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AbstractIndividuals with Williams syndrome (WS) have impairments in visuospatial tasks and in manual visuomotor control, consistent with parietal and cerebellar abnormalities. Here we examined whether individuals with WS also have difficulties in visually controlling whole‐body movements. We investigated visual control of stepping down at a change of level in children with WS (5–16‐year‐olds), who descended a single step while their movement was kinematically recorded. On each trial step height was set unpredictably, so that visual information was necessary to perceive the step depth and position the legs appropriately before landing. Kinematic measures established that children with WS did not use visual information to slow the leg at an appropriate point during the step. This pattern contrasts with that observed in typically developing 3‐ and 4‐year‐old children, implying severe impairment in whole‐body visuomotor control in WS. For children with WS, performance was not significantly predicted by low‐level visual or balance problems, but improved significantly with verbal age. The results suggest some plasticity and development in WS whole‐body control. These data clearly show that visuospatial and visuomotor deficits in WS extend to the locomotor domain. Taken together with evidence for parietal and cerebellar abnormalities in WS, these results also provide new evidence for the role of these circuits in the visual control of whole‐body movement.

Abstract Williams syndrome (WS) is a genetic disorder associated with severe visuocognitive impairment. Individuals with WS also report difficulties with everyday wayfinding. To study the development of body‐, environment‐, and object‐based spatial frames of reference in WS, we tested 45 children and adults with WS on a search task in which the participant and a spatial array are moved with respect to each other. Although individuals with WS showed a marked delay, like young controls they demonstrated independent, additive use of body‐ and environment‐based frames of reference. Crucially, object‐based (intrinsic) representations based on local landmarks within the array were only marginally used even by adults with WS, whereas in typical development these emerge at 5 years. Deficits in landmark use are consistent with wayfinding difficulties in WS, and may also contribute to problems with basic localization, since in typical development landmark‐based representations supplement those based on the body and on self‐motion. Difficulties with inhibition or mental rotation may be further components in the impaired ability to use the correct reference frame in WS.

AbstractIt has been suggested that learning an object's location relative to (1) intramaze landmarks and (2) local boundaries is supported by parallel striatal and hippocampal systems, both of which rely upon input from a third system for orientation. However, little is known about the developmental trajectories of these systems' contributions to spatial learning. The present study tested 5‐ and 7‐year‐old children and adults on a water maze‐like task in which all three types of cue were available. Participants had to remember the location of an object hidden in a circular bounded environment containing a moveable intramaze landmark and surrounded by distal cues. Children performed less accurately than adults, and showed a different pattern of error. While adults relied most on the stable cue provided by the boundary, children relied on both landmark and boundary cues similarly, suggesting a developmental increase in the weighting given to boundary cues. Further, adults were most accurate in coding angular information (dependent on distal cues), whereas children were most accurate in coding distance, suggesting a developing ability to use distal cues to orient. These results indicate that children as young as 5 years use boundary, intramaze landmark, and distal visual cues in parallel, but that the basic accuracy and relative weighting of these cues changes during subsequent development.

AbstractWe report asymmetrical cortical responses (steady‐state visual evoked potentials) to radial expansion and contraction in human infants and adults. Forty‐four infants (22 3‐month‐olds and 22 4‐month‐olds) and nine adults viewed dynamic dot patterns which cyclically (2.1 Hz) alternate between radial expansion (or contraction) and random directional motion. The first harmonic (F1) response in the steady‐state VEP response must arise from mechanisms sensitive to the global radial motion structure. We compared F1 amplitudes between expansion‐random and contraction‐random motion alternations. F1 amplitudes for contraction were significantly larger than those for expansion for the older infants and adults but not for the younger infants. These results suggest that the human cortical motion mechanisms have asymmetrical sensitivity for radial expansion vs. contraction, which develops at around 4 months of age. The relation between development of sensitivity to radial motion and cortical motion mechanisms is discussed.