Predictors of cognitive development in children with neurofibromatosis type 1 and plexiform neurofibromas

Hou, Yang; Allen, Terrence K.; Wolters, Pamela L.; Toledo‐Tamula, Mary Anne; Martin, Staci; Baldwin, Andrea; Reda, Stephanie; Gillespie, Andy; Goodwin, Anne; Widemann, Brigitte C.

doi:10.1111/dmcn.14489

Cited by 15 publications

(18 citation statements)

References 21 publications

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“…A significant finding in this study, both for the NF1 and control groups, was the impact of maternal education on cognitive and behavioural development. In a longitudinal study of cognitive development in children aged 6-18 years with NF1, Hou et al (2020) found that higher parental education related to higher IQ, Maths, reading and cognitive scores (Hou et al, 2020). Two recent studies have also examined the cognitive and academic differences in children with inherited versus de novo NF1 mutations.…”

Section: Discussionmentioning

confidence: 99%

Early Developmental Trajectories in Infants With Neurofibromatosis 1

et al. 2022

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ObjectiveTo examine the trajectories of cognitive, motor and behavioural development in infants with NF1 compared to infants without a family history of neurodevelopmental difficulties.Study designInfants with NF1 and low-risk controls were recruited from 5 months of age and followed longitudinally. Data from standardised tests was gathered at 5, 10 and 14 months and developmental trajectories of motor, language, behaviour, sleep, social development and parent–infant interaction were examined. Linear mixed modelling was used to estimate group differences in cognitive and behavioural measures over time.ResultsNo group differences were observed on Mullen Scale of Early Learning, overall adaptive functioning, temperament or behavioural measures. There were no group differences observed on measures of social communication or parent–infant interaction. Over the course of development, the NF1 group slept less and took more time to settle to sleep as compared to the control group. Maternal education was significantly associated with cognitive and behavioural developmental outcomes in both groups.ConclusionCognitive, social and behavioural impairments are a cause of significant functional morbidity in children with NF1. This report is the first study to investigate the trajectories of cognitive, motor and behavioural development in infancy in NF1. Our results demonstrate that overall cognitive and behavioural developmental trajectories of the NF1 group in the infancy period are similar to controls. Given previous reports of delayed development in the NF1 cohort by 40 months, early clinical interventions strategies to promote sleep hygiene may be beneficial to optimise developmental outcomes.

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Section: Discussionmentioning

confidence: 99%

Early Developmental Trajectories in Infants With Neurofibromatosis 1

et al. 2022

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“…Further research will be necessary, especially to precisely assess the heritability of IQ in both sporadic and familial NF individuals. 32 Additional factors influencing cognitive development in children with NF1 like cerebral lesions in MRI 33 or clinical features like plexiform neurofibromas 34 needs to be taken into consideration as well.…”

Section: Discussionmentioning

confidence: 99%

Psychosocial and Socioeconomic Factors in Children with Neurofibromatosis Type 1

Leppich

Schneider

Eismann

et al. 2021

Journal of Pediatric Neurology

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The aim of this study is to analyze whether children with familial and sporadic neurofibromatosis type 1 (NF1) differ in psychosocial and socioeconomic aspects such as developmental delay as well as in comorbidities. Medical records of 250 children with NF1 at a median age of 10.6 years (range = 2–20 years at time of data collection) were retrospectively reviewed. Specifically, psychosocial and socioeconomic factors from 88 children with a family history for NF1 and 162 sporadic cases were compared. Comparing IQ, familial cases scored significantly lower than sporadic cases (89.8 vs. 96.5; p = 0.015). IQ scores of children with familial and sporadic NF1 differ depending on level of parental education (mean IQ for high education 101.3 [familial] and 102.8 [sporadic] vs. low education 87.5 [familial] and 90.4 [sporadic]; p < 0.001). No significant differences were found regarding the presence of developmental delay, learning disorders, autism spectrum disorder, or attention deficit hyperactivity disease. Children with inherited NF1 had a significantly lower IQ than sporadic cases. Children with familial NF1 more often had parents with a lower level of education, highlighting a possible impact of NF1 through generations. Also, the present study confirmed previous results in literature regarding high frequencies of learning disabilities, autism spectrum disease, and attention deficits in children with NF1.

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“…Ortopedske komplikacije se rešavaju fizikalnom, korektivnom gimnastikom ili, u najtežim slučajevima, zahtevaju hirurško lečenje [2] . Potrebna je sveobuhvatna neuropsihološka procena za decu sa NF 1 [43] . Kognitivni poremećaji, pridruženi ADHD i teškoće u učenju zahtevaju rane i redovne konsultacije dečijeg psihijatra, psihologa i logopeda.…”

Section: Dijagnostika I Lečenjeunclassified

Cognitive phenotype in neurofibromatosis type 1

Todorović¹,

Pavlović²,

Zelić³

et al. 2020

Engrami

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Neurofibromatoses are a set of different genetic disorders that have a common characteristic of the appearance of nervous system tumors. There are three forms of the disease, of which type 1 neurofibromatosis (NF 1) is the most common. NF 1 is an inherited autosomal-dominant disease, with a high rate of new mutations. In addition to the many physical manifestations and complications that occur in persons with NF 1, there are also numerous cognitive difficulties, including lower general intellectual functioning, learning difficulties, but also problems in attention, visual abilities, executive functions, and speech. Attention disorders are up to three times more common in people with NF 1, while learning disabilities are present in more than half of these subjects. Disturbances in the field of visuospatial perception are recognisable even in the preschool period. About 80% of children with NF1 exhibit various speech and language disorders: slow early speech development, slower vocabulary enrichment, syntactic, semantic and phonological speech disorders. Disruption of executive functions will manifest itself in the areas of working memory, organisation, planning / problem solving. This will reflect as the underperformance in academic achievement. Nearly one-third of these persons have emotional and social problems.

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Predictors of cognitive development in children with neurofibromatosis type 1 and plexiform neurofibromas

Cited by 15 publications

References 21 publications

Early Developmental Trajectories in Infants With Neurofibromatosis 1

Early Developmental Trajectories in Infants With Neurofibromatosis 1

Psychosocial and Socioeconomic Factors in Children with Neurofibromatosis Type 1

Cognitive phenotype in neurofibromatosis type 1

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