Atrophy in midbrain & cerebral/cerebellar pedunculi is characteristic for progressive supranuclear palsy – A double-validation whole-brain meta-analysis

Franziska Albrecht, Sandrine Bisenius, Jane Neumann, Jennifer Lynn Whitwell, Matthias L. Schroeter

Research output: Contribution to journalArticle

Abstract

Objective: Progressive supranuclear palsy (PSP) is an atypical parkinsonian syndrome characterized by vertical gaze palsy and postural instability. Midbrain atrophy is suggested as a hallmark, but it has not been validated systematically in whole-brain imaging. Methods: We conducted whole-brain meta-analyses identifying disease-related atrophy in structural MRI. Eighteen studies were identified (N = 315 PSP, 393 controls) and separated into gray or white matter analyses (15/12). All patients were diagnosed according to the National Institute of Neurological Disorders and Stroke and the Society for PSP (NINDS-SPSP criteria, Litvan et al. (1996a)), which are now considered as PSP-Richardson syndrome (Höglinger et al., 2017). With overlay analyses, we double-validated two meta-analytical algorithms: anatomical likelihood estimation and seed-based D mapping. Additionally, we conducted region-of-interest effect size meta-analyses on radiological biomarkers and subtraction analyses differentiating PSP from Parkinson's disease. Results: Whole brain meta-analyses revealed consistent gray matter atrophy in bilateral thalamus, anterior insulae, midbrain, and left caudate nucleus. White matter alterations were consistently detected in bilateral superior/middle cerebellar pedunculi, cerebral pedunculi, and midbrain atrophy. Region-of-interest meta-analyses demonstrated that midbrain metrics generally perform very well in distinguishing PSP from other parkinsonian syndromes with strong effect sizes. Subtraction analyses identified the midbrain as differentiating between PSP and Parkinson's disease. Conclusions: Our meta-analyses identify gray matter atrophy of the midbrain and white matter atrophy of the cerebral/cerebellar pedunculi and midbrain as characteristic for PSP. Results support the incorporation of structural MRI data, and particularly these structures, into the revised PSP diagnostic criteria.

Original languageEnglish (US)
Article number101722
JournalNeuroImage: Clinical
Volume22
DOIs
StatePublished - Jan 1 2019

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Progressive Supranuclear Palsy
Mesencephalon
Atrophy
Meta-Analysis
Brain
National Institute of Neurological Disorders and Stroke
Parkinsonian Disorders
Parkinson Disease
Caudate Nucleus
Thalamus
Neuroimaging
Paralysis
Seeds
Biomarkers

Keywords

  • Anatomical likelihood estimation
  • Cerebellar pedunculi
  • Cerebral pedunculi
  • Imaging biomarker
  • Meta-analysis
  • Midbrain
  • Progressive supranuclear palsy
  • Seed-based D mapping

ASJC Scopus subject areas

  • Radiology Nuclear Medicine and imaging
  • Neurology
  • Clinical Neurology
  • Cognitive Neuroscience

Cite this

Atrophy in midbrain & cerebral/cerebellar pedunculi is characteristic for progressive supranuclear palsy – A double-validation whole-brain meta-analysis. / Albrecht, Franziska; Bisenius, Sandrine; Neumann, Jane; Whitwell, Jennifer Lynn; Schroeter, Matthias L.

In: NeuroImage: Clinical, Vol. 22, 101722, 01.01.2019.

Research output: Contribution to journalArticle

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abstract = "Objective: Progressive supranuclear palsy (PSP) is an atypical parkinsonian syndrome characterized by vertical gaze palsy and postural instability. Midbrain atrophy is suggested as a hallmark, but it has not been validated systematically in whole-brain imaging. Methods: We conducted whole-brain meta-analyses identifying disease-related atrophy in structural MRI. Eighteen studies were identified (N = 315 PSP, 393 controls) and separated into gray or white matter analyses (15/12). All patients were diagnosed according to the National Institute of Neurological Disorders and Stroke and the Society for PSP (NINDS-SPSP criteria, Litvan et al. (1996a)), which are now considered as PSP-Richardson syndrome (H{\"o}glinger et al., 2017). With overlay analyses, we double-validated two meta-analytical algorithms: anatomical likelihood estimation and seed-based D mapping. Additionally, we conducted region-of-interest effect size meta-analyses on radiological biomarkers and subtraction analyses differentiating PSP from Parkinson's disease. Results: Whole brain meta-analyses revealed consistent gray matter atrophy in bilateral thalamus, anterior insulae, midbrain, and left caudate nucleus. White matter alterations were consistently detected in bilateral superior/middle cerebellar pedunculi, cerebral pedunculi, and midbrain atrophy. Region-of-interest meta-analyses demonstrated that midbrain metrics generally perform very well in distinguishing PSP from other parkinsonian syndromes with strong effect sizes. Subtraction analyses identified the midbrain as differentiating between PSP and Parkinson's disease. Conclusions: Our meta-analyses identify gray matter atrophy of the midbrain and white matter atrophy of the cerebral/cerebellar pedunculi and midbrain as characteristic for PSP. Results support the incorporation of structural MRI data, and particularly these structures, into the revised PSP diagnostic criteria.",
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