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November 01, 2022; 99 (18) Research Article
CLINICAL AND STRUCTURAL FINDINGS IN PATIENTS WITH LESION-INDUCED DYSTONIA
DESCRIPTIVE AND QUANTITATIVE ANALYSIS OF PUBLISHED CASES
Daniel T. Corp, View ORCID ProfileChristopher J. Greenwood, Jordan Morrison-Ham,
View ORCID ProfileJaakko Pullinen, Georgia M. McDowall, Ellen F. P. Younger,
Hyder A. Jinnah, View ORCID ProfileMichael D. Fox, Juho Joutsa
First published August 17, 2022, DOI:
https://doi.org/10.1212/WNL.0000000000201042
Daniel T. Corp
From the Cognitive Neuroscience Unit (D.T.C., J.M.-H., G.M., E.Y.), School of
Psychology, Deakin University, Geelong, Australia; Center for Brain Circuit
Therapeutics (D.T.C., M.D.F., J.J.), Brigham and Women's Hospital, Boston, MA;
Deakin University (C.G.), Centre for Social and Early Emotional Development,
School of Psychology, Faculty of Health, Geelong, Australia; Murdoch Children's
Research Institute (C.G.), Centre for Adolescent Health, Melbourne, Australia;
Turku Brain and Mind Center (J.P., J.J.), Clinical Neurosciences, University of
Turku, Finland; Departments of Neurology and Human Genetics (H.J.), Emory
University, School of Medicine, Atlanta, GA; Department of Neurology (M.D.F.),
Harvard Medical School, Boston, MA; and Turku PET Centre (J.J.), Neurocenter,
Turku University Hospital, Finland.
* Find this author on Google Scholar
* Find this author on PubMed
* Search for this author on this site
Christopher J. Greenwood
From the Cognitive Neuroscience Unit (D.T.C., J.M.-H., G.M., E.Y.), School of
Psychology, Deakin University, Geelong, Australia; Center for Brain Circuit
Therapeutics (D.T.C., M.D.F., J.J.), Brigham and Women's Hospital, Boston, MA;
Deakin University (C.G.), Centre for Social and Early Emotional Development,
School of Psychology, Faculty of Health, Geelong, Australia; Murdoch Children's
Research Institute (C.G.), Centre for Adolescent Health, Melbourne, Australia;
Turku Brain and Mind Center (J.P., J.J.), Clinical Neurosciences, University of
Turku, Finland; Departments of Neurology and Human Genetics (H.J.), Emory
University, School of Medicine, Atlanta, GA; Department of Neurology (M.D.F.),
Harvard Medical School, Boston, MA; and Turku PET Centre (J.J.), Neurocenter,
Turku University Hospital, Finland.
* Find this author on Google Scholar
* Find this author on PubMed
* Search for this author on this site
* ORCID record for Christopher J. Greenwood
Jordan Morrison-Ham
From the Cognitive Neuroscience Unit (D.T.C., J.M.-H., G.M., E.Y.), School of
Psychology, Deakin University, Geelong, Australia; Center for Brain Circuit
Therapeutics (D.T.C., M.D.F., J.J.), Brigham and Women's Hospital, Boston, MA;
Deakin University (C.G.), Centre for Social and Early Emotional Development,
School of Psychology, Faculty of Health, Geelong, Australia; Murdoch Children's
Research Institute (C.G.), Centre for Adolescent Health, Melbourne, Australia;
Turku Brain and Mind Center (J.P., J.J.), Clinical Neurosciences, University of
Turku, Finland; Departments of Neurology and Human Genetics (H.J.), Emory
University, School of Medicine, Atlanta, GA; Department of Neurology (M.D.F.),
Harvard Medical School, Boston, MA; and Turku PET Centre (J.J.), Neurocenter,
Turku University Hospital, Finland.
* Find this author on Google Scholar
* Find this author on PubMed
* Search for this author on this site
Jaakko Pullinen
From the Cognitive Neuroscience Unit (D.T.C., J.M.-H., G.M., E.Y.), School of
Psychology, Deakin University, Geelong, Australia; Center for Brain Circuit
Therapeutics (D.T.C., M.D.F., J.J.), Brigham and Women's Hospital, Boston, MA;
Deakin University (C.G.), Centre for Social and Early Emotional Development,
School of Psychology, Faculty of Health, Geelong, Australia; Murdoch Children's
Research Institute (C.G.), Centre for Adolescent Health, Melbourne, Australia;
Turku Brain and Mind Center (J.P., J.J.), Clinical Neurosciences, University of
Turku, Finland; Departments of Neurology and Human Genetics (H.J.), Emory
University, School of Medicine, Atlanta, GA; Department of Neurology (M.D.F.),
Harvard Medical School, Boston, MA; and Turku PET Centre (J.J.), Neurocenter,
Turku University Hospital, Finland.
* Find this author on Google Scholar
* Find this author on PubMed
* Search for this author on this site
* ORCID record for Jaakko Pullinen
Georgia M. McDowall
From the Cognitive Neuroscience Unit (D.T.C., J.M.-H., G.M., E.Y.), School of
Psychology, Deakin University, Geelong, Australia; Center for Brain Circuit
Therapeutics (D.T.C., M.D.F., J.J.), Brigham and Women's Hospital, Boston, MA;
Deakin University (C.G.), Centre for Social and Early Emotional Development,
School of Psychology, Faculty of Health, Geelong, Australia; Murdoch Children's
Research Institute (C.G.), Centre for Adolescent Health, Melbourne, Australia;
Turku Brain and Mind Center (J.P., J.J.), Clinical Neurosciences, University of
Turku, Finland; Departments of Neurology and Human Genetics (H.J.), Emory
University, School of Medicine, Atlanta, GA; Department of Neurology (M.D.F.),
Harvard Medical School, Boston, MA; and Turku PET Centre (J.J.), Neurocenter,
Turku University Hospital, Finland.
* Find this author on Google Scholar
* Find this author on PubMed
* Search for this author on this site
Ellen F. P. Younger
From the Cognitive Neuroscience Unit (D.T.C., J.M.-H., G.M., E.Y.), School of
Psychology, Deakin University, Geelong, Australia; Center for Brain Circuit
Therapeutics (D.T.C., M.D.F., J.J.), Brigham and Women's Hospital, Boston, MA;
Deakin University (C.G.), Centre for Social and Early Emotional Development,
School of Psychology, Faculty of Health, Geelong, Australia; Murdoch Children's
Research Institute (C.G.), Centre for Adolescent Health, Melbourne, Australia;
Turku Brain and Mind Center (J.P., J.J.), Clinical Neurosciences, University of
Turku, Finland; Departments of Neurology and Human Genetics (H.J.), Emory
University, School of Medicine, Atlanta, GA; Department of Neurology (M.D.F.),
Harvard Medical School, Boston, MA; and Turku PET Centre (J.J.), Neurocenter,
Turku University Hospital, Finland.
* Find this author on Google Scholar
* Find this author on PubMed
* Search for this author on this site
Hyder A. Jinnah
From the Cognitive Neuroscience Unit (D.T.C., J.M.-H., G.M., E.Y.), School of
Psychology, Deakin University, Geelong, Australia; Center for Brain Circuit
Therapeutics (D.T.C., M.D.F., J.J.), Brigham and Women's Hospital, Boston, MA;
Deakin University (C.G.), Centre for Social and Early Emotional Development,
School of Psychology, Faculty of Health, Geelong, Australia; Murdoch Children's
Research Institute (C.G.), Centre for Adolescent Health, Melbourne, Australia;
Turku Brain and Mind Center (J.P., J.J.), Clinical Neurosciences, University of
Turku, Finland; Departments of Neurology and Human Genetics (H.J.), Emory
University, School of Medicine, Atlanta, GA; Department of Neurology (M.D.F.),
Harvard Medical School, Boston, MA; and Turku PET Centre (J.J.), Neurocenter,
Turku University Hospital, Finland.
* Find this author on Google Scholar
* Find this author on PubMed
* Search for this author on this site
Michael D. Fox
From the Cognitive Neuroscience Unit (D.T.C., J.M.-H., G.M., E.Y.), School of
Psychology, Deakin University, Geelong, Australia; Center for Brain Circuit
Therapeutics (D.T.C., M.D.F., J.J.), Brigham and Women's Hospital, Boston, MA;
Deakin University (C.G.), Centre for Social and Early Emotional Development,
School of Psychology, Faculty of Health, Geelong, Australia; Murdoch Children's
Research Institute (C.G.), Centre for Adolescent Health, Melbourne, Australia;
Turku Brain and Mind Center (J.P., J.J.), Clinical Neurosciences, University of
Turku, Finland; Departments of Neurology and Human Genetics (H.J.), Emory
University, School of Medicine, Atlanta, GA; Department of Neurology (M.D.F.),
Harvard Medical School, Boston, MA; and Turku PET Centre (J.J.), Neurocenter,
Turku University Hospital, Finland.
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* ORCID record for Michael D. Fox
Juho Joutsa
From the Cognitive Neuroscience Unit (D.T.C., J.M.-H., G.M., E.Y.), School of
Psychology, Deakin University, Geelong, Australia; Center for Brain Circuit
Therapeutics (D.T.C., M.D.F., J.J.), Brigham and Women's Hospital, Boston, MA;
Deakin University (C.G.), Centre for Social and Early Emotional Development,
School of Psychology, Faculty of Health, Geelong, Australia; Murdoch Children's
Research Institute (C.G.), Centre for Adolescent Health, Melbourne, Australia;
Turku Brain and Mind Center (J.P., J.J.), Clinical Neurosciences, University of
Turku, Finland; Departments of Neurology and Human Genetics (H.J.), Emory
University, School of Medicine, Atlanta, GA; Department of Neurology (M.D.F.),
Harvard Medical School, Boston, MA; and Turku PET Centre (J.J.), Neurocenter,
Turku University Hospital, Finland.
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ABSTRACT
Background and Objectives Brain lesions are a well-recognized etiology of
dystonia. These cases are especially valuable because they offer causal insight
into the neuroanatomical substrates of dystonia. To date, knowledge of
lesion-induced dystonia comes mainly from isolated case reports or small case
series, restricting broader description and analysis.
Methods Cases of lesion-induced dystonia were first identified from a systematic
review of published literature. Latent class analysis then investigated whether
patients could be classified into subgroups based on lesion location and body
regions affected by dystonia. Regression analyses subsequently investigated
whether subgroup membership predicted clinical characteristics of dystonia.
Results Three hundred fifty-nine published cases were included. Lesions causing
dystonia occurred in heterogeneous locations, most commonly in the basal ganglia
(46.2%), followed by the thalamus (28.1%), brainstem (22.6%), and white matter
(21.2%). The most common form of lesion-induced dystonia was focal dystonia
(53.2%), with the hand (49.9%) and arm (44.3%) most commonly affected. Of all
cases, 86.6% reported co-occurring neurologic manifestations and 26.1% reported
other movement disorders. Latent class analysis identified 3 distinct subgroups
of patients: those with predominantly limb dystonias, which were associated with
basal ganglia lesions; those with hand dystonia, associated with thalamic
lesions; and those with predominantly cervical dystonia, associated with
brainstem and cerebellar lesions. Regression demonstrated significant
differences between these subgroups on a range of dystonia symptoms, including
dystonic tremor, symptom latency, other movement disorders, and dystonia
variability.
Discussion Although dystonia can be induced by lesions to numerous brain
regions, there are distinct relationships between lesion locations and dystonic
body parts. This suggests that the affected brain networks are different between
types of dystonia.
GLOSSARY
AIC=Akaike information criterion; BIC=Bayesian information criterion; LCA=latent
class analysis; LMR=Lo-Mendell-Rubin
FOOTNOTES
* Go to Neurology.org/N for full disclosures. Funding information and
disclosures deemed relevant by the authors, if any, are provided at the end
of the article.
* Submitted and externally peer reviewed. The handling editor was Peter Hedera,
MD, PhD.
* Editorial, page 777
* CME Course: NPub.org/cmelist
* Received November 17, 2021.
* Accepted in final form June 15, 2022.
* © 2022 American Academy of Neurology
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Citation Toolsclose
Clinical and Structural Findings in Patients With Lesion-Induced Dystonia
Descriptive and Quantitative Analysis of Published Cases
Daniel T. Corp, Christopher J. Greenwood, Jordan Morrison-Ham, Jaakko Pullinen,
Georgia M. McDowall, Ellen F. P. Younger, Hyder A. Jinnah, Michael D. Fox, Juho
Joutsa
Neurology Nov 2022, 99 (18) e1957-e1967; DOI: 10.1212/WNL.0000000000201042
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