Characterization of the Basal Ganglia Using Diffusion Tensor Imaging in Children with Self-Injurious Behavior and Tuberous Sclerosis Complex

Tanjala T. Gipson, Andrea Poretti, Sarah Aminoff Kelley, Kathryn Anne Carson, Michael V Johnston, Thierry A.G.M. Huisman

Research output: Contribution to journalArticle

Abstract

BACKGROUND AND PURPOSE: Tuberous sclerosis complex (TSC) is a rare, genetic disease that is associated with multiple manifestations including epilepsy and autism. Self-injurious behaviors (SIBs) also occur in a subset of patients. This study used diffusion tensor imaging (DTI) in children with TSC for quantitative and volumetric analysis of brain regions that have been associated with SIB in other genetic conditions. METHODS: We used DTI to compare 6 children with TSC-associated SIB and 10 children with TSC without SIB. Atlas-based analysis of DTI data and calculation of number of voxels; fractional anisotropy (FA); and mean, axial, and radial diffusivity were performed for multiple regions; DTI measures were summarized using medians and interquartile ranges and were compared using Wilcoxon rank sum tests and false discovery rates (FDRs). RESULTS: Analysis showed that children with TSC and SIB had reduced numbers of voxels (median) in the bilateral globus pallidus (right: 218 vs. 260, P =.008, FDR =.18; left: 222 vs. 274, P =.002, FDR =.12) and caudate nucleus (right: 712 vs. 896, P =.01, FDR =.26; left: 702 vs. 921, P =.03, FDR =.44) and reduced FA in the bilateral globus pallidus (right:.233 vs.272, P =.003, FDR =.12; left:.223 vs.247, P =.004, FDR =.12) and left caudate nucleus (.162 vs.186, P =.03, FDR =.39) versus children without SIB. No other statistically significant differences were found. CONCLUSIONS: These data support a correlation between lower volumes of the globus pallidus and caudate with SIB in children with TSC.

Original languageEnglish (US)
JournalJournal of Neuroimaging
DOIs
StatePublished - Jan 1 2019

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Self-Injurious Behavior
Tuberous Sclerosis
Diffusion Tensor Imaging
Basal Ganglia
Globus Pallidus
Caudate Nucleus
Anisotropy
Nonparametric Statistics
Inborn Genetic Diseases
Atlases
Autistic Disorder
Rare Diseases
Epilepsy
Brain

Keywords

  • autism
  • behavior
  • diffusion tensor imaging
  • intellectual disability
  • self-injurious behavior
  • Tuberous sclerosis complex

ASJC Scopus subject areas

  • Radiology Nuclear Medicine and imaging
  • Clinical Neurology

Cite this

@article{e15e415426ea41f7bc680ca912651fd8,
title = "Characterization of the Basal Ganglia Using Diffusion Tensor Imaging in Children with Self-Injurious Behavior and Tuberous Sclerosis Complex",
abstract = "BACKGROUND AND PURPOSE: Tuberous sclerosis complex (TSC) is a rare, genetic disease that is associated with multiple manifestations including epilepsy and autism. Self-injurious behaviors (SIBs) also occur in a subset of patients. This study used diffusion tensor imaging (DTI) in children with TSC for quantitative and volumetric analysis of brain regions that have been associated with SIB in other genetic conditions. METHODS: We used DTI to compare 6 children with TSC-associated SIB and 10 children with TSC without SIB. Atlas-based analysis of DTI data and calculation of number of voxels; fractional anisotropy (FA); and mean, axial, and radial diffusivity were performed for multiple regions; DTI measures were summarized using medians and interquartile ranges and were compared using Wilcoxon rank sum tests and false discovery rates (FDRs). RESULTS: Analysis showed that children with TSC and SIB had reduced numbers of voxels (median) in the bilateral globus pallidus (right: 218 vs. 260, P =.008, FDR =.18; left: 222 vs. 274, P =.002, FDR =.12) and caudate nucleus (right: 712 vs. 896, P =.01, FDR =.26; left: 702 vs. 921, P =.03, FDR =.44) and reduced FA in the bilateral globus pallidus (right:.233 vs.272, P =.003, FDR =.12; left:.223 vs.247, P =.004, FDR =.12) and left caudate nucleus (.162 vs.186, P =.03, FDR =.39) versus children without SIB. No other statistically significant differences were found. CONCLUSIONS: These data support a correlation between lower volumes of the globus pallidus and caudate with SIB in children with TSC.",
keywords = "autism, behavior, diffusion tensor imaging, intellectual disability, self-injurious behavior, Tuberous sclerosis complex",
author = "Gipson, {Tanjala T.} and Andrea Poretti and Kelley, {Sarah Aminoff} and Carson, {Kathryn Anne} and Johnston, {Michael V} and Huisman, {Thierry A.G.M.}",
year = "2019",
month = "1",
day = "1",
doi = "10.1111/jon.12628",
language = "English (US)",
journal = "Journal of Neuroimaging",
issn = "1051-2284",
publisher = "Wiley-Blackwell",

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TY - JOUR

T1 - Characterization of the Basal Ganglia Using Diffusion Tensor Imaging in Children with Self-Injurious Behavior and Tuberous Sclerosis Complex

AU - Gipson, Tanjala T.

AU - Poretti, Andrea

AU - Kelley, Sarah Aminoff

AU - Carson, Kathryn Anne

AU - Johnston, Michael V

AU - Huisman, Thierry A.G.M.

PY - 2019/1/1

Y1 - 2019/1/1

N2 - BACKGROUND AND PURPOSE: Tuberous sclerosis complex (TSC) is a rare, genetic disease that is associated with multiple manifestations including epilepsy and autism. Self-injurious behaviors (SIBs) also occur in a subset of patients. This study used diffusion tensor imaging (DTI) in children with TSC for quantitative and volumetric analysis of brain regions that have been associated with SIB in other genetic conditions. METHODS: We used DTI to compare 6 children with TSC-associated SIB and 10 children with TSC without SIB. Atlas-based analysis of DTI data and calculation of number of voxels; fractional anisotropy (FA); and mean, axial, and radial diffusivity were performed for multiple regions; DTI measures were summarized using medians and interquartile ranges and were compared using Wilcoxon rank sum tests and false discovery rates (FDRs). RESULTS: Analysis showed that children with TSC and SIB had reduced numbers of voxels (median) in the bilateral globus pallidus (right: 218 vs. 260, P =.008, FDR =.18; left: 222 vs. 274, P =.002, FDR =.12) and caudate nucleus (right: 712 vs. 896, P =.01, FDR =.26; left: 702 vs. 921, P =.03, FDR =.44) and reduced FA in the bilateral globus pallidus (right:.233 vs.272, P =.003, FDR =.12; left:.223 vs.247, P =.004, FDR =.12) and left caudate nucleus (.162 vs.186, P =.03, FDR =.39) versus children without SIB. No other statistically significant differences were found. CONCLUSIONS: These data support a correlation between lower volumes of the globus pallidus and caudate with SIB in children with TSC.

AB - BACKGROUND AND PURPOSE: Tuberous sclerosis complex (TSC) is a rare, genetic disease that is associated with multiple manifestations including epilepsy and autism. Self-injurious behaviors (SIBs) also occur in a subset of patients. This study used diffusion tensor imaging (DTI) in children with TSC for quantitative and volumetric analysis of brain regions that have been associated with SIB in other genetic conditions. METHODS: We used DTI to compare 6 children with TSC-associated SIB and 10 children with TSC without SIB. Atlas-based analysis of DTI data and calculation of number of voxels; fractional anisotropy (FA); and mean, axial, and radial diffusivity were performed for multiple regions; DTI measures were summarized using medians and interquartile ranges and were compared using Wilcoxon rank sum tests and false discovery rates (FDRs). RESULTS: Analysis showed that children with TSC and SIB had reduced numbers of voxels (median) in the bilateral globus pallidus (right: 218 vs. 260, P =.008, FDR =.18; left: 222 vs. 274, P =.002, FDR =.12) and caudate nucleus (right: 712 vs. 896, P =.01, FDR =.26; left: 702 vs. 921, P =.03, FDR =.44) and reduced FA in the bilateral globus pallidus (right:.233 vs.272, P =.003, FDR =.12; left:.223 vs.247, P =.004, FDR =.12) and left caudate nucleus (.162 vs.186, P =.03, FDR =.39) versus children without SIB. No other statistically significant differences were found. CONCLUSIONS: These data support a correlation between lower volumes of the globus pallidus and caudate with SIB in children with TSC.

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KW - behavior

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KW - intellectual disability

KW - self-injurious behavior

KW - Tuberous sclerosis complex

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