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NFB-11. WHITE MATTER DIFFERENCES IN CHILDREN WITH NF1 COMPARED TO CONTROLS
INTRODUCTION: Neurofibromatosis type 1 (NF1) is a genetic condition in which children develop learning challenges and glioma. White matter tracts (WMT) are implicated in these cognitive functions, while oligodendroglial precursor cells are implicated in both gliomagenesis and white-matter developmen...
Autores principales: | , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Oxford University Press
2020
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7715453/ http://dx.doi.org/10.1093/neuonc/noaa222.614 |
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author | Bruckert, Lisa Travis, Katie McKenna, Emily Yeom, Kristen Campen, Cynthia |
author_facet | Bruckert, Lisa Travis, Katie McKenna, Emily Yeom, Kristen Campen, Cynthia |
author_sort | Bruckert, Lisa |
collection | PubMed |
description | INTRODUCTION: Neurofibromatosis type 1 (NF1) is a genetic condition in which children develop learning challenges and glioma. White matter tracts (WMT) are implicated in these cognitive functions, while oligodendroglial precursor cells are implicated in both gliomagenesis and white-matter development. Specific WMTs have not been well characterized in NF1. METHODS: Twenty NF1 patients aged 1.4–17.6 years (M = 9.5 years, 24 male) and 20 age-and-sex-matched controls underwent dMRI at 3T (25 directions, b=1000 s/mm(2)). Automated segmentation of WMTs extracted fractional anisotropy (FA) and mean diffusivity (MD) of 18 major WMTs. Covariance analysis examined the effect of group (NF1/controls) on FA/MD after controlling for intracranial volume. Regression analyses for WMTs determined the interaction of FA/MD with age for NF1 patients compared to controls. Significance was set at p<0.05 after correcting for multiple comparisons using false discovery rate. RESULTS: Compared to controls, children with NF1 had significantly decreased FA in 8 and increased MD in 12/18 tracts. Differences held after controlling for intracranial volume. The interaction between group and age accounted for a significant proportion of the variance in FA in 9 and in MD in 16/18 tracts. FA and MD differences between children with NF1 and controls were greater at younger than older ages. CONCLUSION: Microstructural differences were observed in WMTs in children with NF1 compared to controls. These differences were not explained by intracranial volume and were most pronounced in younger children with NF1 compared to controls. These findings have implications for understanding neurocognitive deficits and gliomagenesis observed in children with NF1. |
format | Online Article Text |
id | pubmed-7715453 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2020 |
publisher | Oxford University Press |
record_format | MEDLINE/PubMed |
spelling | pubmed-77154532020-12-09 NFB-11. WHITE MATTER DIFFERENCES IN CHILDREN WITH NF1 COMPARED TO CONTROLS Bruckert, Lisa Travis, Katie McKenna, Emily Yeom, Kristen Campen, Cynthia Neuro Oncol Neurofibromatosis INTRODUCTION: Neurofibromatosis type 1 (NF1) is a genetic condition in which children develop learning challenges and glioma. White matter tracts (WMT) are implicated in these cognitive functions, while oligodendroglial precursor cells are implicated in both gliomagenesis and white-matter development. Specific WMTs have not been well characterized in NF1. METHODS: Twenty NF1 patients aged 1.4–17.6 years (M = 9.5 years, 24 male) and 20 age-and-sex-matched controls underwent dMRI at 3T (25 directions, b=1000 s/mm(2)). Automated segmentation of WMTs extracted fractional anisotropy (FA) and mean diffusivity (MD) of 18 major WMTs. Covariance analysis examined the effect of group (NF1/controls) on FA/MD after controlling for intracranial volume. Regression analyses for WMTs determined the interaction of FA/MD with age for NF1 patients compared to controls. Significance was set at p<0.05 after correcting for multiple comparisons using false discovery rate. RESULTS: Compared to controls, children with NF1 had significantly decreased FA in 8 and increased MD in 12/18 tracts. Differences held after controlling for intracranial volume. The interaction between group and age accounted for a significant proportion of the variance in FA in 9 and in MD in 16/18 tracts. FA and MD differences between children with NF1 and controls were greater at younger than older ages. CONCLUSION: Microstructural differences were observed in WMTs in children with NF1 compared to controls. These differences were not explained by intracranial volume and were most pronounced in younger children with NF1 compared to controls. These findings have implications for understanding neurocognitive deficits and gliomagenesis observed in children with NF1. Oxford University Press 2020-12-04 /pmc/articles/PMC7715453/ http://dx.doi.org/10.1093/neuonc/noaa222.614 Text en © The Author(s) 2020. Published by Oxford University Press on behalf of the Society for Neuro-Oncology. http://creativecommons.org/licenses/by-nc/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com |
spellingShingle | Neurofibromatosis Bruckert, Lisa Travis, Katie McKenna, Emily Yeom, Kristen Campen, Cynthia NFB-11. WHITE MATTER DIFFERENCES IN CHILDREN WITH NF1 COMPARED TO CONTROLS |
title | NFB-11. WHITE MATTER DIFFERENCES IN CHILDREN WITH NF1 COMPARED TO CONTROLS |
title_full | NFB-11. WHITE MATTER DIFFERENCES IN CHILDREN WITH NF1 COMPARED TO CONTROLS |
title_fullStr | NFB-11. WHITE MATTER DIFFERENCES IN CHILDREN WITH NF1 COMPARED TO CONTROLS |
title_full_unstemmed | NFB-11. WHITE MATTER DIFFERENCES IN CHILDREN WITH NF1 COMPARED TO CONTROLS |
title_short | NFB-11. WHITE MATTER DIFFERENCES IN CHILDREN WITH NF1 COMPARED TO CONTROLS |
title_sort | nfb-11. white matter differences in children with nf1 compared to controls |
topic | Neurofibromatosis |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7715453/ http://dx.doi.org/10.1093/neuonc/noaa222.614 |
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