ArticleNeurology2024
Changes in Digital Speech Measures in Asymptomatic Carriers of Pathogenic Variants Associated With Frontotemporal Degeneration.
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Article in Neurology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.
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3citing papers in PubMed
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3 citing papers in PubMed, 5 citations in OpenAlex.
- Multi-level language deficits in behavioural frontotemporal degeneration and related disorders.Brain communications · 2026Article
- Design considerations for C9orf72 disease prevention trials.Brain : a journal of neurology · 2025Review
- Developing digital health technologies for frontotemporal degeneration.Alzheimer's & dementia : the journal of the Alzheimer's Association · 2025Review
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Authors and funding
21 authors at 1 institution in 1 country.
Naomi NevlerFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0001-8745-5945
Sunghye ChoFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0003-1569-7608
Katheryn A Q CousinsFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0003-1879-2766
Sharon AshFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0002-6656-1842
Christopher A OlmFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0002-4858-2359
Sanjana ShellikeriFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0002-6436-292X
Galit AgmonFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0001-9588-5953
Carmen Gonzalez-RecoberFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0009-0001-4149-169X
Sharon X XieFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0002-7886-4103
Megan S BarkerFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0001-5385-5908
Masood ManoochehriFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0001-5818-180X
Corey T McmillanFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0002-7581-6405
David J IrwinFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0002-5599-5098
Lauren MassimoFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0002-3407-7335
Laynie DratchFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0002-2179-3199
Gayathri CheranFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.
Edward D HueyFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.
Stephanie A CosentinoFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0001-5684-143X
Vivianna M Van DeerlinFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0002-7400-9097
Mark Y LibermanFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.ORCID 0000-0002-8605-9024
Murray GrossmanFrom the Frontotemporal Degeneration Center, Department of Neurology, (N.N., K.A.Q.C., S.A., C.A.O., S.S., G.A., C.G.-R., C.T.M., D.J.I., L.M., L.D., M.G.), Linguistic Data Consortium, Department of Linguistics (S.C., M.Y.L.), Penn Image Computing and Science Laboratory, Department of Radiology (C.A.O.), Department of Biostatistics, Epidemiology and Informatics (S.X.X.), and Department of Pathology and Laboratory Medicine (V.M.V.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Taub Institute for Research on Alzheimer's Disease and the Aging Brain (M.S.B.,M.M., G.C., E.D.H., S.A.C.); and Department of Neurology (G.C., E.D.H., S.A.C.) and Gertrude H. Sergievsky Center (S.A.C.), Columbia University Irving Medical Center, New York.
Pennsylvania Academic Library Consortium · US
Funding
Research Education ComponentP30AG072979 · NIA · UNIVERSITY OF PENNSYLVANIA · PI DAVID A WOLK · 2021 to 2026
$24.8MUnderstanding Environmental Contributions to Heterogeneity in bvFTD.P01AG066597 · NIA · UNIVERSITY OF PENNSYLVANIA · PI David John Irwin, Corey T McMillan · 2020 to 2026
$18.8MGenetic basis of neuropsychiatric symptoms in Alzheimer's diseaseU01AG079850 · NIA · COLUMBIA UNIVERSITY HEALTH SCIENCES · PI Gary Wayne Beecham, Edward D Huey · 2023 to 2026
$3.0MNeuroanatomical associations with the factor structure underlying neuropsychiatric symptoms in Alzheimer's diseaseR01AG062268 · NIA · COLUMBIA UNIVERSITY HEALTH SCIENCES · PI HUEY, EDWARD D · 2018 to 2022
$2.5MUsing RDoC Negative and Positive Valence Paradigms to Investigate the Mechanisms of Neuropsychiatric Symptoms (NPS) in Alzheimer's Disease and Related DementiasR01MH120794 · NIMH · COLUMBIA UNIVERSITY HEALTH SCIENCES · PI HUEY, EDWARD D · 2019 to 2023
$2.1MNIA NIH HHS P01 AG066597NIA NIH HHS P30 AG072979NIA NIH HHS R01 AG062268NIA NIH HHS U01 AG079850NIMH NIH HHS R01 MH120794
Abstract
BACKGROUND AND
objectivesClinical trials developing therapeutics for frontotemporal degeneration (FTD) focus on pathogenic variant carriers at preclinical stages. Objective, quantitative clinical assessment tools are needed to track stability and delayed disease onset. Natural speech can serve as an accessible, cost-effective assessment tool. We aimed to identify early changes in the natural speech of FTD pathogenic variant carriers before they become symptomatic.
methodsIn this cohort study, speech samples of picture descriptions were collected longitudinally from healthy participants in observational studies at the University of Pennsylvania and Columbia University between 2007 and 2020. Participants were asymptomatic but at risk for familial FTD. Status as "carrier" or "noncarrier" was based on screening for known pathogenic variants in the participant's family. Thirty previously validated digital speech measures derived from automatic speech processing pipelines were selected a priori based on previous studies in patients with FTD and compared between asymptomatic carriers and noncarriers cross-sectionally and longitudinally.
resultsA total of 105 participants, all asymptomatic, included 41 carriers: 12 men [30%], mean age 43 ± 13 years; education, 16 ± 2 years; MMSE 29 ± 1; and 64 noncarriers: 27 men [42%]; mean age, 48 ± 14 years; education, 15 ± 3 years; MMSE 29 ± 1. We identified 4 speech measures that differed between carriers and noncarriers at baseline: mean speech segment duration (mean difference -0.28 seconds, 95% CI -0.55 to -0.02, DISCUSSION: Using automatic processing pipelines, we identified early changes in the natural speech of FTD pathogenic variant carriers in the presymptomatic stage. These findings highlight the potential utility of natural speech as a digital clinical outcome assessment tool in FTD, where objective and quantifiable measures for abnormal behavior and language are lacking.
Indexed as
Frontotemporal DementiaAdultAtrophyCohort StudiesEducational StatusFemaleHumansMaleMiddle AgedObservational Studies as TopicSpeech
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