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Phonics Training Improves Reading in Children with Neurofibromatosis Type 1: A Prospective Intervention Trial
Shelley S Arnold1, Belinda Barton2, Genevieve McArthur3
1Institute for Neuroscience and Muscle Research, The Children's Hospital at Westmead, Australia; Discipline of Paediatrics and Child Health, Faculty of Medicine, University of Sydney, Australia.
Insights
A phonics-training program significantly improved reading skills in children with neurofibromatosis type 1 (NF1). These gains in literacy were maintained after treatment, highlighting the program's effectiveness for this population.
Area of Science:
- Neuroscience
- Pediatrics
- Educational Psychology
Background:
- Neurofibromatosis type 1 (NF1) is a genetic disorder associated with an increased risk of learning difficulties, particularly in reading.
- Children with NF1 often exhibit deficits in phonological processing, which are foundational for reading acquisition.
- Targeted interventions are crucial to address reading challenges in children with NF1 and improve their academic outcomes.
Purpose of the Study:
- To evaluate the efficacy of a home-based, computerized phonics-training program for children diagnosed with neurofibromatosis type 1 (NF1).
- To assess the impact of the intervention on various literacy skills, including reading accuracy, phonological awareness, and reading comprehension.
- To determine if improvements in reading abilities are sustained post-intervention.
Main Methods:
- A 24-week intervention trial involving 30 children (aged 7-12) with NF1 and reading difficulties.
- A double-baseline design with assessments at baseline, after an 8-week no-treatment period, immediately post-treatment, and at an 8-week follow-up.
- Repeated-measures ANOVA and linear regressions were used to analyze changes in literacy outcomes and predictors of treatment effects.
Main Results:
- Significant improvements were observed in reading accuracy (nonword and regular word reading), letter-sound knowledge, blending, nonsense word repetition, phonemic decoding fluency, and reading comprehension.
- Effect sizes ranged from Cohen d = 0.31 to 1.10, indicating moderate to large improvements across literacy measures.
- Improvements were maintained at the 8-week follow-up, and older children with better verbal working memory showed the greatest gains.
Conclusions:
- A home-based, computerized phonics intervention is effective for enhancing reading and related abilities in children with NF1 and reading difficulties.
- The intervention demonstrates lasting benefits, suggesting it is a viable tool for supporting literacy development in this population.
- Individual factors such as age and working memory capacity influence treatment efficacy, informing personalized intervention approaches.
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.
