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 A.
AU - Carson, Kathryn A.
AU - Johnston, Michael V.
AU - Huisman, Thierry A.G.M.
N1 - Funding Information:
and Disclosure: The authors thank Jeffrey Baum for his assistance with data collection. Writing assistance was provided by Robert Schoen, PharmD, and Traci Stuve, MA (ApotheCom, Yardley, PA, USA), and supported by Novartis. K. A. Carson's work on the project was funded by the Johns Hopkins Institute for Clinical and Translational Research, under grant number UL1 TR001079 from the National Center for Advancing Translational Sciences, a component of the National Institutes of Health (NIH) and the NIH Roadmap for Medical Research. T. T. Gipson's work on the project was supported by grant number 2K12NS001696-11A from the National Institute of Neurological Disorders and Stroke. A. Poretti, S. A. Kelley, M. V. Johnston, and T. A. G. M. Huisman report no conflicts of interest. Funding for editorial support was provided by Novartis Pharmaceuticals.
Funding Information:
Acknowledgments and Disclosure: The authors thank Jeffrey Baum for his assistance with data collection. Writing assistance was provided by Robert Schoen, PharmD, and Traci Stuve, MA (ApotheCom, Yardley, PA, USA), and supported by Novartis. K. A. Carson’s work on the project was funded by the Johns Hopkins Institute for Clinical and Translational Research, under grant number UL1 TR001079 from the National Center for Advancing Translational Sciences, a component of the National Institutes of Health (NIH) and the NIH Roadmap for Medical Research. T. T. Gipson’s work on the project was supported by grant number 2K12NS001696-11A from the National Institute of Neurological Disorders and Stroke. A. Poretti, S. A. Kelley, M. V. Johnston, and T. A. G. M. Huisman report no conflicts of interest. Funding for editorial support was provided by Novartis Pharmaceuticals.
Publisher Copyright:
© 2019 The Authors Journal of Neuroimaging published by Wiley Periodicals, Inc. on behalf of American Society of Neuroimaging
PY - 2019/7/1
Y1 - 2019/7/1
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.
KW - Tuberous sclerosis complex
KW - autism
KW - behavior
KW - diffusion tensor imaging
KW - intellectual disability
KW - self-injurious behavior
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U2 - 10.1111/jon.12628
DO - 10.1111/jon.12628
M3 - Article
C2 - 31056796
AN - SCOPUS:85065316133
SN - 1051-2284
VL - 29
SP - 506
EP - 511
JO - Journal of Neuroimaging
JF - Journal of Neuroimaging
IS - 4
ER -