Teaching reading to children with neurofibromatosis type 1: a clinical trial with random assignment to different approaches

Abstract

AIM Neurofibromatosis type 1 (NF1) is a genetic disorder with a cognitive profile that includes visual–spatial perception deficits and a high incidence of reading disability. There is a paucity of information about how this cognitively complex population responds to mainstream reading interventions. The clinical trial goals were to determine whether children and adolescents with NF1 and reading deficits (NF+RD) benefit from mainstream remedial reading programs and whether responsiveness varies with differences in program-related visual–spatial demands. METHOD Forty-nine participants (18 males, 21 females; aged 8–14y) with either NF+RD (n=17, 11 males, 6 females) or idiopathic reading deficit (IRD) (n=32, 17 males, 15 females) were randomly assigned to intensive remedial teaching using one of two multisensory reading programs: one with greater kinesthetic demands and the other with greater visual–spatial demands. Two control groups – wait-list IRD (n=14, 11 males, 3 females) and typically developing readers (n=26, 13 males, 13 females) – received no treatment. Repeated measures and multivariate ANOVA analyses compared each group's growth in reading achievement from pre- to post-testing. RESULTS Treated groups showed significant growth whereas untreated groups did not. Comparing treated groups, the IRD group responded equally well to both interventions, whereas the NF+RD group showed a better response to the more kinesthetic approach. INTERPRETATION Results suggest that multisensory remedial reading teaching that emphasizes kinesthetic demands more than visual–spatial demands is suitable for students with NF+RD.

Cognitive and Behavioral Disorders in Children with Neurofibromatosis Type 1

“…Although further cognitive impairment could be predicted in children with NF1 and concomitant ADHD, some studies have shown no difference in specific skills between NF1 children with ADHD and children with NF1 only. Moreover, Barquero et al ( 11 ) found that scores for ADHD symptoms according to Conners Parent Rating were similar in the four reading groups assessed (NF1 and reading impairment, idiopathic reading deficit, reading deficit reading group, and typical development). In addition, Payne et al ( 22 ) studied SWM and inhibitory control in NF1 children with and without comorbid ADHD.…”

“…Barquero et al ( 11 ) conducted the first evaluation of an intervention for reading deficiencies in 49 children with NF1. Some children received remedial reading instruction from a kinesthetic reading program and others used a program that required greater visuospatial demands.…”

“…In contrast, several studies indicate the presence of attention deficits in children with NF1 that do not seem to affect the performance of specific skills compared with children with NF1 only. Reading ( 11 ), spontaneous and reactive cognitive flexibility tasks ( 26 ), SWM and inhibitory control ( 22 ) were not adversely affected by ADHD symptomatology. The association between attention deficits and motor skills has not been well studied.…”

“…Analysis of the interaction between cognitive and social processes may generate important information that could guide social interventions ( 27 ). In addition, reading interventions could be conducted with larger samples and in combination with pharmaceutical interventions, such as lovastatin ( 11 , 73 ) and MPD, which have shown some benefits for cognitive function ( 45 ). More studies on motor skills are needed to clarify the interdependency between these skills and neurocognitive performance ( 17 ), attention deficits ( 58 ), and language ( 55 ).…”

Role of Neurocognitive Factors in Academic Fluency for Children and Adults With Spina Bifida Myelomeningocele

“…While there are empirically supported interventions that address either reading fluency (Chard, Vaughn, & Tyler, 2002;Wolf & Katzir-Cohen, 2001) or math fact fluency (Codding, Burns, & Lukito, 2011;Fuchs et al, 2010) in TD individuals, there are as yet very few data on the implementation of these programs for individuals with neurodevelopmental disorders such as SBM. For example, we know of only one small case study where an intervention that is efficacious for addressing weak academic skills in TD individuals (math) was shown to also be beneficial in SBM (Coughlin & Montague, 2011), although Barquero, Sefcik, Cutting, and Rimrodt (2015) did implement a reading intervention for individuals with reading difficulty with and without neurofibromatosis.…”