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Case report: Non-Alzheimer's disease tauopathy with logopenic variant primary progressive aphasia diagnosed using amyloid and tau PET
We report a patient with logopenic variant primary progressive aphasia (lv-PPA) who was diagnosed as having non-Alzheimer's disease (AD) tauopathy after multiple biophysical/biological examinations, including amyloid and (18)F-florzolotau tau positron emission tomography (PET), had been perform...
Autores principales: | , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Frontiers Media S.A.
2022
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9705984/ https://www.ncbi.nlm.nih.gov/pubmed/36457868 http://dx.doi.org/10.3389/fneur.2022.1049113 |
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author | Momota, Yuki Konishi, Mika Takahata, Keisuke Kishimoto, Taishiro Tezuka, Toshiki Bun, Shogyoku Tabuchi, Hajime Ito, Daisuke Mimura, Masaru |
author_facet | Momota, Yuki Konishi, Mika Takahata, Keisuke Kishimoto, Taishiro Tezuka, Toshiki Bun, Shogyoku Tabuchi, Hajime Ito, Daisuke Mimura, Masaru |
author_sort | Momota, Yuki |
collection | PubMed |
description | We report a patient with logopenic variant primary progressive aphasia (lv-PPA) who was diagnosed as having non-Alzheimer's disease (AD) tauopathy after multiple biophysical/biological examinations, including amyloid and (18)F-florzolotau tau positron emission tomography (PET), had been performed. A woman in her late 60s who had previously been diagnosed as having AD was referred to us for a further, detailed examination. She had been unaware of any symptoms at the time of AD diagnosis, but she subsequently became gradually aware of a speech impairment. She talked nearly completely and fluently, although she occasionally exhibited word-finding difficulty and made phonological errors during naming, word fluency testing, and sentence repetition; these findings met the criteria for the diagnosis of lv-PPA, which is known to be observed more commonly in AD than in other proteinopathies. Magnetic resonance imaging, single photon emission computed tomography, and plasma phosphorylated tau and plasma neurofilament light chain measurements showed an AD-like pattern. However, both (11)C-Pittsburgh compound-B and (18)F-florbetaben amyloid PET showed negative results, whereas (18)F-florzolotau tau PET yielded positive results, with radio signals predominantly in the left superior temporal gyrus, middle temporal gyrus, supramarginal gyrus, and frontal operculum. Whole-genome sequencing revealed no known dominantly inherited mutations in AD or frontotemporal lobar degeneration genes, including the genes encoding amyloid precursor protein, microtubule-associated protein tau, presenilin 1 and 2. To the best of our knowledge, this patient was a rare case of lv-PPA who was diagnosed as having non-AD tauopathy based on the results of multiple examinations, including whole-genome sequencing, plasma measurement, and amyloid and (18)F-florzolotau tau PET. This case underscores the clinicopathologically heterogeneous nature of this syndrome. |
format | Online Article Text |
id | pubmed-9705984 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-97059842022-11-30 Case report: Non-Alzheimer's disease tauopathy with logopenic variant primary progressive aphasia diagnosed using amyloid and tau PET Momota, Yuki Konishi, Mika Takahata, Keisuke Kishimoto, Taishiro Tezuka, Toshiki Bun, Shogyoku Tabuchi, Hajime Ito, Daisuke Mimura, Masaru Front Neurol Neurology We report a patient with logopenic variant primary progressive aphasia (lv-PPA) who was diagnosed as having non-Alzheimer's disease (AD) tauopathy after multiple biophysical/biological examinations, including amyloid and (18)F-florzolotau tau positron emission tomography (PET), had been performed. A woman in her late 60s who had previously been diagnosed as having AD was referred to us for a further, detailed examination. She had been unaware of any symptoms at the time of AD diagnosis, but she subsequently became gradually aware of a speech impairment. She talked nearly completely and fluently, although she occasionally exhibited word-finding difficulty and made phonological errors during naming, word fluency testing, and sentence repetition; these findings met the criteria for the diagnosis of lv-PPA, which is known to be observed more commonly in AD than in other proteinopathies. Magnetic resonance imaging, single photon emission computed tomography, and plasma phosphorylated tau and plasma neurofilament light chain measurements showed an AD-like pattern. However, both (11)C-Pittsburgh compound-B and (18)F-florbetaben amyloid PET showed negative results, whereas (18)F-florzolotau tau PET yielded positive results, with radio signals predominantly in the left superior temporal gyrus, middle temporal gyrus, supramarginal gyrus, and frontal operculum. Whole-genome sequencing revealed no known dominantly inherited mutations in AD or frontotemporal lobar degeneration genes, including the genes encoding amyloid precursor protein, microtubule-associated protein tau, presenilin 1 and 2. To the best of our knowledge, this patient was a rare case of lv-PPA who was diagnosed as having non-AD tauopathy based on the results of multiple examinations, including whole-genome sequencing, plasma measurement, and amyloid and (18)F-florzolotau tau PET. This case underscores the clinicopathologically heterogeneous nature of this syndrome. Frontiers Media S.A. 2022-11-15 /pmc/articles/PMC9705984/ /pubmed/36457868 http://dx.doi.org/10.3389/fneur.2022.1049113 Text en Copyright © 2022 Momota, Konishi, Takahata, Kishimoto, Tezuka, Bun, Tabuchi, Ito and Mimura. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms. |
spellingShingle | Neurology Momota, Yuki Konishi, Mika Takahata, Keisuke Kishimoto, Taishiro Tezuka, Toshiki Bun, Shogyoku Tabuchi, Hajime Ito, Daisuke Mimura, Masaru Case report: Non-Alzheimer's disease tauopathy with logopenic variant primary progressive aphasia diagnosed using amyloid and tau PET |
title | Case report: Non-Alzheimer's disease tauopathy with logopenic variant primary progressive aphasia diagnosed using amyloid and tau PET |
title_full | Case report: Non-Alzheimer's disease tauopathy with logopenic variant primary progressive aphasia diagnosed using amyloid and tau PET |
title_fullStr | Case report: Non-Alzheimer's disease tauopathy with logopenic variant primary progressive aphasia diagnosed using amyloid and tau PET |
title_full_unstemmed | Case report: Non-Alzheimer's disease tauopathy with logopenic variant primary progressive aphasia diagnosed using amyloid and tau PET |
title_short | Case report: Non-Alzheimer's disease tauopathy with logopenic variant primary progressive aphasia diagnosed using amyloid and tau PET |
title_sort | case report: non-alzheimer's disease tauopathy with logopenic variant primary progressive aphasia diagnosed using amyloid and tau pet |
topic | Neurology |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9705984/ https://www.ncbi.nlm.nih.gov/pubmed/36457868 http://dx.doi.org/10.3389/fneur.2022.1049113 |
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