Longitudinal anatomic, functional, and molecular characterization of Pick disease phenotypes.


Journal

Neurology
ISSN: 1526-632X
Titre abrégé: Neurology
Pays: United States
ID NLM: 0401060

Informations de publication

Date de publication:
15 12 2020
Historique:
received: 17 04 2020
accepted: 30 07 2020
pubmed: 30 9 2020
medline: 26 1 2021
entrez: 29 9 2020
Statut: ppublish

Résumé

To characterize longitudinal MRI and PET abnormalities in autopsy-confirmed Pick disease (PiD) and determine how patterns of neurodegeneration differ with respect to clinical syndrome. Seventeen patients with PiD were identified who had antemortem MRI (8 with behavioral variant frontotemporal dementia [bvFTD-PiD], 6 with nonfluent/agrammatic primary progressive aphasia [naPPA-PiD], 1 with semantic primary progressive aphasia, 1 with unclassified primary progressive aphasia, and 1 with corticobasal syndrome). Thirteen patients had serial MRI for a total of 56 MRIs, 7 had [ The bvFTD-PiD and naPPA-PiD groups showed different foci of volume loss and hypometabolism early in the disease, with bvFTD-PiD involving bilateral prefrontal and anterior temporal cortices and naPPA-PiD involving left inferior frontal gyrus, insula, and orbitofrontal cortex. However, patterns merged over time, with progressive spread into prefrontal and anterior temporal lobe in naPPA-PiD, and eventual involvement of posterior temporal lobe, motor cortex, and parietal lobe in both groups. Rates of frontotemporal atrophy were faster in bvFTD-PiD than naPPA-PiD. One patient was β-amyloid-positive on PET with low Alzheimer neuropathologic changes at autopsy. Flortaucipir PET showed elevated uptake in frontotemporal white matter. Patterns of atrophy and hypometabolism differ in PiD according to presenting syndrome, although patterns of neurodegeneration appear to converge over time.

Identifiants

pubmed: 32989107
pii: WNL.0000000000010948
doi: 10.1212/WNL.0000000000010948
pmc: PMC7836669
doi:

Substances chimiques

Amyloid beta-Peptides 0

Types de publication

Journal Article Research Support, N.I.H., Extramural

Langues

eng

Sous-ensembles de citation

IM

Pagination

e3190-e3202

Subventions

Organisme : NIDCD NIH HHS
ID : R01 DC014942
Pays : United States
Organisme : NIA NIH HHS
ID : R01 AG011378
Pays : United States
Organisme : NIDCD NIH HHS
ID : R01 DC012519
Pays : United States
Organisme : NIDCD NIH HHS
ID : R01 DC010367
Pays : United States
Organisme : NIA NIH HHS
ID : P30 AG062677
Pays : United States
Organisme : NINDS NIH HHS
ID : R21 NS094684
Pays : United States
Organisme : NIA NIH HHS
ID : U01 AG006786
Pays : United States

Informations de copyright

© 2020 American Academy of Neurology.

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Auteurs

Jennifer L Whitwell (JL)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL. Whitwell.jennifer@mayo.edu.

Nirubol Tosakulwong (N)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Christopher C Schwarz (CC)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Matthew L Senjem (ML)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Anthony J Spychalla (AJ)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Joseph R Duffy (JR)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Jon Graff-Radford (J)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Mary M Machulda (MM)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Bradley F Boeve (BF)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

David S Knopman (DS)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Ronald C Petersen (RC)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Val J Lowe (VJ)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Clifford R Jack (CR)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Dennis W Dickson (DW)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Joseph E Parisi (JE)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

Keith A Josephs (KA)

From the Departments of Radiology (J.L.W., C.C.S., M.L.S., A.J.S., V.J.L., C.R.J.), Health Sciences Research (N.T.), Neurology (J.R.D., J.G.-R., B.F.B., D.S.K., R.C.P., K.A.J.), Psychiatry and Psychology (M.M.M.), and Neuropathology (J.E.P.), Mayo Clinic, Rochester, MN; and Department of Neuropathology (D.W.D.), Mayo Clinic, Jacksonville, FL.

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