Dysphagia in primary progressive aphasia: Clinical predictors and neuroanatomical basis.
Alzheimer dementia
dysphagia
primary progressive aphasia
semantic dementia
swallowing
Journal
European journal of neurology
ISSN: 1468-1331
Titre abrégé: Eur J Neurol
Pays: England
ID NLM: 9506311
Informations de publication
Date de publication:
21 Jun 2024
21 Jun 2024
Historique:
revised:
29
04
2024
received:
23
02
2024
accepted:
12
05
2024
medline:
16
7
2024
pubmed:
16
7
2024
entrez:
16
7
2024
Statut:
aheadofprint
Résumé
Dysphagia is an important feature of neurodegenerative diseases and potentially life-threatening in primary progressive aphasia (PPA) but remains poorly characterized in these syndromes. We hypothesized that dysphagia would be more prevalent in nonfluent/agrammatic variant (nfv)PPA than other PPA syndromes, predicted by accompanying motor features, and associated with atrophy affecting regions implicated in swallowing control. In a retrospective case-control study at our tertiary referral centre, we recruited 56 patients with PPA (21 nfvPPA, 22 semantic variant [sv]PPA, 13 logopenic variant [lv]PPA). Using a pro forma based on caregiver surveys and clinical records, we documented dysphagia (present/absent) and associated, potentially predictive clinical, cognitive, and behavioural features. These were used to train a machine learning model. Patients' brain magnetic resonance imaging scans were assessed using voxel-based morphometry and region-of-interest analyses comparing differential atrophy profiles associated with dysphagia presence/absence. Dysphagia was significantly more prevalent in nfvPPA (43% vs. 5% svPPA and no lvPPA). The machine learning model revealed a hierarchy of features predicting dysphagia in the nfvPPA group, with excellent classification accuracy (90.5%, 95% confidence interval = 77.9-100); the strongest predictor was orofacial apraxia, followed by older age, parkinsonism, more severe behavioural disturbance, and more severe cognitive impairment. Significant grey matter atrophy correlates of dysphagia in nfvPPA were identified in left middle frontal, right superior frontal, and right supramarginal gyri and right caudate. Dysphagia is a common feature of nfvPPA, linked to underlying corticosubcortical network dysfunction. Clinicians should anticipate this symptom particularly in the context of other motor features and more severe disease.
Sections du résumé
BACKGROUND AND PURPOSE
OBJECTIVE
Dysphagia is an important feature of neurodegenerative diseases and potentially life-threatening in primary progressive aphasia (PPA) but remains poorly characterized in these syndromes. We hypothesized that dysphagia would be more prevalent in nonfluent/agrammatic variant (nfv)PPA than other PPA syndromes, predicted by accompanying motor features, and associated with atrophy affecting regions implicated in swallowing control.
METHODS
METHODS
In a retrospective case-control study at our tertiary referral centre, we recruited 56 patients with PPA (21 nfvPPA, 22 semantic variant [sv]PPA, 13 logopenic variant [lv]PPA). Using a pro forma based on caregiver surveys and clinical records, we documented dysphagia (present/absent) and associated, potentially predictive clinical, cognitive, and behavioural features. These were used to train a machine learning model. Patients' brain magnetic resonance imaging scans were assessed using voxel-based morphometry and region-of-interest analyses comparing differential atrophy profiles associated with dysphagia presence/absence.
RESULTS
RESULTS
Dysphagia was significantly more prevalent in nfvPPA (43% vs. 5% svPPA and no lvPPA). The machine learning model revealed a hierarchy of features predicting dysphagia in the nfvPPA group, with excellent classification accuracy (90.5%, 95% confidence interval = 77.9-100); the strongest predictor was orofacial apraxia, followed by older age, parkinsonism, more severe behavioural disturbance, and more severe cognitive impairment. Significant grey matter atrophy correlates of dysphagia in nfvPPA were identified in left middle frontal, right superior frontal, and right supramarginal gyri and right caudate.
CONCLUSIONS
CONCLUSIONS
Dysphagia is a common feature of nfvPPA, linked to underlying corticosubcortical network dysfunction. Clinicians should anticipate this symptom particularly in the context of other motor features and more severe disease.
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
e16370Subventions
Organisme : National Institute for Health Research Rare Disease Translational Research Collaboration
ID : BRC149/NS/MH
Organisme : Medical Research Council Clinician Scientist Fellowship
ID : MR/M008525/1
Organisme : Alzheimer's Research UK
Organisme : Alzheimer's Society
Pays : United Kingdom
Organisme : National Brain Appeal
Organisme : UK Dementia Research Institute
Organisme : Invention for Innovation
ID : NIHR204280
Organisme : Association of British Neurologists Clinical Research Training Fellowship
Organisme : Miriam Marks Brain Research UK Senior Fellowship
Organisme : Wellcome Institutional Strategic Support Fund Award
ID : 204841/Z/16/Z
Organisme : The National Brain Appeal Royal National Institute for Deaf People (Discovery Grant G105_WARREN)National Institute for Health Research University College London Hospitals Biomedical Research Centre NIHR Advanced Fellowship NIHR302240 UKRI and Wellcome Trust
ID : 204841/Z/16/Z
Informations de copyright
© 2024 The Author(s). European Journal of Neurology published by John Wiley & Sons Ltd on behalf of European Academy of Neurology.
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