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Early Developmental Trajectories in Infants With Neurofibromatosis 1

OBJECTIVE: To examine the trajectories of cognitive, motor and behavioural development in infants with NF1 compared to infants without a family history of neurodevelopmental difficulties. STUDY DESIGN: Infants with NF1 and low-risk controls were recruited from 5 months of age and followed longitudin...

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Autores principales: Garg, Shruti, Wan, Ming Wai, Begum-Ali, Jannath, Kolesnik-Taylor, Anna, Green, Jonathan, Johnson, Mark H., Jones, Emily
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9355323/
https://www.ncbi.nlm.nih.gov/pubmed/35936291
http://dx.doi.org/10.3389/fpsyg.2022.795951
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author Garg, Shruti
Wan, Ming Wai
Begum-Ali, Jannath
Kolesnik-Taylor, Anna
Green, Jonathan
Johnson, Mark H.
Jones, Emily
author_facet Garg, Shruti
Wan, Ming Wai
Begum-Ali, Jannath
Kolesnik-Taylor, Anna
Green, Jonathan
Johnson, Mark H.
Jones, Emily
author_sort Garg, Shruti
collection PubMed
description OBJECTIVE: To examine the trajectories of cognitive, motor and behavioural development in infants with NF1 compared to infants without a family history of neurodevelopmental difficulties. STUDY DESIGN: Infants 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. RESULTS: No 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. CONCLUSION: Cognitive, 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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spelling pubmed-93553232022-08-06 Early Developmental Trajectories in Infants With Neurofibromatosis 1 Garg, Shruti Wan, Ming Wai Begum-Ali, Jannath Kolesnik-Taylor, Anna Green, Jonathan Johnson, Mark H. Jones, Emily Front Psychol Psychology OBJECTIVE: To examine the trajectories of cognitive, motor and behavioural development in infants with NF1 compared to infants without a family history of neurodevelopmental difficulties. STUDY DESIGN: Infants 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. RESULTS: No 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. CONCLUSION: Cognitive, 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. Frontiers Media S.A. 2022-07-22 /pmc/articles/PMC9355323/ /pubmed/35936291 http://dx.doi.org/10.3389/fpsyg.2022.795951 Text en Copyright © 2022 Garg, Wan, Begum-Ali, Kolesnik-Taylor, Green, Johnson and Jones. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Psychology
Garg, Shruti
Wan, Ming Wai
Begum-Ali, Jannath
Kolesnik-Taylor, Anna
Green, Jonathan
Johnson, Mark H.
Jones, Emily
Early Developmental Trajectories in Infants With Neurofibromatosis 1
title Early Developmental Trajectories in Infants With Neurofibromatosis 1
title_full Early Developmental Trajectories in Infants With Neurofibromatosis 1
title_fullStr Early Developmental Trajectories in Infants With Neurofibromatosis 1
title_full_unstemmed Early Developmental Trajectories in Infants With Neurofibromatosis 1
title_short Early Developmental Trajectories in Infants With Neurofibromatosis 1
title_sort early developmental trajectories in infants with neurofibromatosis 1
topic Psychology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9355323/
https://www.ncbi.nlm.nih.gov/pubmed/35936291
http://dx.doi.org/10.3389/fpsyg.2022.795951
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