Focal white matter changes in spasmodic dysphonia: A combined diffusion tensor imaging and neuropathological study

Kristina Simonyan, Fernanda Tovar-Moll, John Ostuni, Mark Hallett, Victor F. Kalasinsky, Michael R. Lewin-Smith, Elisabeth J. Rushing, Alexander Vortmeyer, Christy L. Ludlow

Research output: Contribution to journalArticle

87 Citations (Scopus)

Abstract

Spasmodic dysphonia is a neurological disorder characterized by involuntary spasms in the laryngeal muscles during speech production. Although the clinical symptoms are well characterized, the pathophysiology of this voice disorder is unknown. We describe here, for the first time to our knowledge, disorder-specific brain abnormalities in these patients as determined by a combined approach of diffusion tensor imaging (DTI) and postmortem histopathology. We used DTI to identify brain changes and to target those brain regions for neuropathological examination. DTI showed right-sided decrease of fractional anisotropy in the genu of the internal capsule and bilateral increase of overall water diffusivity in the white matter along the corticobulbar/ corticospinal tract in 20 spasmodic dysphonia patients compared to 20 healthy subjects. In addition, water diffusivity was bilaterally increased in the lentiform nucleus, ventral thalamus and cerebellar white and grey matter in the patients. These brain changes were substantiated with focal histopathological abnormalities presented as a loss of axonal density and myelin content in the right genu of the internal capsule and clusters of mineral depositions, containing calcium, phosphorus and iron, in the parenchyma and vessel walls of the posterior limb of the internal capsule, putamen, globus pallidus and cerebellum in the postmortem brain tissue from one patient compared to three controls. The specificity of these brain abnormalities is confirmed by their localization, limited only to the corticobulbar/corticospinal tract and its main input/output structures. We also found positive correlation between the diffusivity changes and clinical symptoms of spasmodic dysphonia (r = 0.509, P = 0.037). These brain abnormalities may alter the central control of voluntary voice production and, therefore, may underlie the pathophysiology of this disorder.

Original languageEnglish (US)
Pages (from-to)447-459
Number of pages13
JournalBrain
Volume131
Issue number2
DOIs
StatePublished - Jan 1 2008
Externally publishedYes

Fingerprint

Dysphonia
Diffusion Tensor Imaging
Pyramidal Tracts
Internal Capsule
Brain
Voice Disorders
Laryngeal Muscles
Corpus Striatum
Globus Pallidus
Water
Putamen
Anisotropy
Spasm
Brain Diseases
Myelin Sheath
Nervous System Diseases
Thalamus
Phosphorus
Cerebellum
Minerals

Keywords

  • Basal ganglia
  • Corticobulbar tract
  • Laryngeal dystonia
  • Neuroimaging
  • Neuropathology

ASJC Scopus subject areas

  • Clinical Neurology

Cite this

Simonyan, K., Tovar-Moll, F., Ostuni, J., Hallett, M., Kalasinsky, V. F., Lewin-Smith, M. R., ... Ludlow, C. L. (2008). Focal white matter changes in spasmodic dysphonia: A combined diffusion tensor imaging and neuropathological study. Brain, 131(2), 447-459. https://doi.org/10.1093/brain/awm303

Focal white matter changes in spasmodic dysphonia : A combined diffusion tensor imaging and neuropathological study. / Simonyan, Kristina; Tovar-Moll, Fernanda; Ostuni, John; Hallett, Mark; Kalasinsky, Victor F.; Lewin-Smith, Michael R.; Rushing, Elisabeth J.; Vortmeyer, Alexander; Ludlow, Christy L.

In: Brain, Vol. 131, No. 2, 01.01.2008, p. 447-459.

Research output: Contribution to journalArticle

Simonyan, K, Tovar-Moll, F, Ostuni, J, Hallett, M, Kalasinsky, VF, Lewin-Smith, MR, Rushing, EJ, Vortmeyer, A & Ludlow, CL 2008, 'Focal white matter changes in spasmodic dysphonia: A combined diffusion tensor imaging and neuropathological study', Brain, vol. 131, no. 2, pp. 447-459. https://doi.org/10.1093/brain/awm303
Simonyan K, Tovar-Moll F, Ostuni J, Hallett M, Kalasinsky VF, Lewin-Smith MR et al. Focal white matter changes in spasmodic dysphonia: A combined diffusion tensor imaging and neuropathological study. Brain. 2008 Jan 1;131(2):447-459. https://doi.org/10.1093/brain/awm303
Simonyan, Kristina ; Tovar-Moll, Fernanda ; Ostuni, John ; Hallett, Mark ; Kalasinsky, Victor F. ; Lewin-Smith, Michael R. ; Rushing, Elisabeth J. ; Vortmeyer, Alexander ; Ludlow, Christy L. / Focal white matter changes in spasmodic dysphonia : A combined diffusion tensor imaging and neuropathological study. In: Brain. 2008 ; Vol. 131, No. 2. pp. 447-459.
@article{ea53ef75222f41f3800a695bb037ecf5,
title = "Focal white matter changes in spasmodic dysphonia: A combined diffusion tensor imaging and neuropathological study",
abstract = "Spasmodic dysphonia is a neurological disorder characterized by involuntary spasms in the laryngeal muscles during speech production. Although the clinical symptoms are well characterized, the pathophysiology of this voice disorder is unknown. We describe here, for the first time to our knowledge, disorder-specific brain abnormalities in these patients as determined by a combined approach of diffusion tensor imaging (DTI) and postmortem histopathology. We used DTI to identify brain changes and to target those brain regions for neuropathological examination. DTI showed right-sided decrease of fractional anisotropy in the genu of the internal capsule and bilateral increase of overall water diffusivity in the white matter along the corticobulbar/ corticospinal tract in 20 spasmodic dysphonia patients compared to 20 healthy subjects. In addition, water diffusivity was bilaterally increased in the lentiform nucleus, ventral thalamus and cerebellar white and grey matter in the patients. These brain changes were substantiated with focal histopathological abnormalities presented as a loss of axonal density and myelin content in the right genu of the internal capsule and clusters of mineral depositions, containing calcium, phosphorus and iron, in the parenchyma and vessel walls of the posterior limb of the internal capsule, putamen, globus pallidus and cerebellum in the postmortem brain tissue from one patient compared to three controls. The specificity of these brain abnormalities is confirmed by their localization, limited only to the corticobulbar/corticospinal tract and its main input/output structures. We also found positive correlation between the diffusivity changes and clinical symptoms of spasmodic dysphonia (r = 0.509, P = 0.037). These brain abnormalities may alter the central control of voluntary voice production and, therefore, may underlie the pathophysiology of this disorder.",
keywords = "Basal ganglia, Corticobulbar tract, Laryngeal dystonia, Neuroimaging, Neuropathology",
author = "Kristina Simonyan and Fernanda Tovar-Moll and John Ostuni and Mark Hallett and Kalasinsky, {Victor F.} and Lewin-Smith, {Michael R.} and Rushing, {Elisabeth J.} and Alexander Vortmeyer and Ludlow, {Christy L.}",
year = "2008",
month = "1",
day = "1",
doi = "10.1093/brain/awm303",
language = "English (US)",
volume = "131",
pages = "447--459",
journal = "Brain",
issn = "0006-8950",
publisher = "Oxford University Press",
number = "2",

}

TY - JOUR

T1 - Focal white matter changes in spasmodic dysphonia

T2 - A combined diffusion tensor imaging and neuropathological study

AU - Simonyan, Kristina

AU - Tovar-Moll, Fernanda

AU - Ostuni, John

AU - Hallett, Mark

AU - Kalasinsky, Victor F.

AU - Lewin-Smith, Michael R.

AU - Rushing, Elisabeth J.

AU - Vortmeyer, Alexander

AU - Ludlow, Christy L.

PY - 2008/1/1

Y1 - 2008/1/1

N2 - Spasmodic dysphonia is a neurological disorder characterized by involuntary spasms in the laryngeal muscles during speech production. Although the clinical symptoms are well characterized, the pathophysiology of this voice disorder is unknown. We describe here, for the first time to our knowledge, disorder-specific brain abnormalities in these patients as determined by a combined approach of diffusion tensor imaging (DTI) and postmortem histopathology. We used DTI to identify brain changes and to target those brain regions for neuropathological examination. DTI showed right-sided decrease of fractional anisotropy in the genu of the internal capsule and bilateral increase of overall water diffusivity in the white matter along the corticobulbar/ corticospinal tract in 20 spasmodic dysphonia patients compared to 20 healthy subjects. In addition, water diffusivity was bilaterally increased in the lentiform nucleus, ventral thalamus and cerebellar white and grey matter in the patients. These brain changes were substantiated with focal histopathological abnormalities presented as a loss of axonal density and myelin content in the right genu of the internal capsule and clusters of mineral depositions, containing calcium, phosphorus and iron, in the parenchyma and vessel walls of the posterior limb of the internal capsule, putamen, globus pallidus and cerebellum in the postmortem brain tissue from one patient compared to three controls. The specificity of these brain abnormalities is confirmed by their localization, limited only to the corticobulbar/corticospinal tract and its main input/output structures. We also found positive correlation between the diffusivity changes and clinical symptoms of spasmodic dysphonia (r = 0.509, P = 0.037). These brain abnormalities may alter the central control of voluntary voice production and, therefore, may underlie the pathophysiology of this disorder.

AB - Spasmodic dysphonia is a neurological disorder characterized by involuntary spasms in the laryngeal muscles during speech production. Although the clinical symptoms are well characterized, the pathophysiology of this voice disorder is unknown. We describe here, for the first time to our knowledge, disorder-specific brain abnormalities in these patients as determined by a combined approach of diffusion tensor imaging (DTI) and postmortem histopathology. We used DTI to identify brain changes and to target those brain regions for neuropathological examination. DTI showed right-sided decrease of fractional anisotropy in the genu of the internal capsule and bilateral increase of overall water diffusivity in the white matter along the corticobulbar/ corticospinal tract in 20 spasmodic dysphonia patients compared to 20 healthy subjects. In addition, water diffusivity was bilaterally increased in the lentiform nucleus, ventral thalamus and cerebellar white and grey matter in the patients. These brain changes were substantiated with focal histopathological abnormalities presented as a loss of axonal density and myelin content in the right genu of the internal capsule and clusters of mineral depositions, containing calcium, phosphorus and iron, in the parenchyma and vessel walls of the posterior limb of the internal capsule, putamen, globus pallidus and cerebellum in the postmortem brain tissue from one patient compared to three controls. The specificity of these brain abnormalities is confirmed by their localization, limited only to the corticobulbar/corticospinal tract and its main input/output structures. We also found positive correlation between the diffusivity changes and clinical symptoms of spasmodic dysphonia (r = 0.509, P = 0.037). These brain abnormalities may alter the central control of voluntary voice production and, therefore, may underlie the pathophysiology of this disorder.

KW - Basal ganglia

KW - Corticobulbar tract

KW - Laryngeal dystonia

KW - Neuroimaging

KW - Neuropathology

UR - http://www.scopus.com/inward/record.url?scp=38849191142&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=38849191142&partnerID=8YFLogxK

U2 - 10.1093/brain/awm303

DO - 10.1093/brain/awm303

M3 - Article

C2 - 18083751

AN - SCOPUS:38849191142

VL - 131

SP - 447

EP - 459

JO - Brain

JF - Brain

SN - 0006-8950

IS - 2

ER -