Cited by 127Neurobiology of Language and BilingualismDementia and Cognitive Impairment ResearchVoice and Speech Disorders

Retraining speech production and fluency in non-fluent/agrammatic primary progressive aphasia

Open access
Henry, Maya L, Hubbard, H Isabel, Grasso, Stephanie M, Mandelli, Maria Luisa, Wilson, Stephen M, Sathishkumar, Mithra T, Fridriksson, Julius, Daigle, Wylin +3 more·2018·Brain

Abstract

The non-fluent/agrammatic variant of primary progressive aphasia (nfvPPA) presents with a gradual decline in grammar and motor speech resulting from selective degeneration of speech-language regions in the brain. There has been considerable progress in identifying treatment approaches to remediate language deficits in other primary progressive aphasia variants; however, interventions for the core deficits in nfvPPA have yet to be systematically investigated. Further, the neural mechanisms that support behavioural restitution in the context of neurodegeneration are not well understood. We examined the immediate and long-term benefits of video implemented script training for aphasia (VISTA) in 10 individuals with nfvPPA. The treatment approach involved repeated rehearsal of individualized scripts via structured treatment with a clinician as well as intensive home practice with an audiovisual model using 'speech entrainment'. We evaluated accuracy of script production as well as overall intelligibility and grammaticality for trained and untrained scripts. These measures and standardized test scores were collected at post-treatment and 3-, 6-, and 12-month follow-up visits. Treatment resulted in significant improvement in production of correct, intelligible scripted words for trained topics, a reduction in grammatical errors for trained topics, and an overall increase in intelligibility for trained as well as untrained topics at post-treatment. Follow-up testing revealed maintenance of gains for trained scripts up to 1 year post-treatment on the primary outcome measure. Performance on untrained scripts and standardized tests remained relatively stable during the follow-up period, indicating that treatment helped to stabilize speech and language despite disease progression. To identify neural predictors of responsiveness to intervention, we examined treatment effect sizes relative to grey matter volumes in regions of interest derived from a previously identified speech production network. Regions of significant atrophy within this network included bilateral inferior frontal cortices and supplementary motor area as well as left striatum. Volumes in a left middle/inferior temporal region of interest were significantly correlated with the magnitude of treatment effects. This region, which was relatively spared anatomically in nfvPPA patients, has been implicated in syntactic production as well as visuo-motor facilitation of speech. This is the first group study to document the benefits of behavioural intervention that targets both linguistic and motoric deficits in nfvPPA. Findings indicate that behavioural intervention may result in lasting and generalized improvement of communicative function in individuals with neurodegenerative disease and that the integrity of spared regions within the speech-language network may be an important predictor of treatment response.

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  • For VISTA, despite significant declines relative to post-treatment, no measures showed a significant decline relative to pre-treatment at any time point.

    “Despite significant declines in performance relative to post-treatment, no measures showed a significant decline in performance relative to pre-treatment at any time point.”

    From <p>Investigating the utility of teletherapy in individuals with primary progressive aphasia</p> (2019) · cites this paper as [48]

    The ‘Stability of treatment effects at follow-up time points through 1 year post-treatment’ section reports that the group’s primary outcome remained significantly above pretreatment at every follow-up, while other measures were either above pretreatment or not significantly different from it. It describes a steady decline in two standardized tests after treatment, but does not establish the claimed significant declines relative to post-treatment. The claim is supported for the group-level pretreatment comparisons, not for that significance qualifier. (LLM, conf=0.84)

    Experimental — accuracy not yet measured
  • VISTA resulted in significant improvement in the production of correct, intelligible scripted words for trained scripts relative to pre-treatment, with gains maintained up to 1-year follow-up.

    “As reported in Henry et al,48 analyses of group data revealed a significant improvement in the production of correct, intelligible scripted words for trained scripts relative to pre-treatment, with gains maintained up to 1-year follow-up.”
  • VISTA produced no significant changes on the NAT or WAB AQ.

    “No significant changes were observed on the NAT or WAB AQ.”

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Research works printed but not linked: 3

  1. [2]2. Andrews G, Howie PM, Dozsa M, Guitar BE. Stuttering: speech pat- tern characteristics under fluency-inducing conditions. J Speech Lang Hear Res 1982; 25: 208-16.
  2. [11]11. Carthery-Goulart MT, da Silveira AD, Machado TH, Mansur LL, Parente MA, Senaha MLH, et al. Nonpharmacological interventions for cognitive impairments following primary progressive aphasia. Dement Neuropsychol 2013; 7: 121-31.
  3. [64]64. Mandelli ML, Vilaplana E, Brown JA, Hubbard HI, Binney RJ, Attyagalle S, et al. Healthy brain connectivity predicts atrophy pro- gression in nonfluent variant of primary progressive aphasia. Brain 2016; 139: 2778-91.

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Authors
Henry, M. L., Hubbard, H. I., Grasso, S. M., Mandelli, M. L., Wilson, S. M., Sathishkumar, M. T., Fridriksson, J., Daigle, W., Boxer, A. L., Miller, B. L., & Gorno-Tempini, M. L.
Affiliations
  • The University of Texas at Austin (US)
  • University of Alberta (CA)
  • University of California, San Francisco (US)
  • Vanderbilt University Medical Center (US)
  • University of South Carolina (US)
  • Department of Neurology, Memory and Aging Center, University of California San Francisco, CA, USA
Corresponding author
Henry, Maya L
Venue
Brain
Published
2018-04-30
Volume
141
Issue
6
Pages
1799-1814
ISSN
  • 0006-8950 (ISSN-L)
  • 1460-2156
OpenAlex
W2800976177
Cited by
127
References
99
Citations by year
2026: 12 · 2025: 13 · 2024: 15 · 2023: 17 · 2022: 12 · 2021: 24 · 2020: 15 · 2019: 16 · 2018: 2
FWCI
8.49
Type
journal article
Language
en
Open access
Locations
  • Brain (publishedVersion)
  • Europe PMC (PubMed Central) (publishedVersion)
  • PubMed Central (publishedVersion)
  • PubMed (publishedVersion)
  • eScholarship (California Digital Library) (submittedVersion)
Funders
National Institutes of Health (M.H., NIDCD R01 DC016291, M.H., NIDCD R03 DC013403, M.GT., NINDS R01 NS050915, M.GT., NIDCD K24 DC015544, S.W., NIDCD R01 DC013270, S.W., NIDCD R21 DC016080, B.M., NIA P01 AG019724, B.M., NIA P50 AG023501, A.B., NINDS U54 NS092089, A.B., NIA R01 AG038791, J.F., NIDCD P50 DC014664)
Topics
  • Life Sciences › Neuroscience › Cognitive Neuroscience › Neurobiology of Language and Bilingualism
  • Health Sciences › Medicine › Psychiatry and Mental health › Dementia and Cognitive Impairment Research
  • Health Sciences › Medicine › Physiology › Voice and Speech Disorders
Keywords
aphasia rehabilitation, speech entrainment, speech production, fluency, intelligibility, grammaticality, gray matter atrophy, predictors of treatment response, supplementary motor area, inferior frontal cortex, neurodegeneration, behavioral interventions
MeSH terms
Aged, Aphasia, Wernicke, Female, Follow-Up Studies, Humans, Image Processing, Computer-Assisted, Magnetic Resonance Imaging, Male, Middle Aged, Neuropsychological Tests, Speech, Speech Therapy, Treatment Outcome, Aphasia, Primary Progressive

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