Early Developmental Trajectories in Infants With Neurofibromatosis 1

Abstract

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.

Developmental trajectories in infants and pre-school children with Neurofibromatosis 1

“…Consistent with a previous longitudinal study of NF1 children aged 21-40 months, 13 we largely found no sex differences in the trajectories of cognitive, behavioural or social development. These sex differences may emerge at a later timepoint, as there are significant sex differences observed in cross-sectional studies in school-age children with NF1.…”

“…Previous papers from our group examined trajectories of cognitive and adaptive behavioural development in infants with NF1 aged 5 to 14 months compared with typically developing (TD) children. 13 At this early stage in development, no group differences were observed in trajectories of cognitive and adaptive functioning, nor differences on social communication measures. However, early differences in neural processing including auditory processing and excitation/inhibition balance were observed, both of which were related to later autistic traits.…”

“…Understanding this is crucial for identifying early markers of later neurodevelopmental outcomes, and has clinical implications for screening and intervention, particularly educational planning. 13 Two previous studies have analysed trajectories of cognitive development in toddlers. 14,15 Lorenzo et al (2015) assessed the early development of 39 children with NF1 aged 21, 30 and 40 months compared with matched controls; the NF1 cohort demonstrated lower cognitive functioning than controls.…”

“…16,17 Our study aim is to examine the early cognitive, behavioural, autism and ADHD trait development of infants and pre-school aged children with NF1, compared with a cohort of TD children. Our objectives were to build on the earlier work of our team 13 by examining longitudinal trajectories of cognitive and behavioural development from 5-36 months, and ADHD trait development at 24 and 36 months. We also aimed to examine emergence of autism traits at 24 and 36 months.…”

“…Understanding this is crucial for identifying early markers of later neurodevelopmental outcomes, and has clinical implications for screening and intervention, particularly educational planning. 13…”