Progressive phonagnosia in a telephone operator carrying a C9orf72 expansion.


Journal

Cortex; a journal devoted to the study of the nervous system and behavior
ISSN: 1973-8102
Titre abrégé: Cortex
Pays: Italy
ID NLM: 0100725

Informations de publication

Date de publication:
11 2020
Historique:
received: 30 08 2019
revised: 21 02 2020
accepted: 13 05 2020
pubmed: 23 9 2020
medline: 22 6 2021
entrez: 22 9 2020
Statut: ppublish

Résumé

Selectivity is the rule, rather than the exception, in neurodegenerative disease. A retired telephone operator carrying a C9orf72 expansion developed phonagnosia, a selective impairment of voice recognition, contrasting with intact person knowledge and recognition of faces, as a presenting sign of genetically confirmed fronto-temporal dementia. Since the dysfunction in this patient fell into his area of professional expertise, we discuss if overload in voice related neural networks might have caused failure propagating to connected nodes. The interaction with downstream molecular events, triggered by the C9orf72 expansion, may have led to breakdown at the network level, leading to this specific phenotype.

Identifiants

pubmed: 32961393
pii: S0010-9452(20)30295-1
doi: 10.1016/j.cortex.2020.05.022
pii:
doi:

Substances chimiques

C9orf72 Protein 0
C9orf72 protein, human 0

Banques de données

ClinicalTrials.gov
['NCT02363062']

Types de publication

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

Langues

eng

Sous-ensembles de citation

IM

Pagination

92-98

Informations de copyright

Copyright © 2020 Elsevier Ltd. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of competing interest Nothing to report. Publicly archived anonymised public study data or digital study materials are not available for ethical reasons, but readers seeking access to the data can contact the lead author. No part of the study procedures or analysis plans was preregistered in an institutional registry prior to the research being conducted.

Auteurs

Mira Didic (M)

APHM, Timone, Service de Neurologie et Neuropsychologie, Hôpital Timone Adultes, Marseille, France; Aix Marseille Univ, INSERM, INS, Inst Neurosci Syst, Marseille, France. Electronic address: mira.didic@ap-hm.fr.

Virginia Aglieri (V)

Institut de Neurosciences de la Timone, UMR 7289, CNRS and Aix-Marseille Université, 13005 Marseille, France. Electronic address: vi.aglieri@gmail.com.

Eve Tramoni-Nègre (E)

APHM, Timone, Service de Neurologie et Neuropsychologie, Hôpital Timone Adultes, Marseille, France; Aix Marseille Univ, INSERM, INS, Inst Neurosci Syst, Marseille, France. Electronic address: eve.tramoni@ap-hm.fr.

Lucas Ronat (L)

Aix Marseille Univ, INSERM, INS, Inst Neurosci Syst, Marseille, France. Electronic address: lucas.ronat@ap-hm.fr.

Isabelle Le Ber (I)

Sorbonne Universités, UPMC Univ Paris 06, Inserm U1127, CNRS UMR 7225, Institut du Cerveau et la Moelle épinière (ICM), AP-HP - Hôpital Pitié-Salpêtrière, Paris, France; Centre de référence des démences rares ou précoces, IM2A, Département de Neurologie, AP-HP - Hôpital Pitié-Salpêtrière, Paris, France. Electronic address: isabelle.leber@upmc.fr.

Mathieu Ceccaldi (M)

APHM, Timone, Service de Neurologie et Neuropsychologie, Hôpital Timone Adultes, Marseille, France; Aix Marseille Univ, INSERM, INS, Inst Neurosci Syst, Marseille, France. Electronic address: mathieu-pierre.ceccaldi@ap-hm.fr.

Pascal Belin (P)

Institut de Neurosciences de la Timone, UMR 7289, CNRS and Aix-Marseille Université, 13005 Marseille, France; Department of Psychology, Université de Montréal, Montreal, QC H2V 2S9, Canada. Electronic address: pascal.belin@univ-amu.fr.

Olivier Felician (O)

APHM, Timone, Service de Neurologie et Neuropsychologie, Hôpital Timone Adultes, Marseille, France; Aix Marseille Univ, INSERM, INS, Inst Neurosci Syst, Marseille, France. Electronic address: olivier.felician@ap-hm.fr.

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Classifications MeSH