Learning to read in Williams syndrome and Down syndrome: syndrome-specific precursors and developmental trajectories.

BACKGROUND In typical development, early reading is underpinned by language skills, like vocabulary and phonological awareness (PA), as well as taught skills like letter knowledge. Less is understood about how early reading develops in children with neurodevelopmental disorders who display specific profiles of linguistic strengths and weaknesses, such as Down syndrome (DS) and Williams syndrome (WS). METHODS Early reading, letter knowledge, rhyme matching, phoneme matching and receptive vocabulary were assessed in 26 children with DS and 26 children with WS between 4 and 8 years, as well as in two groups of typically developing (TD) children matched on nonverbal mental age (NVMA controls) or reading (RA controls). Reading was also measured 1 year later in DS, WS and RA controls to assess reading growth and its longitudinal predictors. RESULTS Despite poor PA and vocabulary, children with DS displayed good reading and letter knowledge, compared with NVMA controls. Performance of children with WS was equivalent to RA controls and superior to NVMA controls on all tasks. Longitudinal delays emerged in reading in both DS and WS compared with RA controls. Vocabulary was a significant longitudinal predictor of reading growth for all children, but, for both children with DS and WS, and unlike RA controls, letter knowledge and PA were not. CONCLUSIONS Children with DS and WS display atypical developmental patterns in the earliest stages of reading, further underlining the importance of cross-syndrome, longitudinal research, which tracks all levels of development in neurodevelopmental disorders. Identifying early syndrome-specific profiles of strengths and weaknesses underlying literacy development is critical for planning intervention programmes.

[1]  Gaia Scerif,et al.  The multiple subfunctions of attention: differential developmental gateways to literacy and numeracy. , 2012, Child development.

[2]  Charles Hulme,et al.  The growth of reading skills in children with Down Syndrome. , 2012, Developmental science.

[3]  C. Mervis,et al.  Precursors to language and early language , 2011 .

[4]  K. Eklund,et al.  Language Development, Literacy Skills, and Predictive Connections to Reading in Finnish Children With and Without Familial Risk for Dyslexia , 2010, Journal of learning disabilities.

[5]  A. Meyer,et al.  The evolutionary significance of ancient genome duplications , 2009, Nature Reviews Genetics.

[6]  Cláudia Cardoso-Martins,et al.  Component reading skills in Down Syndrome , 2009 .

[7]  Christopher Jarrold,et al.  A comparison between word and nonword reading in Down syndrome: the role of phonological awareness. , 2008, Journal of communication disorders.

[8]  K. Nation,et al.  A case of exceptional reading accuracy in a child with Down syndrome: Underlying skills and the relation to reading comprehension , 2006, Cognitive neuropsychology.

[9]  S. Vicari,et al.  Reading skills and phonological awareness acquisition in Down syndrome. , 2006, Journal of intellectual disability research : JIDR.

[10]  Stylianos E. Antonarakis,et al.  Chromosome 21 and Down syndrome: from genomics to pathophysiology , 2004, Nature Reviews Genetics.

[11]  C. Hulme,et al.  Phonemes, rimes, vocabulary, and grammatical skills as foundations of early reading development: evidence from a longitudinal study. , 2004, Developmental psychology.

[12]  Julia M. Carroll,et al.  Language and phonological skills in children at high risk of reading difficulties. , 2004, Journal of child psychology and psychiatry, and allied disciplines.

[13]  A. Baddeley,et al.  What Links Verbal Short-Term Memory Performance and Vocabulary Level? Evidence of Changing Relationships among Individuals with Learning Disability. , 2004 .

[14]  M. Coltheart,et al.  Is there a causal link from phonological awareness to success in learning to read? , 2004, Cognition.

[15]  S. Majerus,et al.  An Investigation of Verbal Short-term Memory and Phonological Processing in Four Children With Williams Syndrome , 2003, Neurocase.

[16]  Charles Hulme,et al.  The development of phonological awareness in preschool children. , 2003, Developmental psychology.

[17]  Uta Frith,et al.  Family risk of dyslexia is continuous: individual differences in the precursors of reading skill. , 2003, Child development.

[18]  Paula J. Clarke,et al.  General cognitive ability in children with reading comprehension difficulties. , 2002, The British journal of educational psychology.

[19]  Charles Hulme,et al.  A deficit in rime awareness in children with Down syndrome , 2002 .

[20]  D. Gunn,et al.  Relationships between reading, phonological skills and language development in individuals with Down syndrome: A five year follow-up study , 2002 .

[21]  Daniel Ansari,et al.  Atypical development of language and social communication in toddlers with Williams syndrome , 2002 .

[22]  Margaret J. Snowling,et al.  The effects of global similarity between stimuli on children's judgment of rime and alliteration , 2001, Applied Psycholinguistics.

[23]  A. Karmiloff-Smith,et al.  Learning to read in Williams syndrome: looking beneath the surface of atypical reading development. , 2001, Journal of child psychology and psychiatry, and allied disciplines.

[24]  Carolyn B. Mervis,et al.  The Williams Syndrome Cognitive Profile , 2000, Brain and Cognition.

[25]  A. Karmiloff-Smith,et al.  Williams syndrome: from genotype through to the cognitive phenotype. , 2000, American journal of medical genetics.

[26]  A. Karmiloff-Smith Development itself is the key to understanding developmental disorders , 1998, Trends in Cognitive Sciences.

[27]  Margaret J. Snowling,et al.  Semantic processing and the development of word-recognition skills: Evidence from children with reading comprehension difficulties. , 1998 .

[28]  P. Howlin,et al.  Cognitive functioning in adults with Williams syndrome. , 1998, Journal of child psychology and psychiatry, and allied disciplines.

[29]  C. Hulme,et al.  Segmentation, not rhyming, predicts early progress in learning to read. , 1997, Journal of experimental child psychology.

[30]  M. Davies,et al.  A LONGITUDINAL STUDY OF COGNITIVE ABILITIES AND EDUCATIONAL ATTAINMENT IN WILLIAMS SYNDROME , 1996, Developmental medicine and child neurology.

[31]  Peter J. Hatcher,et al.  Ameliorating Early Reading Failure by Integrating the Teaching of Reading and Phonological Skills: The Phonological Linkage Hypothesis , 1994 .

[32]  U. Goswami,et al.  Phonological Skills and Learning to Read , 1993, Annals of the New York Academy of Sciences.

[33]  H. Scarborough,et al.  Very early language deficits in dyslexic children. , 1990, Child development.

[34]  R. Pagon,et al.  Williams syndrome: features in late childhood and adolescence. , 1987, Pediatrics.

[35]  G. Scerif,et al.  Neurocognitive development of attention across genetic syndromes: inspecting a disorder's dynamics through the lens of another. , 2011, Progress in brain research.

[36]  J. Atkinson,et al.  Gene expression to neurobiology and behavior : human brain development and developmental disorders , 2011 .

[37]  M. Coltheart,et al.  Early orthographic influences on phonemic awareness tasks: evidence from a preschool training study. , 2011, Journal of Experimental Child Psychology.

[38]  H. Fletcher,et al.  Phonological awareness in children with Down syndrome. , 2002, Down's syndrome, research and practice : the journal of the Sarah Duffen Centre.

[39]  C. Hulme,et al.  Segmentation, not rhyming, predicts early progress in learning to read. , 1998, Journal of experimental child psychology.

[40]  C. Hulme,et al.  Learning to read: a theoretical synthesis. , 1993, Advances in child development and behavior.

[41]  Donald Shankweiler,et al.  Phonological processes in literacy : a tribute to Isabelle Y. Liberman , 1991 .

[42]  Anne E. Fowler,et al.  How early phonological development might set the stage for phoneme awareness. , 1991 .