TY - JOUR
T1 - Cross-sectional and longitudinal assessment of cognitive development in Williams syndrome
AU - Farran, Emily K
AU - Purser, Harry R M
AU - Jarrold, Christopher
AU - Thomas, Michael
AU - Scerif, Gaia
AU - Stojanovik, Vesna
AU - Van Herwegen, Jo
N1 - Funding Information:
Thank you to the parents and individuals with WS and to the Williams Syndrome Foundation, UK. Thank you to lab members who contributed to the legacy data set. This research was supported by funding from the Williams Syndrome Foundation UK to Jo Van Herwegen and Michael Thomas. Legacy data were collected under multiple grants to the authors.
Publisher Copyright:
© 2023 The Authors. Developmental Science published by John Wiley & Sons Ltd.
PY - 2023/6/7
Y1 - 2023/6/7
N2 - Research HighlightsCross-sectional and longitudinal developmental trajectories of verbal and non-verbal development, in the largest sample of individuals with Williams syndrome (WS) to-date, are presented.This research is unique due to large sample sizes, independence between cross-sectional and longitudinal samples, and a reliance on minimum three timepoints for longitudinal trajectories. Data support the WS cognitive profile of stronger verbal than non-verbal ability and a developmental model of delayed onset and a delayed rate. Cross-sectional and longitudinal data are discussed with reference to validating cross-sectional developmental patterns using longitudinal data and the importance of individual differences in understanding development. AbstractWilliams syndrome (WS) is a rare genetic syndrome. As with all rare syndromes, obtaining adequately powered sample sizes is a challenge. Here we present legacy data from seven UK labs, enabling the characterisation of cross-sectional and longitudinal developmental trajectories of verbal and non-verbal development in the largest sample of individuals with WS to-date. In Study 1, we report cross-sectional data from between N=102 and N=209 children and adults with WS on measures of verbal and non-verbal ability. In Study 2, we report longitudinal data from N=17 to N=54 children and adults with WS who had been tested on at least three timepoints on these measures. Data support the WS characteristic cognitive profile of stronger verbal than non-verbal ability, and shallow developmental progression for both domains. Both cross-sectional and longitudinal data demonstrate steeper rates of development in the child participants than the adolescent and adults in our sample. Cross-sectional data indicate steeper development in verbal than non-verbal ability, and that individual differences in the discrepancy between verbal and non-verbal ability are largely accounted for by level of intellectual functioning. A diverging developmental discrepancy between verbal and non-verbal ability, whilst marginal, is not mirrored statistically in the longitudinal data. Cross-sectional and longitudinal data are discussed with reference to validating cross-sectional developmental patterns using longitudinal data and the importance of individual differences in understanding developmental progression.
AB - Research HighlightsCross-sectional and longitudinal developmental trajectories of verbal and non-verbal development, in the largest sample of individuals with Williams syndrome (WS) to-date, are presented.This research is unique due to large sample sizes, independence between cross-sectional and longitudinal samples, and a reliance on minimum three timepoints for longitudinal trajectories. Data support the WS cognitive profile of stronger verbal than non-verbal ability and a developmental model of delayed onset and a delayed rate. Cross-sectional and longitudinal data are discussed with reference to validating cross-sectional developmental patterns using longitudinal data and the importance of individual differences in understanding development. AbstractWilliams syndrome (WS) is a rare genetic syndrome. As with all rare syndromes, obtaining adequately powered sample sizes is a challenge. Here we present legacy data from seven UK labs, enabling the characterisation of cross-sectional and longitudinal developmental trajectories of verbal and non-verbal development in the largest sample of individuals with WS to-date. In Study 1, we report cross-sectional data from between N=102 and N=209 children and adults with WS on measures of verbal and non-verbal ability. In Study 2, we report longitudinal data from N=17 to N=54 children and adults with WS who had been tested on at least three timepoints on these measures. Data support the WS characteristic cognitive profile of stronger verbal than non-verbal ability, and shallow developmental progression for both domains. Both cross-sectional and longitudinal data demonstrate steeper rates of development in the child participants than the adolescent and adults in our sample. Cross-sectional data indicate steeper development in verbal than non-verbal ability, and that individual differences in the discrepancy between verbal and non-verbal ability are largely accounted for by level of intellectual functioning. A diverging developmental discrepancy between verbal and non-verbal ability, whilst marginal, is not mirrored statistically in the longitudinal data. Cross-sectional and longitudinal data are discussed with reference to validating cross-sectional developmental patterns using longitudinal data and the importance of individual differences in understanding developmental progression.
U2 - 10.1111/desc.13421
DO - 10.1111/desc.13421
M3 - Article (Academic Journal)
C2 - 37287370
SN - 1363-755X
VL - 27
JO - Developmental Science
JF - Developmental Science
IS - 1
M1 - e13421
ER -