Detection of TDP-43 seeding activity in the olfactory mucosa from patients with frontotemporal dementia.

TAR DNA-binding protein 43 frontotemporal dementia frontotemporal lobar degeneration olfactory mucosa seeding seeding amplification assays

Journal

Alzheimer's & dementia : the journal of the Alzheimer's Association
ISSN: 1552-5279
Titre abrégé: Alzheimers Dement
Pays: United States
ID NLM: 101231978

Informations de publication

Date de publication:
31 Oct 2023
Historique:
revised: 10 10 2023
received: 25 07 2023
accepted: 11 10 2023
medline: 1 11 2023
pubmed: 1 11 2023
entrez: 1 11 2023
Statut: aheadofprint

Résumé

We assessed TAR DNA-binding protein 43 (TDP-43) seeding activity and aggregates detection in olfactory mucosa of patients with frontotemporal lobar degeneration with TDP-43-immunoreactive pathology (FTLD-TDP) by TDP-43 seeding amplification assay (TDP43-SAA) and immunocytochemical analysis. The TDP43-SAA was optimized using frontal cortex samples from 16 post mortem cases with FTLD-TDP, FTLD with tau inclusions, and controls. Subsequently, olfactory mucosa samples were collected from 17 patients with FTLD-TDP, 15 healthy controls, and three patients carrying MAPT variants. TDP43-SAA discriminated with 100% accuracy post mortem cases presenting or lacking TDP-43 neuropathology. TDP-43 seeding activity was detectable in the olfactory mucosa, and 82.4% of patients with FTLD-TDP tested positive, whereas 86.7% of controls tested negative (P < 0.001). Two out of three patients with MAPT mutations tested negative. In TDP43-SAA positive samples, cytoplasmatic deposits of phosphorylated TDP-43 in the olfactory neural cells were detected. TDP-43 aggregates can be detectable in olfactory mucosa, suggesting that TDP43-SAA might be useful for identifying and monitoring FTLD-TDP in living patients.

Identifiants

pubmed: 37908186
doi: 10.1002/alz.13541
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Subventions

Organisme : NIA NIH HHS
ID : P30 AG010133
Pays : United States

Informations de copyright

© 2023 The Authors. Alzheimer's & Dementia published by Wiley Periodicals LLC on behalf of Alzheimer's Association.

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Auteurs

Elena Fontana (E)

Department of Neuroscience, Biomedicine and Movement Sciences, University of Verona, Verona, Italy.

Matilde Bongianni (M)

Department of Neuroscience, Biomedicine and Movement Sciences, University of Verona, Verona, Italy.

Alberto Benussi (A)

Department of Clinical and Experimental Sciences, University of Brescia, Brescia, Italy.
Department of Continuity of Care and Frailty, ASST Spedali Civili Brescia Hospital, Brescia, Italy.

Erika Bronzato (E)

Department of Neuroscience, Biomedicine and Movement Sciences, University of Verona, Verona, Italy.

Carlo Scialo (C)

Department of Quantitative Biomedicine, University of Zurich, Zurich, Switzerland.

Luca Sacchetto (L)

Department of Surgery, Dentistry, Paediatrics and Gynaecology, Otolaryngology Section, University of Verona, Verona, Italy.

Annachiara Cagnin (A)

Neurology Unit, Department of Neuroscience, University of Padova, Padua, Italy.
Padova Neuroscience Center (PNC), University of Padova, Padua, Italy.

Santina Castriciano (S)

Copan, S.P.A, Brescia, Italy.

Emanuele Buratti (E)

International Centre for Genetic Engineering and Biotechnology (ICGEB), Trieste, Italy.

Fabrizio Gardoni (F)

Department of Pharmacological and Biomolecular Sciences (DiSFeB) "Rodolfo Paoletti,", University of Milan, Milan, Italy.

Maria Italia (M)

Department of Pharmacological and Biomolecular Sciences (DiSFeB) "Rodolfo Paoletti,", University of Milan, Milan, Italy.

Alberto Schreiber (A)

Otorhinolaryngology Unit, Head and Neck Surgery, ASST Spedali Civili, University of Brescia, Brescia, Italy.

Chiara Ferracin (C)

Laboratory of Prion Biology, Department of Neuroscience, Scuola Internazionale Superiore Di Studi Avanzati (SISSA), Trieste, Italy.

Michele Fiorini (M)

Department of Neuroscience, Biomedicine and Movement Sciences, University of Verona, Verona, Italy.

Kathy L Newell (KL)

Department of Pathology and Laboratory Medicine, School of Medicine, Indiana University, Indianapolis, Indiana, USA.

Laura Cracco (L)

Department of Pathology and Laboratory Medicine, School of Medicine, Indiana University, Indianapolis, Indiana, USA.

Holly J Garringer (HJ)

Department of Pathology and Laboratory Medicine, School of Medicine, Indiana University, Indianapolis, Indiana, USA.

Maria Paola Cecchini (MP)

Department of Neuroscience, Biomedicine and Movement Sciences, University of Verona, Verona, Italy.

Magdalini Polymenidou (M)

Department of Quantitative Biomedicine, University of Zurich, Zurich, Switzerland.

Alessandro Padovani (A)

Department of Clinical and Experimental Sciences, University of Brescia, Brescia, Italy.
Department of Continuity of Care and Frailty, ASST Spedali Civili Brescia Hospital, Brescia, Italy.

Salvatore Monaco (S)

Department of Neuroscience, Biomedicine and Movement Sciences, University of Verona, Verona, Italy.

Giuseppe Legname (G)

Laboratory of Prion Biology, Department of Neuroscience, Scuola Internazionale Superiore Di Studi Avanzati (SISSA), Trieste, Italy.

Bernardino Ghetti (B)

Department of Pathology and Laboratory Medicine, School of Medicine, Indiana University, Indianapolis, Indiana, USA.

Barbara Borroni (B)

Department of Clinical and Experimental Sciences, University of Brescia, Brescia, Italy.
Department of Continuity of Care and Frailty, ASST Spedali Civili Brescia Hospital, Brescia, Italy.

Gianluigi Zanusso (G)

Department of Neuroscience, Biomedicine and Movement Sciences, University of Verona, Verona, Italy.

Classifications MeSH