Phonics Training Improves Reading in Children with Neurofibromatosis Type 1: A Prospective Intervention Trial

Objective To examine the efficacy of a phonics-training program in children with neurofibromatosis type 1 (NF1) and reading difficulties. Study design Thirty children (7-12 years of age) with NF1 completed a double-baseline, 24-week intervention trial. Literacy outcome measures were assessed at 4 ti...

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Bibliographic Details
Published in:The Journal of pediatrics 2016-10, Vol.177, p.219-226.e2
Main Authors: Arnold, Shelley S., BA, Barton, Belinda, PhD, McArthur, Genevieve, PhD, North, Kathryn N., MD, Payne, Jonathan M., DPsych
Format: Article
Language:English
Subjects:
Child
Dyslexia - etiology
Dyslexia - therapy
Early Intervention (Education)
Female
Humans
intervention
Male
Neurofibromatosis 1 - complications
neurofibromatosis type 1
Pediatrics
phonics training
phonological awareness
Prospective Studies
reading deficit
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Summary:Objective To examine the efficacy of a phonics-training program in children with neurofibromatosis type 1 (NF1) and reading difficulties. Study design Thirty children (7-12 years of age) with NF1 completed a double-baseline, 24-week intervention trial. Literacy outcome measures were assessed at 4 time points: (1) at baseline; (2) after an 8-week no-treatment period; (3) immediately post-treatment; and (4) at follow-up 8 weeks post-treatment. Repeated-measures ANOVA were conducted to examine change over time for all outcome measures, and significant main effects were explored with planned comparisons. Predictors of treatment effects were examined by linear regressions. Results Ninety percent of participants completed the intervention. Intervention-specific improvements were observed across a range of literacy outcomes, including reading accuracy (nonword reading, Cohen d  = 1.10; regular-word reading, Cohen d  = 0.32), letter-sound knowledge (Cohen d  = 0.80), blending (Cohen d  = 0.88), repetition of nonsense words (Cohen d  = 0.94), phonemic decoding fluency (Cohen d  = 0.55), and reading comprehension (Cohen d  = 0.31). Improvements were maintained 8 weeks post-treatment. Age ( P  = .03) and working memory ( P  = .02) significantly influenced efficacy, with greatest improvements observed in older children with stronger verbal working memory capacity. Conclusions Home-based, computerized reading intervention was effective in improving the reading and reading-related abilities of children with NF1 and reading difficulty. Trial registration Australian and New Zealand Clinical Trials Registry: ACTRN12611000779976
ISSN:0022-3476
1097-6833
DOI:10.1016/j.jpeds.2016.06.037