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White matter microstructure of patients with neurofibromatosis type 1 and its relation to inhibitory control

Neurofibromatosis Type 1 (NF1) is commonly associated with deficits in executive functions such as working memory and inhibitory control. A valid biomarker to describe the pathological basis of these deficits in NF1 is not available. The aim of this study was to investigate whether any abnormalities...

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Autores principales: Koini, M., Rombouts, S. A. R. B., Veer, I. M., Van Buchem, M. A., Huijbregts, S. C. J.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer US 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5707233/
https://www.ncbi.nlm.nih.gov/pubmed/27796732
http://dx.doi.org/10.1007/s11682-016-9641-3
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author Koini, M.
Rombouts, S. A. R. B.
Veer, I. M.
Van Buchem, M. A.
Huijbregts, S. C. J.
author_facet Koini, M.
Rombouts, S. A. R. B.
Veer, I. M.
Van Buchem, M. A.
Huijbregts, S. C. J.
author_sort Koini, M.
collection PubMed
description Neurofibromatosis Type 1 (NF1) is commonly associated with deficits in executive functions such as working memory and inhibitory control. A valid biomarker to describe the pathological basis of these deficits in NF1 is not available. The aim of this study was to investigate whether any abnormalities in white matter integrity of the executive function related anterior thalamic radiation (ATR), cingulate bundle (CB), and superior longitudinal fasciculus (SLF) may be regarded as a pathological basis for inhibitory control deficits in adolescents with NF1. Sixteen NF1 patients and 32 healthy controls underwent 3 T DTI MRI scanning. Whole brain-, ATR-, CB-, and SLF-white matter integrity were studied using fractional anisotropy, mean (MD), radial, and axial (DA) diffusivity. Correlation analyses between white matter metrics and inhibitory control (as measured with a computerized task) were performed. Also, verbal and performance abilities (IQ-estimates) were assessed and correlated with white matter metrics. Patients showed significant whole brain- and local microstructural pathology when compared to healthy controls in all measures. In NF1-patients, whole-brain (MD: r = .646 and DA: r = .673) and ATR- (r-range: −.405–.771), but not the CB- (r-range: −.307–.472) and SLF- (r-range: −.187–.406) white matter integrity, were correlated with inhibitory control. Verbal and performance abilities were not associated with white matter pathology. In NF1, white matter abnormalities are observed throughout the brain, but damage to the ATR seems specifically, or at least most strongly related to inhibitory control. Future studies should examine whether reduced white matter integrity in other brain regions or tracts is (more strongly) associated with different aspects of the cognitive-behavioral phenotype associated with NF1.
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spelling pubmed-57072332017-12-04 White matter microstructure of patients with neurofibromatosis type 1 and its relation to inhibitory control Koini, M. Rombouts, S. A. R. B. Veer, I. M. Van Buchem, M. A. Huijbregts, S. C. J. Brain Imaging Behav Original Research Neurofibromatosis Type 1 (NF1) is commonly associated with deficits in executive functions such as working memory and inhibitory control. A valid biomarker to describe the pathological basis of these deficits in NF1 is not available. The aim of this study was to investigate whether any abnormalities in white matter integrity of the executive function related anterior thalamic radiation (ATR), cingulate bundle (CB), and superior longitudinal fasciculus (SLF) may be regarded as a pathological basis for inhibitory control deficits in adolescents with NF1. Sixteen NF1 patients and 32 healthy controls underwent 3 T DTI MRI scanning. Whole brain-, ATR-, CB-, and SLF-white matter integrity were studied using fractional anisotropy, mean (MD), radial, and axial (DA) diffusivity. Correlation analyses between white matter metrics and inhibitory control (as measured with a computerized task) were performed. Also, verbal and performance abilities (IQ-estimates) were assessed and correlated with white matter metrics. Patients showed significant whole brain- and local microstructural pathology when compared to healthy controls in all measures. In NF1-patients, whole-brain (MD: r = .646 and DA: r = .673) and ATR- (r-range: −.405–.771), but not the CB- (r-range: −.307–.472) and SLF- (r-range: −.187–.406) white matter integrity, were correlated with inhibitory control. Verbal and performance abilities were not associated with white matter pathology. In NF1, white matter abnormalities are observed throughout the brain, but damage to the ATR seems specifically, or at least most strongly related to inhibitory control. Future studies should examine whether reduced white matter integrity in other brain regions or tracts is (more strongly) associated with different aspects of the cognitive-behavioral phenotype associated with NF1. Springer US 2016-10-29 2017 /pmc/articles/PMC5707233/ /pubmed/27796732 http://dx.doi.org/10.1007/s11682-016-9641-3 Text en © The Author(s) 2016 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made.
spellingShingle Original Research
Koini, M.
Rombouts, S. A. R. B.
Veer, I. M.
Van Buchem, M. A.
Huijbregts, S. C. J.
White matter microstructure of patients with neurofibromatosis type 1 and its relation to inhibitory control
title White matter microstructure of patients with neurofibromatosis type 1 and its relation to inhibitory control
title_full White matter microstructure of patients with neurofibromatosis type 1 and its relation to inhibitory control
title_fullStr White matter microstructure of patients with neurofibromatosis type 1 and its relation to inhibitory control
title_full_unstemmed White matter microstructure of patients with neurofibromatosis type 1 and its relation to inhibitory control
title_short White matter microstructure of patients with neurofibromatosis type 1 and its relation to inhibitory control
title_sort white matter microstructure of patients with neurofibromatosis type 1 and its relation to inhibitory control
topic Original Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5707233/
https://www.ncbi.nlm.nih.gov/pubmed/27796732
http://dx.doi.org/10.1007/s11682-016-9641-3
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