TY - JOUR
T1 - Atrophy in midbrain & cerebral/cerebellar pedunculi is characteristic for progressive supranuclear palsy – A double-validation whole-brain meta-analysis
AU - Albrecht, Franziska
AU - Bisenius, Sandrine
AU - Neumann, Jane
AU - Whitwell, Jennifer
AU - Schroeter, Matthias L.
N1 - Funding Information:
This study has been supported by the Parkinson's Disease Foundation ( PDF-IRG-1307 ), the Michael J Fox Foundation ( MJFF-11362 ), the German Federal Ministry of Education and Research (BMBF; FKZ 01GI1007A ; German FTLD consortium), the German Research Foundation (DFG, SCHR 774/5-1 ), the National Institutes of Health (NIH, R01-NS89757 ), and the International Max Planck Research School (IMPRS) NeuroCom by the Max Planck Society. Jane Neumann is supported by the Federal Ministry of Education and Research Germany (BMBF, FKZ: 01EO1001 ), and the German Research Foundation ( DFG-SFB1052 ).
Publisher Copyright:
© 2019 The Authors
PY - 2019/1/1
Y1 - 2019/1/1
N2 - 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.
AB - 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.
KW - Anatomical likelihood estimation
KW - Cerebellar pedunculi
KW - Cerebral pedunculi
KW - Imaging biomarker
KW - Meta-analysis
KW - Midbrain
KW - Progressive supranuclear palsy
KW - Seed-based D mapping
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U2 - 10.1016/j.nicl.2019.101722
DO - 10.1016/j.nicl.2019.101722
M3 - Article
C2 - 30831462
AN - SCOPUS:85062212725
SN - 2213-1582
VL - 22
JO - NeuroImage: Clinical
JF - NeuroImage: Clinical
M1 - 101722
ER -