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High diagnostic value of second generation CSF RT-QuIC across the wide spectrum of CJD prions

An early and accurate in vivo diagnosis of rapidly progressive dementia remains challenging, despite its critical importance for the outcome of treatable forms, and the formulation of prognosis. Real-Time Quaking-Induced Conversion (RT-QuIC) is an in vitro assay that, for the first time, specificall...

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Autores principales: Franceschini, Alessia, Baiardi, Simone, Hughson, Andrew G., McKenzie, Neil, Moda, Fabio, Rossi, Marcello, Capellari, Sabina, Green, Alison, Giaccone, Giorgio, Caughey, Byron, Parchi, Piero
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5587608/
https://www.ncbi.nlm.nih.gov/pubmed/28878311
http://dx.doi.org/10.1038/s41598-017-10922-w
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author Franceschini, Alessia
Baiardi, Simone
Hughson, Andrew G.
McKenzie, Neil
Moda, Fabio
Rossi, Marcello
Capellari, Sabina
Green, Alison
Giaccone, Giorgio
Caughey, Byron
Parchi, Piero
author_facet Franceschini, Alessia
Baiardi, Simone
Hughson, Andrew G.
McKenzie, Neil
Moda, Fabio
Rossi, Marcello
Capellari, Sabina
Green, Alison
Giaccone, Giorgio
Caughey, Byron
Parchi, Piero
author_sort Franceschini, Alessia
collection PubMed
description An early and accurate in vivo diagnosis of rapidly progressive dementia remains challenging, despite its critical importance for the outcome of treatable forms, and the formulation of prognosis. Real-Time Quaking-Induced Conversion (RT-QuIC) is an in vitro assay that, for the first time, specifically discriminates patients with prion disease. Here, using cerebrospinal fluid (CSF) samples from 239 patients with definite or probable prion disease and 100 patients with a definite alternative diagnosis, we compared the performance of the first (PQ-CSF) and second generation (IQ-CSF) RT-QuIC assays, and investigated the diagnostic value of IQ-CSF across the broad spectrum of human prions. Our results confirm the high sensitivity of IQ-CSF for detecting human prions with a sub-optimal sensitivity for the sporadic CJD subtypes MM2C and MM2T, and a low sensitivity limited to variant CJD, Gerstmann-Sträussler-Scheinker syndrome and fatal familial insomnia. While we found no difference in specificity between PQ-CSF and IQ-CSF, the latter showed a significant improvement in sensitivity, allowing prion detection in about 80% of PQ-CSF negative CJD samples. Our results strongly support the implementation of IQ-CSF in clinical practice. By rapidly confirming or excluding CJD with high accuracy the assay is expected to improve the outcome for patients and their enrollment in therapeutic trials.
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spelling pubmed-55876082017-09-13 High diagnostic value of second generation CSF RT-QuIC across the wide spectrum of CJD prions Franceschini, Alessia Baiardi, Simone Hughson, Andrew G. McKenzie, Neil Moda, Fabio Rossi, Marcello Capellari, Sabina Green, Alison Giaccone, Giorgio Caughey, Byron Parchi, Piero Sci Rep Article An early and accurate in vivo diagnosis of rapidly progressive dementia remains challenging, despite its critical importance for the outcome of treatable forms, and the formulation of prognosis. Real-Time Quaking-Induced Conversion (RT-QuIC) is an in vitro assay that, for the first time, specifically discriminates patients with prion disease. Here, using cerebrospinal fluid (CSF) samples from 239 patients with definite or probable prion disease and 100 patients with a definite alternative diagnosis, we compared the performance of the first (PQ-CSF) and second generation (IQ-CSF) RT-QuIC assays, and investigated the diagnostic value of IQ-CSF across the broad spectrum of human prions. Our results confirm the high sensitivity of IQ-CSF for detecting human prions with a sub-optimal sensitivity for the sporadic CJD subtypes MM2C and MM2T, and a low sensitivity limited to variant CJD, Gerstmann-Sträussler-Scheinker syndrome and fatal familial insomnia. While we found no difference in specificity between PQ-CSF and IQ-CSF, the latter showed a significant improvement in sensitivity, allowing prion detection in about 80% of PQ-CSF negative CJD samples. Our results strongly support the implementation of IQ-CSF in clinical practice. By rapidly confirming or excluding CJD with high accuracy the assay is expected to improve the outcome for patients and their enrollment in therapeutic trials. Nature Publishing Group UK 2017-09-06 /pmc/articles/PMC5587608/ /pubmed/28878311 http://dx.doi.org/10.1038/s41598-017-10922-w Text en © The Author(s) 2017 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/.
spellingShingle Article
Franceschini, Alessia
Baiardi, Simone
Hughson, Andrew G.
McKenzie, Neil
Moda, Fabio
Rossi, Marcello
Capellari, Sabina
Green, Alison
Giaccone, Giorgio
Caughey, Byron
Parchi, Piero
High diagnostic value of second generation CSF RT-QuIC across the wide spectrum of CJD prions
title High diagnostic value of second generation CSF RT-QuIC across the wide spectrum of CJD prions
title_full High diagnostic value of second generation CSF RT-QuIC across the wide spectrum of CJD prions
title_fullStr High diagnostic value of second generation CSF RT-QuIC across the wide spectrum of CJD prions
title_full_unstemmed High diagnostic value of second generation CSF RT-QuIC across the wide spectrum of CJD prions
title_short High diagnostic value of second generation CSF RT-QuIC across the wide spectrum of CJD prions
title_sort high diagnostic value of second generation csf rt-quic across the wide spectrum of cjd prions
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5587608/
https://www.ncbi.nlm.nih.gov/pubmed/28878311
http://dx.doi.org/10.1038/s41598-017-10922-w
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