An Examination of Lexical and Sublexical Reading Skills in Children with Neurofibromatosis Type 1

Watt, Sharon; Shores, E. Arthur; North, Klaus

doi:10.1080/09297040701595505

Cited by 36 publications

(35 citation statements)

References 37 publications

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“…Of note, we found strong correlations between phonological awareness and letter‐sound knowledge in our NF1 cohort, which suggests these two skills typically develop in parallel in NF1; or, at least, that when there is a delayed development in one of these skills, there is usually poor development in the other. Taken together, these findings suggest that weak letter‐sound knowledge ultimately appears to be the most important predictor of early spelling ability in NF1, and that deficits in letter‐sound knowledge in combination with phonological impairments are likely to significantly contribute to the literacy difficulties that are common in school‐aged children with the condition …”

Section: Discussionmentioning

confidence: 83%

Preliteracy impairments in children with neurofibromatosis type 1

Arnold

Payne

Lorenzo

et al. 2018

Develop Med Child Neuro

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Section: Discussionmentioning

confidence: 83%

Preliteracy impairments in children with neurofibromatosis type 1

Arnold

Payne

Lorenzo

et al. 2018

Develop Med Child Neuro

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“…Although it is true that most children with NF-1 show visuospatial impairment, a variety of other (and more academically relevant) deficits have been noted, including those in the reading, language, and memory domains (Hyman, Shores, & North, 2006; Krab et al 2008; Levine, Materek, Abel, O’Donnell, & Cutting, 2006; Watt, Shores, & North, 2008). Because of the tremendous importance of reading for life achievement, particularly regarding their children’s school and employment future, weaknesses in reading are often of utmost concern for parents and teachers (Krab et al; Stine and Adams, 1989), and almost all studies that have examined reading abilities in NF-1 have reported deficits in this area (e.g., Brewer, Moore, & Hiscock, 1997; Cutting, Koth, & Denckla, 2000; Dilts et al, 1996; Hofman, Marris, Bryan, & Denckla, 1994; Mazzocco et al, 1995; Mautner, Kluwe, Thakker, & Leark, 2002; North et al, 1994; North, 2000; Watts et al, 2008).…”

Section: Learning Disabilities In Nf-1mentioning

confidence: 99%

Cognitive Profile of Children with Neurofibromatosis and Reading Disabilities

Cutting

Levine

2010

Child Neuropsychology

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A large percentage of children with Neurofibromatosis Type 1(NF-1) have learning disabilities, often in the realm of reading. Previous studies have indicated that children with NF-1 show a neuropsychological profile similar to idiopathic reading disabilities (IRD); however, studies typically have not subdivided children with NF-1 into those who do and not have RD (NF+RD and NFnoRD, respectively). The current study examined the cognitive profile of children with NF-1 with and without RD and compared them to children with IRD as well as to typically developing readers (Controls). Findings showed that children with NF+RD performed similarly to children with IRD on phonological, rapid naming, and reading comprehension measures; however, children with NF +RD displayed pronounced visual spatial deficits as compared to IRD and Control groups. In addition, when comparing the NF-1 groups to each other as well as to the CNT and IRD groups, the current study reported that there were no oral language differences; lack of findings in the realm of oral language was attributed to the fact that groups were equated on IQ. Overall, findings suggest that a more refined classification of children with NF-1 may be helpful for tailoring academic interventions. KeywordsNeurofibromatosis; Reading Disabilities; Learning Disabilities; Cognitive; Neuropsychology Neurofibromatosis Type 1 (NF-1) is a genetic disorder that has an incidence of 1:3,500 and is equally pervasive across ethnicities and gender. Currently, about 30% to 50% of the cases are due to spontaneous mutation, whereas the remainder of are due to familial mutation. A clinical diagnosis of NF-1 must include two or more of the following: six or more cafe au lait macules; two or more neurofibromas, or one plexiform neurofibroma; freckling in the axilla or inguinal region; an optic glioma; two or more Lisch Nodules; a distinct osseous lesion; and a first degree relative with NF-1. Learning Disabilities in NF-1Although a specific cognitive profile associated with NF-1 has remained elusive, one consistent finding is that Learning Disabilities (LDs) and other cognitive and behavioral difficulties are much more prevalent in the NF-1 population than in the general population, with approximately 70 percent showing LDs and/or behavioral difficulties such as Attention Deficit Disorder and NIH Public Access Author ManuscriptChild Neuropsychol. Author manuscript; available in PMC 2011 September 1. NIH-PA Author ManuscriptNIH-PA Author Manuscript NIH-PA Author Manuscript other neuropsychological deficits Mautner, Kluwe, Thakker, & Leark, 2002;Moore, 2009; Noll et al. 2007;Rosser & Packer, 2003).Initially, the cognitive profile of children with NF1 was characterized as "nonverbal" because most individuals with NF-1 show impairment in visual perceptual (specifically visuospatial) functioning. Although it is true that most children with NF-1 show visuospatial impairment, a variety of other (and more academically relevant) deficits have been noted, including those in the reading, lan...

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“…Concerning the DL findings, to the best of our knowledge, only one study examined lexical and phonological reading skills in children with NF1 (Watt et al, 2008). The authors reported 67% of the sample exhibited deficits in one or more reading skills.…”

Section: Discussionmentioning

confidence: 99%

“…SLD have been reported in 30–70% of children with NF1 (Hyman et al, 2005; Acosta, 2007). Nevertheless, the diagnosis of SLD has mainly focused on IQ-achievement discrepancy (Hyman et al, 2005; Watt et al, 2008). The discrepancy model has been criticized for numerous authors.…”

Section: Introductionmentioning

confidence: 99%

“…To the best of our knowledge, the identification of DD focused on defective basic numerical capacities and math fluency has not been previously addressed in children with NF1 and only one study evaluated DL subtypes considering phonological and lexical deficits in this genetic disorder (Watt et al, 2008). The present study aims to evaluate a group of children with NF1 in order to determine the presence of DD and DL based on profiles of defective core capacities, using neurocognitive tests specifically designed and standardized for the diagnosis of these SLD.…”

Section: Introductionmentioning

confidence: 99%

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Neurocognitive profiles of learning disabled children with neurofibromatosis type 1

Orraca-Castillo¹,

Estévez-Pérez

Reigosa-Crespo

2014

Front. Hum. Neurosci.

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Neurofibromatosis 1 (NF1) is a genetic condition generally associated with intellectual deficiency and learning disabilities. Although there have been groundbreaking advances in the understanding of the molecular, cellular, and neural systems underlying learning deficits associated to NF1 in animal models, much remains to be learned about the spectrum of neurocognitive phenotype associated with the NF1 clinical syndrome. In the present study, 32 children with NF1 ranging from 7 to 14 years were evaluated with neurocognitive tests dedicated to assess basic capacities which are involved in reading and mathematical achievement. Deficits in lexical and phonological strategies and poor number facts retrieval were found underlying reading and arithmetic disorders, respectively. Additionally, efficiencies in lexical/phonological strategies and mental arithmetic were significant predictors of individual differences in reading attainment and math. However, deficits in core numeric capacities were not found in the sample, suggesting that it is not responsible for calculation dysfluency. The estimated prevalence of Developmental Dyscalculia was 18.8%, and the male:female ratio was 5:1. On the other hand, the prevalence of Developmental Dyslexia was almost 3 times as high (50%), and no gender differences were found (male: female ratio = 1:1). This study offers new evidence to the neurocognitive phenotype of NF1 contributing to an in depth understanding of this condition, but also to possible treatments for the cognitive deficits associated with NF1.

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An Examination of Lexical and Sublexical Reading Skills in Children with Neurofibromatosis Type 1

Cited by 36 publications

References 37 publications

Preliteracy impairments in children with neurofibromatosis type 1

Preliteracy impairments in children with neurofibromatosis type 1

Cognitive Profile of Children with Neurofibromatosis and Reading Disabilities

Neurocognitive profiles of learning disabled children with neurofibromatosis type 1

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