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

TitleRetraining speech production and fluency in non-fluent/agrammatic primary progressive aphasia.
Publication TypeJournal Article
Year of Publication2018
AuthorsHenry ML, H Hubbard I, Grasso SM, Mandelli MLuisa, Wilson SM, Sathishkumar MT, Fridriksson J, Daigle W, Boxer AL, Miller BL, Gorno-Tempini MLuisa
JournalBrain
Volume141
Issue6
Pagination1799-1814
Date Published2018 06 01
ISSN1460-2156
KeywordsAged, Aphasia, Primary Progressive, Aphasia, Wernicke, Female, Follow-Up Studies, Humans, Image Processing, Computer-Assisted, Magnetic Resonance Imaging, Male, Middle Aged, Neuropsychological Tests, Speech, Speech Therapy, Treatment Outcome
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.

DOI10.1093/brain/awy101
Alternate JournalBrain
PubMed ID29718131
PubMed Central IDPMC5972572
Grant ListP50 AG023501 / AG / NIA NIH HHS / United States
P50 DC014664 / DC / NIDCD NIH HHS / United States
R03 DC013403 / DC / NIDCD NIH HHS / United States
R01 AG038791 / AG / NIA NIH HHS / United States
R21 DC016080 / DC / NIDCD NIH HHS / United States
R01 DC013270 / DC / NIDCD NIH HHS / United States
P01 AG019724 / AG / NIA NIH HHS / United States
R01 DC016291 / DC / NIDCD NIH HHS / United States
R01 NS100440 / NS / NINDS NIH HHS / United States
R01 NS050915 / NS / NINDS NIH HHS / United States
U54 NS092089 / NS / NINDS NIH HHS / United States
K24 DC015544 / DC / NIDCD NIH HHS / United States