Quantitative MRI demonstrates abnormalities of the third ventricle subventricular zone in neurofibromatosis type-1 and sporadic paediatric optic pathway glioma

Background: The subventricular zone of the third ventricle (TVZ) is a germinal stem cell niche, identified as the possible location of optic pathway glioma (OPG) cell origin. Paediatric OPGs are predominantly diagnosed as low-grade astrocytomas, which are either sporadic or are associated with neuro...

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Main Authors: Natalie R. Boonzaier, Patrick W. Hales, Felice D'Arco, Bronwen C. Walters, Ramneek Kaur, Kshitij Mankad, Jessica Cooper, Alki Liasis, Victoria Smith, Patricia O'Hare, Darren Hargrave, Christopher A. Clark
Format: Article
Language:English
Published: Elsevier 2020-01-01
Series:NeuroImage: Clinical
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Online Access:http://www.sciencedirect.com/science/article/pii/S2213158220302849
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author Natalie R. Boonzaier
Patrick W. Hales
Felice D'Arco
Bronwen C. Walters
Ramneek Kaur
Kshitij Mankad
Jessica Cooper
Alki Liasis
Victoria Smith
Patricia O'Hare
Darren Hargrave
Christopher A. Clark
author_facet Natalie R. Boonzaier
Patrick W. Hales
Felice D'Arco
Bronwen C. Walters
Ramneek Kaur
Kshitij Mankad
Jessica Cooper
Alki Liasis
Victoria Smith
Patricia O'Hare
Darren Hargrave
Christopher A. Clark
author_sort Natalie R. Boonzaier
collection DOAJ
description Background: The subventricular zone of the third ventricle (TVZ) is a germinal stem cell niche, identified as the possible location of optic pathway glioma (OPG) cell origin. Paediatric OPGs are predominantly diagnosed as low-grade astrocytomas, which are either sporadic or are associated with neurofibromatosis type-1 (NF1). These tumours often cause a significant impairment to visual acuity (VA). Infiltrative/invasive tumour activity is associated with increased apparent diffusion coefficient (ADC) and cerebral blood flow (CBF). This study aimed to determine whether TVZ imaging features differed between sporadic-OPG, NF1-OPG and controls, and whether the ADC and CBF profile at the germinal stem cell niche (the TVZ) correlated with the primary outcome of VA. Methods: ADC and CBF MRI data were acquired from 30 paediatric OPG patients (median age 6 years; range 8 months–17 years), along with VA measurements, during clinical surveillance of their tumour. Values for mean ADC and maximum CBF were measured at the TVZ, and normalized to normal-appearing grey matter. These values were compared between the two OPG groups and the healthy control subjects, and multivariate linear regression was used to test the linear association between these values and patient’s VA. Results: In the TVZ, normalized mean ADC was higher in NF1-associated OPG patients (N = 15), compared to both sporadic OPG patients (N = 15; p = 0.010) and healthy controls (N = 14; p < 0.001). In the same region, normalized maximum CBF was higher in sporadic OPG patients compared to both NF1-OPG patients (p = 0.016) and healthy controls (p < 0.001). In sporadic OPG patients only, normalized mean ADC in the TVZ was significantly correlated with visual acuity (R2 = 0.41, p = 0.019). No significant correlations were found between TVZ CBF and ADC values and visual acuity in the NF1-associated OPG patients. Conclusion: Quantitative MRI detects TVZ abnormalities in both sporadic and NF1-OPG patients, and identifies TVZ features that differentiate the two. TVZ features may be useful MRI markers of interest in future predictive studies involving sporadic OPG.
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spelling doaj.art-61a189f666f04397863aa0c2d55d4c342022-12-21T22:33:59ZengElsevierNeuroImage: Clinical2213-15822020-01-0128102447Quantitative MRI demonstrates abnormalities of the third ventricle subventricular zone in neurofibromatosis type-1 and sporadic paediatric optic pathway gliomaNatalie R. Boonzaier0Patrick W. Hales1Felice D'Arco2Bronwen C. Walters3Ramneek Kaur4Kshitij Mankad5Jessica Cooper6Alki Liasis7Victoria Smith8Patricia O'Hare9Darren Hargrave10Christopher A. Clark11Developmental Imaging and Biophysics Section, Developmental Neurosciences, University College London Great Ormond Street Institute of Child Health, London, UKDevelopmental Imaging and Biophysics Section, Developmental Neurosciences, University College London Great Ormond Street Institute of Child Health, London, UK; Corresponding author at: Developmental Imaging &amp; Biophysics Section, UCL Great Ormond Street Institute of Child Health, 30 Guilford Street, London WC1N 1EH, UK.Great Ormond Street Hospital for Children NHS Foundation Trust, London, UKGreat Ormond Street Hospital for Children NHS Foundation Trust, London, UKDevelopmental Imaging and Biophysics Section, Developmental Neurosciences, University College London Great Ormond Street Institute of Child Health, London, UKGreat Ormond Street Hospital for Children NHS Foundation Trust, London, UKGreat Ormond Street Hospital for Children NHS Foundation Trust, London, UKGreat Ormond Street Hospital for Children NHS Foundation Trust, London, UK; University of Pittsburgh Medical Center, Children’s Hospital of Pittsburgh, Pittsburgh, USAGreat Ormond Street Hospital for Children NHS Foundation Trust, London, UKGreat Ormond Street Hospital for Children NHS Foundation Trust, London, UKDevelopmental Imaging and Biophysics Section, Developmental Neurosciences, University College London Great Ormond Street Institute of Child Health, London, UK; Great Ormond Street Hospital for Children NHS Foundation Trust, London, UKDevelopmental Imaging and Biophysics Section, Developmental Neurosciences, University College London Great Ormond Street Institute of Child Health, London, UKBackground: The subventricular zone of the third ventricle (TVZ) is a germinal stem cell niche, identified as the possible location of optic pathway glioma (OPG) cell origin. Paediatric OPGs are predominantly diagnosed as low-grade astrocytomas, which are either sporadic or are associated with neurofibromatosis type-1 (NF1). These tumours often cause a significant impairment to visual acuity (VA). Infiltrative/invasive tumour activity is associated with increased apparent diffusion coefficient (ADC) and cerebral blood flow (CBF). This study aimed to determine whether TVZ imaging features differed between sporadic-OPG, NF1-OPG and controls, and whether the ADC and CBF profile at the germinal stem cell niche (the TVZ) correlated with the primary outcome of VA. Methods: ADC and CBF MRI data were acquired from 30 paediatric OPG patients (median age 6 years; range 8 months–17 years), along with VA measurements, during clinical surveillance of their tumour. Values for mean ADC and maximum CBF were measured at the TVZ, and normalized to normal-appearing grey matter. These values were compared between the two OPG groups and the healthy control subjects, and multivariate linear regression was used to test the linear association between these values and patient’s VA. Results: In the TVZ, normalized mean ADC was higher in NF1-associated OPG patients (N = 15), compared to both sporadic OPG patients (N = 15; p = 0.010) and healthy controls (N = 14; p < 0.001). In the same region, normalized maximum CBF was higher in sporadic OPG patients compared to both NF1-OPG patients (p = 0.016) and healthy controls (p < 0.001). In sporadic OPG patients only, normalized mean ADC in the TVZ was significantly correlated with visual acuity (R2 = 0.41, p = 0.019). No significant correlations were found between TVZ CBF and ADC values and visual acuity in the NF1-associated OPG patients. Conclusion: Quantitative MRI detects TVZ abnormalities in both sporadic and NF1-OPG patients, and identifies TVZ features that differentiate the two. TVZ features may be useful MRI markers of interest in future predictive studies involving sporadic OPG.http://www.sciencedirect.com/science/article/pii/S2213158220302849Optic pathway gliomaThird ventricle subventricular zoneNeurofibromatosis type-1Quantitative magnetic resonance imagingPaediatric
spellingShingle Natalie R. Boonzaier
Patrick W. Hales
Felice D'Arco
Bronwen C. Walters
Ramneek Kaur
Kshitij Mankad
Jessica Cooper
Alki Liasis
Victoria Smith
Patricia O'Hare
Darren Hargrave
Christopher A. Clark
Quantitative MRI demonstrates abnormalities of the third ventricle subventricular zone in neurofibromatosis type-1 and sporadic paediatric optic pathway glioma
NeuroImage: Clinical
Optic pathway glioma
Third ventricle subventricular zone
Neurofibromatosis type-1
Quantitative magnetic resonance imaging
Paediatric
title Quantitative MRI demonstrates abnormalities of the third ventricle subventricular zone in neurofibromatosis type-1 and sporadic paediatric optic pathway glioma
title_full Quantitative MRI demonstrates abnormalities of the third ventricle subventricular zone in neurofibromatosis type-1 and sporadic paediatric optic pathway glioma
title_fullStr Quantitative MRI demonstrates abnormalities of the third ventricle subventricular zone in neurofibromatosis type-1 and sporadic paediatric optic pathway glioma
title_full_unstemmed Quantitative MRI demonstrates abnormalities of the third ventricle subventricular zone in neurofibromatosis type-1 and sporadic paediatric optic pathway glioma
title_short Quantitative MRI demonstrates abnormalities of the third ventricle subventricular zone in neurofibromatosis type-1 and sporadic paediatric optic pathway glioma
title_sort quantitative mri demonstrates abnormalities of the third ventricle subventricular zone in neurofibromatosis type 1 and sporadic paediatric optic pathway glioma
topic Optic pathway glioma
Third ventricle subventricular zone
Neurofibromatosis type-1
Quantitative magnetic resonance imaging
Paediatric
url http://www.sciencedirect.com/science/article/pii/S2213158220302849
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