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- Title
Preliteracy impairments in children with neurofibromatosis type 1.
- Authors
Arnold, Shelley S.; Payne, Jonathan M.; Lorenzo, Jennifer; North, Kathryn N.; Barton, Belinda
- Abstract
<bold>Aim: </bold>This cross-sectional study aimed to examine the preliteracy abilities of young children with neurofibromatosis type 1 (NF1) and to identify which of these abilities best predicted conventional literacy (spelling).<bold>Method: </bold>Forty-two children with NF1 (23 males, 19 females; mean age [SD] 5y 6mo [6mo]) were compared with 32 unaffected children (15 males, 17 females; mean age [SD] 5y 4mo [6mo]). All children completed a comprehensive cognitive assessment including measures of phonological processing (phonological awareness, phonological memory, rapid automatic naming) and letter-sound knowledge.<bold>Results: </bold>Children with NF1 performed significantly poorer than the comparison group across all cognitive and preliteracy domains, with specific weaknesses evident in phonological awareness (F1,68 =14.13, p<0.001, partial η2 =0.17), phonological memory (F1,68 =13.87, p<0.001, partial η2 =0.17), and letter-sound knowledge (F1,71 =5.65, p=0.020, partial η2 =0.07). Within the group with NF1 group, over a third of children demonstrated impairment in at least one phonological processing domain and the risk of phonological impairment was 5.60 times that of unaffected children. Children's letter-sound knowledge was the strongest predictor of conventional literacy (spelling).<bold>Interpretation: </bold>This study establishes that preliteracy deficits are present and detectable in young children with NF1. As a result of the high incidence of preliteracy impairment, we recommend screening phonological awareness and letter-sound knowledge to identify risk of future learning disorders.<bold>What This Paper Adds: </bold>Young children with neurofibromatosis type 1 are at elevated risk of preliteracy deficits. The most affected domains are phonological awareness and phonological memory. Letter-sound knowledge is the strongest predictor of conventional literacy (spelling).
- Subjects
NEUROFIBROMATOSIS 1; NEUROFIBROMATOSIS in children; PRELITERACY; LEARNING disabilities; PHONOLOGICAL awareness; DISABILITIES; COGNITION disorders diagnosis; ANALYSIS of variance; COGNITION; COGNITION disorders; COMPARATIVE studies; DEVELOPMENTAL disabilities; LONGITUDINAL method; DYSLEXIA; NEUROPSYCHOLOGICAL tests; RESEARCH methodology; MEDICAL cooperation; READABILITY (Literary style); RESEARCH; EVALUATION research; CROSS-sectional method; DISEASE complications; DIAGNOSIS
- Publication
Developmental Medicine & Child Neurology, 2018, Vol 60, Issue 7, p703
- ISSN
0012-1622
- Publication type
journal article
- DOI
10.1111/dmcn.13768