Diagnostic and prognostic value of cerebrospinal fluid SNAP-25 and neurogranin in Creutzfeldt-Jakob disease in a clinical setting cohort of rapidly progressive dementias.

Cognitive Disorders Creutzfeldt-Jakob Dementia Neurofilament Neurogranin Prion disease SNAP-25 Synapses Tau

Journal

Alzheimer's research & therapy
ISSN: 1758-9193
Titre abrégé: Alzheimers Res Ther
Pays: England
ID NLM: 101511643

Informations de publication

Date de publication:
08 09 2023
Historique:
received: 10 03 2023
accepted: 27 08 2023
medline: 11 9 2023
pubmed: 9 9 2023
entrez: 8 9 2023
Statut: epublish

Résumé

The levels of synaptic markers synaptosomal-associated protein 25 (SNAP-25) and neurogranin (Ng) have been shown to increase early in the cerebrospinal fluid (CSF) of patients with Creutzfeldt-Jakob disease (CJD) and to have prognostic potential. However, no validation studies assessed these biomarkers' diagnostic and prognostic value in a large clinical setting cohort of rapidly progressive dementia. In this retrospective study, using commercially available immunoassays, we measured the levels of SNAP-25, Ng, 14-3-3, total-tau (t-tau), neurofilament light chain (NfL), and phospho-tau181 (p-tau) in CSF samples from consecutive patients with CJD (n = 220) or non-prion rapidly progressive dementia (np-RPD) (n = 213). We evaluated and compared the diagnostic accuracy of each CSF biomarker and biomarker combination by receiver operating characteristics curve (ROC) analyses, studied SNAP-25 and Ng CSF concentrations distribution across CJD subtypes, and estimated their association with survival using multivariable Cox regression analyses. CSF SNAP-25 and Ng levels were higher in CJD than in np-RPD (SNAP-25: 582, 95% CI 240-1250 vs. 115, 95% CI 78-157 pg/ml, p < 0.0001; Ng: 841, 95% CI 411-1473 vs. 390, 95% CI 260-766 pg/ml, p < 0.001). SNAP-25 diagnostic accuracy (AUC 0.902, 95% CI 0.873-0.931) exceeded that of 14-3-3 (AUC 0.853, 95% CI 0.816-0.889), t-tau (AUC 0.878, 95% CI 0.845-0.901), and the t-tau/p-tau ratio (AUC 0.884, 95% CI 0.851-0.916). In contrast, Ng performed worse (AUC 0.697, 95% CI 0.626-0.767) than all other surrogate biomarkers, except for NfL (AUC 0.649, 95% CI 0.593-0.705). SNAP-25 maintained a relatively high diagnostic value even for atypical CJD subtypes (AUC 0.792, 95% CI 0.729-0.854). In Cox regression analyses, SNAP-25 levels were significantly associated with survival in CJD (hazard ratio [HR] 1.71 95% CI 1.40-2.09). Conversely, Ng was associated with survival only in the most rapidly progressive CJD subtypes (sCJD MM(V)1 and gCJD M1) (HR 1.81 95% CI 1.21-2.93). In the clinical setting, CSF SNAP-25 is a viable alternative to t-tau, 14-3-3, and the t-tau/p-tau ratio in discriminating the CJD subtypes from other RPDs. Additionally, SNAP-25 and, to a lesser extent, Ng predict survival in CJD, showing prognostic power in the range of CSF t-tau/14-3-3 and NfL, respectively.

Sections du résumé

BACKGROUND
The levels of synaptic markers synaptosomal-associated protein 25 (SNAP-25) and neurogranin (Ng) have been shown to increase early in the cerebrospinal fluid (CSF) of patients with Creutzfeldt-Jakob disease (CJD) and to have prognostic potential. However, no validation studies assessed these biomarkers' diagnostic and prognostic value in a large clinical setting cohort of rapidly progressive dementia.
METHODS
In this retrospective study, using commercially available immunoassays, we measured the levels of SNAP-25, Ng, 14-3-3, total-tau (t-tau), neurofilament light chain (NfL), and phospho-tau181 (p-tau) in CSF samples from consecutive patients with CJD (n = 220) or non-prion rapidly progressive dementia (np-RPD) (n = 213). We evaluated and compared the diagnostic accuracy of each CSF biomarker and biomarker combination by receiver operating characteristics curve (ROC) analyses, studied SNAP-25 and Ng CSF concentrations distribution across CJD subtypes, and estimated their association with survival using multivariable Cox regression analyses.
RESULTS
CSF SNAP-25 and Ng levels were higher in CJD than in np-RPD (SNAP-25: 582, 95% CI 240-1250 vs. 115, 95% CI 78-157 pg/ml, p < 0.0001; Ng: 841, 95% CI 411-1473 vs. 390, 95% CI 260-766 pg/ml, p < 0.001). SNAP-25 diagnostic accuracy (AUC 0.902, 95% CI 0.873-0.931) exceeded that of 14-3-3 (AUC 0.853, 95% CI 0.816-0.889), t-tau (AUC 0.878, 95% CI 0.845-0.901), and the t-tau/p-tau ratio (AUC 0.884, 95% CI 0.851-0.916). In contrast, Ng performed worse (AUC 0.697, 95% CI 0.626-0.767) than all other surrogate biomarkers, except for NfL (AUC 0.649, 95% CI 0.593-0.705). SNAP-25 maintained a relatively high diagnostic value even for atypical CJD subtypes (AUC 0.792, 95% CI 0.729-0.854). In Cox regression analyses, SNAP-25 levels were significantly associated with survival in CJD (hazard ratio [HR] 1.71 95% CI 1.40-2.09). Conversely, Ng was associated with survival only in the most rapidly progressive CJD subtypes (sCJD MM(V)1 and gCJD M1) (HR 1.81 95% CI 1.21-2.93).
CONCLUSIONS
In the clinical setting, CSF SNAP-25 is a viable alternative to t-tau, 14-3-3, and the t-tau/p-tau ratio in discriminating the CJD subtypes from other RPDs. Additionally, SNAP-25 and, to a lesser extent, Ng predict survival in CJD, showing prognostic power in the range of CSF t-tau/14-3-3 and NfL, respectively.

Identifiants

pubmed: 37684653
doi: 10.1186/s13195-023-01300-y
pii: 10.1186/s13195-023-01300-y
pmc: PMC10485978
doi:

Substances chimiques

Neurogranin 132654-77-4
Synaptosomal-Associated Protein 25 0
Prions 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

150

Informations de copyright

© 2023. BioMed Central Ltd., part of Springer Nature.

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Auteurs

Giuseppe Mario Bentivenga (GM)

Department of Biomedical and Neuromotor Sciences (DiBiNeM), University of Bologna, Bologna, Italy.

Simone Baiardi (S)

Department of Biomedical and Neuromotor Sciences (DiBiNeM), University of Bologna, Bologna, Italy.

Andrea Mastrangelo (A)

Department of Biomedical and Neuromotor Sciences (DiBiNeM), University of Bologna, Bologna, Italy.

Corrado Zenesini (C)

IRCCS, Istituto delle Scienze Neurologiche di Bologna, Programma Neuropatologia delle Malattie Neurodegenerative, Bologna, Italy.

Angela Mammana (A)

IRCCS, Istituto delle Scienze Neurologiche di Bologna, Programma Neuropatologia delle Malattie Neurodegenerative, Bologna, Italy.

Barbara Polischi (B)

IRCCS, Istituto delle Scienze Neurologiche di Bologna, Programma Neuropatologia delle Malattie Neurodegenerative, Bologna, Italy.

Sabina Capellari (S)

Department of Biomedical and Neuromotor Sciences (DiBiNeM), University of Bologna, Bologna, Italy.
IRCCS, Istituto delle Scienze Neurologiche di Bologna, Programma Neuropatologia delle Malattie Neurodegenerative, Bologna, Italy.

Piero Parchi (P)

Department of Biomedical and Neuromotor Sciences (DiBiNeM), University of Bologna, Bologna, Italy. piero.parchi@unibo.it.
IRCCS, Istituto delle Scienze Neurologiche di Bologna, Programma Neuropatologia delle Malattie Neurodegenerative, Bologna, Italy. piero.parchi@unibo.it.

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