Affected functional networks associated with sentence production in classic galactosemia

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Abstract

Patients with the inherited metabolic disorder classic galactosemia have language production impairments in several planning stages. Here, we assessed potential deviations in recruitment and connectivity across brain areas responsible for language production that may explain these deficits. We used functional magnetic resonance imaging (fMRI) to study neural activity and connectivity while participants carried out a language production task. This study included 13 adolescent patients and 13 age- and gender-matched healthy controls. Participants passively watched or actively described an animated visual scene using two conditions, varying in syntactic complexity (single words versus a sentence). Results showed that patients recruited additional and more extensive brain regions during sentence production. Both groups showed modulations with syntactic complexity in left inferior frontal gyrus (IFG), a region associated with syntactic planning, and in right insula. In addition, patients showed a modulation with syntax in left superior temporal gyrus (STG), whereas the controls did not. Further, patients showed increased activity in right STG and right supplementary motor area (SMA). The functional connectivity data showed similar patterns, with more extensive connectivity with frontal and motor regions, and restricted and weaker connectivity with superior temporal regions. Patients also showed higher baseline cerebral blood flow (CBF) in right IFG and trends towards higher CBF in bilateral STG, SMA and the insula. Taken together, the data demonstrate that language abnormalities in classic galactosemia are associated with specific changes within the language network. These changes point towards impairments related to both syntactic planning and speech motor planning in these patients. (C) 2015 Elsevier B.V. All rights reserved.
Original languageEnglish
Pages (from-to)166-176
Number of pages11
JournalBrain Research
Volume1616
Early online date13 May 2015
DOIs
Publication statusPublished - 7 Aug 2015

Keywords

  • Classic galactosemia
  • Functional magnetic resonance imaging
  • Functional connectivity
  • Language production
  • Syntactic encoding
  • LANGUAGE IMPAIRMENT
  • SYNTACTIC STRUCTURE
  • SPEECH PRODUCTION
  • FMRI
  • CHILDREN
  • COMPREHENSION
  • DISORDERS
  • PHENOTYPE
  • MUTATION
  • APRAXIA

Cite this

@article{06b88f50a436466aafce86964c8cda06,
title = "Affected functional networks associated with sentence production in classic galactosemia",
abstract = "Patients with the inherited metabolic disorder classic galactosemia have language production impairments in several planning stages. Here, we assessed potential deviations in recruitment and connectivity across brain areas responsible for language production that may explain these deficits. We used functional magnetic resonance imaging (fMRI) to study neural activity and connectivity while participants carried out a language production task. This study included 13 adolescent patients and 13 age- and gender-matched healthy controls. Participants passively watched or actively described an animated visual scene using two conditions, varying in syntactic complexity (single words versus a sentence). Results showed that patients recruited additional and more extensive brain regions during sentence production. Both groups showed modulations with syntactic complexity in left inferior frontal gyrus (IFG), a region associated with syntactic planning, and in right insula. In addition, patients showed a modulation with syntax in left superior temporal gyrus (STG), whereas the controls did not. Further, patients showed increased activity in right STG and right supplementary motor area (SMA). The functional connectivity data showed similar patterns, with more extensive connectivity with frontal and motor regions, and restricted and weaker connectivity with superior temporal regions. Patients also showed higher baseline cerebral blood flow (CBF) in right IFG and trends towards higher CBF in bilateral STG, SMA and the insula. Taken together, the data demonstrate that language abnormalities in classic galactosemia are associated with specific changes within the language network. These changes point towards impairments related to both syntactic planning and speech motor planning in these patients. (C) 2015 Elsevier B.V. All rights reserved.",
keywords = "Classic galactosemia, Functional magnetic resonance imaging, Functional connectivity, Language production, Syntactic encoding, LANGUAGE IMPAIRMENT, SYNTACTIC STRUCTURE, SPEECH PRODUCTION, FMRI, CHILDREN, COMPREHENSION, DISORDERS, PHENOTYPE, MUTATION, APRAXIA",
author = "I. Timmers and {van den Hurk}, J. and P.A.M. Hofman and L.J.I. Zimmermann and K. Uludag and B.M. Jansma and M.E. Rubio-Gozalbo",
year = "2015",
month = "8",
day = "7",
doi = "10.1016/j.brainres.2015.05.007",
language = "English",
volume = "1616",
pages = "166--176",
journal = "Brain Research",
issn = "0006-8993",
publisher = "Elsevier",

}

Affected functional networks associated with sentence production in classic galactosemia. / Timmers, I.; van den Hurk, J.; Hofman, P.A.M.; Zimmermann, L.J.I.; Uludag, K.; Jansma, B.M.; Rubio-Gozalbo, M.E.

In: Brain Research, Vol. 1616, 07.08.2015, p. 166-176.

Research output: Contribution to journalArticleAcademicpeer-review

TY - JOUR

T1 - Affected functional networks associated with sentence production in classic galactosemia

AU - Timmers, I.

AU - van den Hurk, J.

AU - Hofman, P.A.M.

AU - Zimmermann, L.J.I.

AU - Uludag, K.

AU - Jansma, B.M.

AU - Rubio-Gozalbo, M.E.

PY - 2015/8/7

Y1 - 2015/8/7

N2 - Patients with the inherited metabolic disorder classic galactosemia have language production impairments in several planning stages. Here, we assessed potential deviations in recruitment and connectivity across brain areas responsible for language production that may explain these deficits. We used functional magnetic resonance imaging (fMRI) to study neural activity and connectivity while participants carried out a language production task. This study included 13 adolescent patients and 13 age- and gender-matched healthy controls. Participants passively watched or actively described an animated visual scene using two conditions, varying in syntactic complexity (single words versus a sentence). Results showed that patients recruited additional and more extensive brain regions during sentence production. Both groups showed modulations with syntactic complexity in left inferior frontal gyrus (IFG), a region associated with syntactic planning, and in right insula. In addition, patients showed a modulation with syntax in left superior temporal gyrus (STG), whereas the controls did not. Further, patients showed increased activity in right STG and right supplementary motor area (SMA). The functional connectivity data showed similar patterns, with more extensive connectivity with frontal and motor regions, and restricted and weaker connectivity with superior temporal regions. Patients also showed higher baseline cerebral blood flow (CBF) in right IFG and trends towards higher CBF in bilateral STG, SMA and the insula. Taken together, the data demonstrate that language abnormalities in classic galactosemia are associated with specific changes within the language network. These changes point towards impairments related to both syntactic planning and speech motor planning in these patients. (C) 2015 Elsevier B.V. All rights reserved.

AB - Patients with the inherited metabolic disorder classic galactosemia have language production impairments in several planning stages. Here, we assessed potential deviations in recruitment and connectivity across brain areas responsible for language production that may explain these deficits. We used functional magnetic resonance imaging (fMRI) to study neural activity and connectivity while participants carried out a language production task. This study included 13 adolescent patients and 13 age- and gender-matched healthy controls. Participants passively watched or actively described an animated visual scene using two conditions, varying in syntactic complexity (single words versus a sentence). Results showed that patients recruited additional and more extensive brain regions during sentence production. Both groups showed modulations with syntactic complexity in left inferior frontal gyrus (IFG), a region associated with syntactic planning, and in right insula. In addition, patients showed a modulation with syntax in left superior temporal gyrus (STG), whereas the controls did not. Further, patients showed increased activity in right STG and right supplementary motor area (SMA). The functional connectivity data showed similar patterns, with more extensive connectivity with frontal and motor regions, and restricted and weaker connectivity with superior temporal regions. Patients also showed higher baseline cerebral blood flow (CBF) in right IFG and trends towards higher CBF in bilateral STG, SMA and the insula. Taken together, the data demonstrate that language abnormalities in classic galactosemia are associated with specific changes within the language network. These changes point towards impairments related to both syntactic planning and speech motor planning in these patients. (C) 2015 Elsevier B.V. All rights reserved.

KW - Classic galactosemia

KW - Functional magnetic resonance imaging

KW - Functional connectivity

KW - Language production

KW - Syntactic encoding

KW - LANGUAGE IMPAIRMENT

KW - SYNTACTIC STRUCTURE

KW - SPEECH PRODUCTION

KW - FMRI

KW - CHILDREN

KW - COMPREHENSION

KW - DISORDERS

KW - PHENOTYPE

KW - MUTATION

KW - APRAXIA

U2 - 10.1016/j.brainres.2015.05.007

DO - 10.1016/j.brainres.2015.05.007

M3 - Article

VL - 1616

SP - 166

EP - 176

JO - Brain Research

JF - Brain Research

SN - 0006-8993

ER -