = 0.79). Participants with neurological disorders had a greater press latency compared to their typically developing peers (p < 0.001). Target fixation duration shows a significant association with parent-reported attention problems (r = −0.39, p < 0.001). We conclude that eye tracking during a distraction task reveals potentially valid clinical information that may contribute to the assessment of dysfunctional attentional processes. Further research on the validity and reliability of this paradigm is recommended. [ABSTRACT FROM AUTHOR]

Introduction: Children with early brain damage or dysfunction are at risk of developing cerebral visual impairment (CVI), including visual processing dysfunctions (VPD), which currently remain largely undetected until school age. Our aim was to systematically screen for possible VPD in children born very or extremely preterm from 1 to 2 years corrected age (CA) and to evaluate the effectiveness of early referral. Method: We included N = 48 children born < 30 weeks from 1 year CA. They underwent a two-step VPD screening based on (1) neurological signs indicative of visual brain damage evaluated by neonatologists and/or pediatric neurologist and (2) a functional assessment of visual orienting functions (VOF) with an eye tracking-based test. If at least one of these assessments was abnormal for their age, the children were classified as a risk of VPD and referred to undergo conventional visual diagnostics: ophthalmic exam and visual function assessment (VFA). At 2 years CA, VOF screening was repeated and neurodevelopment was assessed. Results: 18 children (38%) were classified as at risk of VPD at 1 year CA. 7 children had abnormal neurological signs, 5 children had abnormal VOF, and 6 children had both. Subsequent ophthalmic exams (N = 14) showed severe hypermetropia in 21% and strabismus in 14%. VFA (N = 10) showed abnormal visual function and behavior in only 1 child. At 2 years CA, the total group showed an increase in abnormal VOF. Whereas the children at risk showed some normalization, the group without VPD risk at 1 year CA showed deterioration of VOF. Neurodevelopmental outcome did not clearly differ between risk groups. Conclusion: Our findings show a substantial risk of VPD during visual screening (in 38%) at 1 year CA, but relatively few deficits on subsequent conventional ophthalmic exams and VFA. The data suggest that most conventional visual diagnostic methods at this young age are not related to the established VPD risks. VOF assessment should be used complimentary to these methods. The fact that at 2 years CA the number of children with a VPD risk based on abnormal VOF increased argues for more extensive and continuous screening in risk groups, at least until school age. [ABSTRACT FROM AUTHOR]

Aim: Children born extremely preterm are at risk of visual processing problems related to brain damage. Damage in visual pathways can remain undetected by conventional magnetic resonance imaging (MRI) and functional consequences cannot always be predicted. The aim of this study was to assess the efficacy of processing visual information in infants born extremely preterm at a corrected age of 1 year using a communication-free visual function test based on eye tracking.Method: Infants born extremely preterm (<29wks' gestation) without apparent white and grey matter damage on conventional MRI at 30 weeks' postmenstrual age were included (19 males, 1.01y [0.96-1.24] (median [25th-75th centiles]); 11 females, 0.99y [0.98-1.01]). At the corrected age of 1 year, reaction times to fixation (RTF) of specific visual properties displayed on an eye-tracker monitor were quantified and compared with results from a comparison group (eight males, 1.28y [1.01-1.33]; nine females, 1.10y [0.90-1.20]).Results: The infants in the preterm group had longer response times in detecting colour patterns (red-green) and motion compared with infants in the comparison group. No impairments were detected in oculomotor functions (saccades, pursuit, and fixations).Interpretation: The data suggest that delays in processing visual information can be identified in children born extremely preterm. The delays might be ascribed to deficits in neuronal connectivity in visual pathways at a microstructural level. [ABSTRACT FROM AUTHOR]

Purpose Measurements of visual and oculomotor functions are essential for providing tailored support to visually impaired children. In young or intellectually disabled children these measurements can be difficult or even impossible to perform. Recordings of orienting gaze in response to specific visual information, made with eye tracking, may offer a solution. The aim of this study was to observe and quantify eye tracking (ET)-based gaze responses to provide information about visual and oculomotor functioning, and to compare this information with standard visual function assessments (VFA). Methods One hundred and twenty-six visually impaired children from 1-14 years underwent a VFA. Next they underwent a remote ET test. Four aspects of oculomotor control (nystagmus, fixation, saccades, pursuit) and three visual functions (visual field, contrast, colour) were selected to compare both methods. Performance was assessed (1) during VFA using standard behavioural observation and test scores and (2) after ET by observing and scoring the eye movement recordings. Validity, in terms of agreement between results, was measured by correlation analyses. From the orienting gaze responses, quantitative parameters (gain, fixation duration and directional saccades) were calculated to characterize visual performance. Results Good agreement between the two test methods was found for observational assessment of oculomotor control and visual functions (correlations ranging from rs = 0.39 to rs = 0.69). The quantitative parameters of visual performance showed distinct results between children with and without specific functional impairments, both in children aged 1-6 and 7-14 years. Conclusion Eye tracking-based gaze recordings are a promising tool to assess oculomotor and visual performance in a communication-free manner. Calculating quantitative parameters from specific gaze responses could assist in the characterization of functional visual performance in children, independent of age. Gaze responses are a useful addition to standard VFA in clinical practice. [ABSTRACT FROM AUTHOR]

Background Displaying screen savers with gain-framed messages are effective to improve hand hygiene, but the design of screen savers has not been studied yet. Methods Based on the literature, scientific propositions were developed for the design of screen savers, exploring 2 strategies to subconsciously influence hand hygiene behavior; the first was to gain attention, and the second was to exert peer pressure. The designed screen savers were tested for attention with an eye-tracking study (N = 27) and for the influence of peer pressure with a questionnaire (N = 25). Results Twenty-five propositions for gaining attention concerned the format and color of the screen saver itself and color, position, and style of visual and text elements. Seven propositions for peer pressure concerned the influence of peers, role models, and feelings of being watched. Eye-tracking measurements showed that text on the 4 screen savers based on propositions gained more, earlier, and longer attention and the visual elements gained earlier and longer attention than the control screen savers. The questionnaire results showed that feelings of peer pressure were evoked by 3 screen savers; of these, one was not based on propositions. Conclusions Screen savers designed according to scientific propositions for visual attention and peer pressure have the potential to alter hand hygiene behavior. [ABSTRACT FROM AUTHOR]

Background: To provide insight into early neurosensory development in children born very preterm, we assessed the association between early structural brain damage and functional visuospatial attention and motion processing from one to two years corrected age.Methods: In 112 children born at less than 32 weeks gestational age, we assessed brain damage and growth with a standardized scoring system on magnetic resonance imaging (MRI; 1.5 Tesla) scans performed at 29 to 35 weeks gestational age. Of the children with an MRI scan, 82 participated in an eye tracking-based assessment of visuospatial attention and motion processing (Tobii T60XL) at one year corrected age and 59 at two years corrected age.Results: MRI scoring showed good intra- and inter-rater reproducibility. At one year, 10% children had delayed attentional reaction times and 23% had delayed motion reaction times. Moderate to severe brain damage significantly correlated with slower visuospatial reaction times. At two years, despite attention and motion reaction times becoming significantly faster, 20% had delayed attentional reaction times and 35% had delayed motion reaction times, but no correlations with MRI scores were found. The presence of structural brain damage was associated with abnormal functional performance over age.Conclusions: The present study indicates an association between moderate to severe brain damage and visuospatial attention and motion processing dysfunction at one year corrected age. This provides a new perspective on comprehensive MRI scoring and quantitative functional visuospatial assessments and their applicability in children born very preterm in their first years of life. [ABSTRACT FROM AUTHOR]

Abstract Background Peripheral and central visual processing development highly depends on the integrity of the visual sensory system and the allocation of visuospatial attention. Method We quantitatively followed visual sensory functions (VSF) and visuospatial orienting functions (VOF) over two years in 77 children (1–13 years) with different types of visual pathology. Results Within the clinical groups, VSF were relatively constant over two years, except visual acuity, and VOF were characterized by longer reaction time, shorter fixation duration, and lower fixation accuracy than normal for their age. Children with peripheral pathology had high rates of abnormal VSF, of changes to abnormal visual acuity at 1–6 years, and larger and more abnormal VOF (fixation inaccuracy). Children with central pathology had relatively good VSF, whereas two-third had delayed orienting reaction times that differed from other groups mainly at 1–6 years. Conclusion The distinct patterns of quantitative VSF and VOF over time between the visual pathology groups, and the finding that both methods provided complementary information, argues for combining both types of assessments to provide comprehensive monitoring of visual functioning in children from a young age. [ABSTRACT FROM AUTHOR]