Resting-State fMRI Networks in Children with Tuberous Sclerosis Complex

on behalf of the TACERN Study Group

doi:10.1111/jon.12653

Resting-State fMRI Networks in Children with Tuberous Sclerosis Complex

on behalf of the TACERN Study Group

Pediatrics

Research output: Contribution to journal › Article › peer-review

6 Scopus citations

Abstract

BACKGROUND AND PURPOSE: There are no published studies examining resting state networks (RSNs) and their relationship with neurodevelopmental metrics in tuberous sclerosis complex (TSC). We aimed to identify major resting-state functional magnetic resonance imaging (rs-fMRI) networks in infants with TSC and correlate network analyses with neurodevelopmental assessments, autism diagnosis, and seizure history. METHODS: Rs-fMRI data from 34 infants with TSC, sedated with propofol during the scan, were analyzed to identify auditory, motor, and visual RSNs. We examined the correlations between auditory, motor, and visual RSNs at approximately 11.5 months, neurodevelopmental outcome at approximately 18.5 months, and diagnosis of autism spectrum disorders at approximately 36 months of age. RESULTS: RSNs were obtained in 76.5% (26/34) of infants. We observed significant negative correlations between auditory RSN and auditory comprehension test scores (p =.038; r = −.435), as well as significant positive correlations between motor RSN and gross motor skills test scores (p =.023; r =.564). Significant positive correlations between motor RSNs and gross motor skills (p =.012; r =.754) were observed in TSC infants without autism, but not in TSC infants with autism, which could suggest altered motor processing. There were no significant differences in RSNs according to seizure history. CONCLUSIONS: Negative correlation between auditory RSN, as well as positive correlation between motor RSN and developmental outcome measures might reflect different brain mechanisms and, when identified, may be helpful in predicting later function. A larger study of TSC patients with a healthy control group is needed before auditory and motor RSNs could be considered as neurodevelopmental outcome biomarkers.

Original language	English (US)
Pages (from-to)	750-759
Number of pages	10
Journal	Journal of Neuroimaging
Volume	29
Issue number	6
DOIs	https://doi.org/10.1111/jon.12653
State	Published - Nov 1 2019

Funding

Research reported in this publication was supported by the National Institute of Neurological Disorders And Stroke of the National Institutes of Health (NINDS) and Eunice Kennedy Shriver National Institute Of Child Health & Human Development (NICHD) under Award Numbers U01NS082320 and P20-NS080199, the Tuberous Sclerosis Alliance, as well as in part by NIH grants R01 NS079788, R01 EB019483, and U54 HD090255. Darcy A. Krueger has received support from Novartis Pharmaceuticals. Joyce Y. Wu has received research funding from Novartis, GW Pharmaceutical, NINDS/NIH (R01 NS082649, U01 NS082320, U54 NS092090, U01 NS092595), and the Today's and Tomorrow's Children Fund from UCLA Mattel Children's Hospital at the University of California Los Angeles. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.We are sincerely indebted to the generosity of the families and patients in TSC clinics across the United States who contributed their time and effort to this study. We would also like to thank the Tuberous Sclerosis Alliance for their continued support in TSC research. We also would like to thank Borjan Gagoski, PhD; Amalya Wilson; Silvina Ferradal, PhD; and Matthew Hutchison, PhD for their assistance.We also would like to acknowledge the members of the Tuberous Sclerosis Autism Center of Excellence Research Network (TACERN) whose names are not in the main author list: Monisha Goyal, MD, Birmingham, AL; Deborah A. Pearson, PhD, Houston, TX; Marian E. Williams, PhD, Los Angeles, CA; Ellen Hanson, PhD, Boston, MA; Nicole Bing, PsyD, Cincinnati, OH; Bridget Kent, MA, CCC-SLP, Cincinnati, OH; Sarah O'Kelley, PhD, Birmingham, AL; Rajna Filip-Dhima, MS, Boston, MA; Kira Dies, ScM, CGC, Boston, MA; Stephanie Bruns, CCRP, Cincinnati, OH; Benoit Scherrer, PhD, Boston, MA; Gary Cutter, PhD, Birmingham, AL; Donna S. Murray, PhD, Boston, MA; Steven L. Roberds, PhD, Silver Spring, MD.

Keywords

Autism spectrum disorders
propofol
resting state functional resonance imaging
resting state networks
tuberous sclerosis complex

ASJC Scopus subject areas

Radiology Nuclear Medicine and imaging
Clinical Neurology

Access to Document

10.1111/jon.12653

Cite this

@article{dd870135356e4640bd82f35a11723f6f,

title = "Resting-State fMRI Networks in Children with Tuberous Sclerosis Complex",

abstract = "BACKGROUND AND PURPOSE: There are no published studies examining resting state networks (RSNs) and their relationship with neurodevelopmental metrics in tuberous sclerosis complex (TSC). We aimed to identify major resting-state functional magnetic resonance imaging (rs-fMRI) networks in infants with TSC and correlate network analyses with neurodevelopmental assessments, autism diagnosis, and seizure history. METHODS: Rs-fMRI data from 34 infants with TSC, sedated with propofol during the scan, were analyzed to identify auditory, motor, and visual RSNs. We examined the correlations between auditory, motor, and visual RSNs at approximately 11.5 months, neurodevelopmental outcome at approximately 18.5 months, and diagnosis of autism spectrum disorders at approximately 36 months of age. RESULTS: RSNs were obtained in 76.5% (26/34) of infants. We observed significant negative correlations between auditory RSN and auditory comprehension test scores (p =.038; r = −.435), as well as significant positive correlations between motor RSN and gross motor skills test scores (p =.023; r =.564). Significant positive correlations between motor RSNs and gross motor skills (p =.012; r =.754) were observed in TSC infants without autism, but not in TSC infants with autism, which could suggest altered motor processing. There were no significant differences in RSNs according to seizure history. CONCLUSIONS: Negative correlation between auditory RSN, as well as positive correlation between motor RSN and developmental outcome measures might reflect different brain mechanisms and, when identified, may be helpful in predicting later function. A larger study of TSC patients with a healthy control group is needed before auditory and motor RSNs could be considered as neurodevelopmental outcome biomarkers.",

keywords = "Autism spectrum disorders, propofol, resting state functional resonance imaging, resting state networks, tuberous sclerosis complex",

author = "{on behalf of the TACERN Study Group} and Banu Ahtam and Mathieu Dehaes and Sliva, {Danielle D.} and Peters, {Jurriaan M.} and Krueger, {Darcy A.} and Bebin, {Elizabeth Martina} and Hope Northrup and Wu, {Joyce Y.} and Warfield, {Simon K.} and Mustafa Sahin and Grant, {Patricia Ellen}",

note = "Funding Information: Research reported in this publication was supported by the National Institute of Neurological Disorders And Stroke of the National Institutes of Health (NINDS) and Eunice Kennedy Shriver National Institute Of Child Health & Human Development (NICHD) under Award Numbers U01NS082320 and P20-NS080199, the Tuberous Sclerosis Alliance, as well as in part by NIH grants R01 NS079788, R01 EB019483, and U54 HD090255. Darcy A. Krueger has received support from Novartis Pharmaceuticals. Joyce Y. Wu has received research funding from Novartis, GW Pharmaceutical, NINDS/NIH (R01 NS082649, U01 NS082320, U54 NS092090, U01 NS092595), and the Today's and Tomorrow's Children Fund from UCLA Mattel Children's Hospital at the University of California Los Angeles. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.We are sincerely indebted to the generosity of the families and patients in TSC clinics across the United States who contributed their time and effort to this study. We would also like to thank the Tuberous Sclerosis Alliance for their continued support in TSC research. We also would like to thank Borjan Gagoski, PhD; Amalya Wilson; Silvina Ferradal, PhD; and Matthew Hutchison, PhD for their assistance.We also would like to acknowledge the members of the Tuberous Sclerosis Autism Center of Excellence Research Network (TACERN) whose names are not in the main author list: Monisha Goyal, MD, Birmingham, AL; Deborah A. Pearson, PhD, Houston, TX; Marian E. Williams, PhD, Los Angeles, CA; Ellen Hanson, PhD, Boston, MA; Nicole Bing, PsyD, Cincinnati, OH; Bridget Kent, MA, CCC-SLP, Cincinnati, OH; Sarah O'Kelley, PhD, Birmingham, AL; Rajna Filip-Dhima, MS, Boston, MA; Kira Dies, ScM, CGC, Boston, MA; Stephanie Bruns, CCRP, Cincinnati, OH; Benoit Scherrer, PhD, Boston, MA; Gary Cutter, PhD, Birmingham, AL; Donna S. Murray, PhD, Boston, MA; Steven L. Roberds, PhD, Silver Spring, MD. Publisher Copyright: {\textcopyright} 2019 by the American Society of Neuroimaging",

year = "2019",

month = nov,

day = "1",

doi = "10.1111/jon.12653",

language = "English (US)",

volume = "29",

pages = "750--759",

journal = "Journal of Neuroimaging",

issn = "1051-2284",

publisher = "Wiley-Blackwell",

number = "6",

}

TY - JOUR

T1 - Resting-State fMRI Networks in Children with Tuberous Sclerosis Complex

AU - on behalf of the TACERN Study Group

AU - Ahtam, Banu

AU - Dehaes, Mathieu

AU - Sliva, Danielle D.

AU - Peters, Jurriaan M.

AU - Krueger, Darcy A.

AU - Bebin, Elizabeth Martina

AU - Northrup, Hope

AU - Wu, Joyce Y.

AU - Warfield, Simon K.

AU - Sahin, Mustafa

AU - Grant, Patricia Ellen

N1 - Funding Information: Research reported in this publication was supported by the National Institute of Neurological Disorders And Stroke of the National Institutes of Health (NINDS) and Eunice Kennedy Shriver National Institute Of Child Health & Human Development (NICHD) under Award Numbers U01NS082320 and P20-NS080199, the Tuberous Sclerosis Alliance, as well as in part by NIH grants R01 NS079788, R01 EB019483, and U54 HD090255. Darcy A. Krueger has received support from Novartis Pharmaceuticals. Joyce Y. Wu has received research funding from Novartis, GW Pharmaceutical, NINDS/NIH (R01 NS082649, U01 NS082320, U54 NS092090, U01 NS092595), and the Today's and Tomorrow's Children Fund from UCLA Mattel Children's Hospital at the University of California Los Angeles. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.We are sincerely indebted to the generosity of the families and patients in TSC clinics across the United States who contributed their time and effort to this study. We would also like to thank the Tuberous Sclerosis Alliance for their continued support in TSC research. We also would like to thank Borjan Gagoski, PhD; Amalya Wilson; Silvina Ferradal, PhD; and Matthew Hutchison, PhD for their assistance.We also would like to acknowledge the members of the Tuberous Sclerosis Autism Center of Excellence Research Network (TACERN) whose names are not in the main author list: Monisha Goyal, MD, Birmingham, AL; Deborah A. Pearson, PhD, Houston, TX; Marian E. Williams, PhD, Los Angeles, CA; Ellen Hanson, PhD, Boston, MA; Nicole Bing, PsyD, Cincinnati, OH; Bridget Kent, MA, CCC-SLP, Cincinnati, OH; Sarah O'Kelley, PhD, Birmingham, AL; Rajna Filip-Dhima, MS, Boston, MA; Kira Dies, ScM, CGC, Boston, MA; Stephanie Bruns, CCRP, Cincinnati, OH; Benoit Scherrer, PhD, Boston, MA; Gary Cutter, PhD, Birmingham, AL; Donna S. Murray, PhD, Boston, MA; Steven L. Roberds, PhD, Silver Spring, MD. Publisher Copyright: © 2019 by the American Society of Neuroimaging

PY - 2019/11/1

Y1 - 2019/11/1

N2 - BACKGROUND AND PURPOSE: There are no published studies examining resting state networks (RSNs) and their relationship with neurodevelopmental metrics in tuberous sclerosis complex (TSC). We aimed to identify major resting-state functional magnetic resonance imaging (rs-fMRI) networks in infants with TSC and correlate network analyses with neurodevelopmental assessments, autism diagnosis, and seizure history. METHODS: Rs-fMRI data from 34 infants with TSC, sedated with propofol during the scan, were analyzed to identify auditory, motor, and visual RSNs. We examined the correlations between auditory, motor, and visual RSNs at approximately 11.5 months, neurodevelopmental outcome at approximately 18.5 months, and diagnosis of autism spectrum disorders at approximately 36 months of age. RESULTS: RSNs were obtained in 76.5% (26/34) of infants. We observed significant negative correlations between auditory RSN and auditory comprehension test scores (p =.038; r = −.435), as well as significant positive correlations between motor RSN and gross motor skills test scores (p =.023; r =.564). Significant positive correlations between motor RSNs and gross motor skills (p =.012; r =.754) were observed in TSC infants without autism, but not in TSC infants with autism, which could suggest altered motor processing. There were no significant differences in RSNs according to seizure history. CONCLUSIONS: Negative correlation between auditory RSN, as well as positive correlation between motor RSN and developmental outcome measures might reflect different brain mechanisms and, when identified, may be helpful in predicting later function. A larger study of TSC patients with a healthy control group is needed before auditory and motor RSNs could be considered as neurodevelopmental outcome biomarkers.

AB - BACKGROUND AND PURPOSE: There are no published studies examining resting state networks (RSNs) and their relationship with neurodevelopmental metrics in tuberous sclerosis complex (TSC). We aimed to identify major resting-state functional magnetic resonance imaging (rs-fMRI) networks in infants with TSC and correlate network analyses with neurodevelopmental assessments, autism diagnosis, and seizure history. METHODS: Rs-fMRI data from 34 infants with TSC, sedated with propofol during the scan, were analyzed to identify auditory, motor, and visual RSNs. We examined the correlations between auditory, motor, and visual RSNs at approximately 11.5 months, neurodevelopmental outcome at approximately 18.5 months, and diagnosis of autism spectrum disorders at approximately 36 months of age. RESULTS: RSNs were obtained in 76.5% (26/34) of infants. We observed significant negative correlations between auditory RSN and auditory comprehension test scores (p =.038; r = −.435), as well as significant positive correlations between motor RSN and gross motor skills test scores (p =.023; r =.564). Significant positive correlations between motor RSNs and gross motor skills (p =.012; r =.754) were observed in TSC infants without autism, but not in TSC infants with autism, which could suggest altered motor processing. There were no significant differences in RSNs according to seizure history. CONCLUSIONS: Negative correlation between auditory RSN, as well as positive correlation between motor RSN and developmental outcome measures might reflect different brain mechanisms and, when identified, may be helpful in predicting later function. A larger study of TSC patients with a healthy control group is needed before auditory and motor RSNs could be considered as neurodevelopmental outcome biomarkers.

KW - Autism spectrum disorders

KW - propofol

KW - resting state functional resonance imaging

KW - resting state networks

KW - tuberous sclerosis complex

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ER -

Resting-State fMRI Networks in Children with Tuberous Sclerosis Complex

Abstract

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