An Anti-β-Amyloid Vaccine for Treating Cognitive Deficits in a Mouse Model of Down Syndrome.

In Down syndrome (DS) or trisomy of chromosome 21, the β-amyloid (Aβ) peptide product of the amyloid precursor protein (APP) is present in excess. Evidence points to increased APP gene dose and Aβ as playing a critical role in cognitive difficulties experienced by people with DS. Particularly, Aβ is...

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Main Authors: Pavel V Belichenko, Rime Madani, Lorianne Rey-Bellet, Maria Pihlgren, Ann Becker, Adeline Plassard, Stephanie Vuillermot, Valérie Giriens, Rachel L Nosheny, Alexander M Kleschevnikov, Janice S Valletta, Sara K S Bengtsson, Gordon R Linke, Michael T Maloney, David T Hickman, Pedro Reis, Anne Granet, Dorin Mlaki, Maria Pilar Lopez-Deber, Long Do, Nishant Singhal, Eliezer Masliah, Matthew L Pearn, Andrea Pfeifer, Andreas Muhs, William C Mobley
Format: Article
Language:English
Published: Public Library of Science (PLoS) 2016-01-01
Series:PLoS ONE
Online Access:https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0152471&type=printable
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author Pavel V Belichenko
Rime Madani
Lorianne Rey-Bellet
Maria Pihlgren
Ann Becker
Adeline Plassard
Stephanie Vuillermot
Valérie Giriens
Rachel L Nosheny
Alexander M Kleschevnikov
Janice S Valletta
Sara K S Bengtsson
Gordon R Linke
Michael T Maloney
David T Hickman
Pedro Reis
Anne Granet
Dorin Mlaki
Maria Pilar Lopez-Deber
Long Do
Nishant Singhal
Eliezer Masliah
Matthew L Pearn
Andrea Pfeifer
Andreas Muhs
William C Mobley
author_facet Pavel V Belichenko
Rime Madani
Lorianne Rey-Bellet
Maria Pihlgren
Ann Becker
Adeline Plassard
Stephanie Vuillermot
Valérie Giriens
Rachel L Nosheny
Alexander M Kleschevnikov
Janice S Valletta
Sara K S Bengtsson
Gordon R Linke
Michael T Maloney
David T Hickman
Pedro Reis
Anne Granet
Dorin Mlaki
Maria Pilar Lopez-Deber
Long Do
Nishant Singhal
Eliezer Masliah
Matthew L Pearn
Andrea Pfeifer
Andreas Muhs
William C Mobley
author_sort Pavel V Belichenko
collection DOAJ
description In Down syndrome (DS) or trisomy of chromosome 21, the β-amyloid (Aβ) peptide product of the amyloid precursor protein (APP) is present in excess. Evidence points to increased APP gene dose and Aβ as playing a critical role in cognitive difficulties experienced by people with DS. Particularly, Aβ is linked to the late-life emergence of dementia as associated with neuropathological markers of Alzheimer's disease (AD). At present, no treatment targets Aβ-related pathogenesis in people with DS. Herein we used a vaccine containing the Aβ 1-15 peptide embedded into liposomes together with the adjuvant monophosphoryl lipid A (MPLA). Ts65Dn mice, a model of DS, were immunized with the anti-Aβ vaccine at 5 months of age and were examined for cognitive measures at 8 months of age. The status of basal forebrain cholinergic neurons and brain levels of APP and its proteolytic products were measured. Immunization of Ts65Dn mice resulted in robust anti-Aβ IgG titers, demonstrating the ability of the vaccine to break self-tolerance. The vaccine-induced antibodies reacted with Aβ without detectable binding to either APP or its C-terminal fragments. Vaccination of Ts65Dn mice resulted in a modest, but non-significant reduction in brain Aβ levels relative to vehicle-treated Ts65Dn mice, resulting in similar levels of Aβ as diploid (2N) mice. Importantly, vaccinated Ts65Dn mice showed resolution of memory deficits in the novel object recognition and contextual fear conditioning tests, as well as reduction of cholinergic neuron atrophy. No treatment adverse effects were observed; vaccine did not result in inflammation, cellular infiltration, or hemorrhage. These data are the first to show that an anti-Aβ immunotherapeutic approach may act to target Aβ-related pathology in a mouse model of DS.
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spelling doaj-art-e6e5615e619c41a0ad64eafb049c71c72025-08-20T01:51:24ZengPublic Library of Science (PLoS)PLoS ONE1932-62032016-01-01113e015247110.1371/journal.pone.0152471An Anti-β-Amyloid Vaccine for Treating Cognitive Deficits in a Mouse Model of Down Syndrome.Pavel V BelichenkoRime MadaniLorianne Rey-BelletMaria PihlgrenAnn BeckerAdeline PlassardStephanie VuillermotValérie GiriensRachel L NoshenyAlexander M KleschevnikovJanice S VallettaSara K S BengtssonGordon R LinkeMichael T MaloneyDavid T HickmanPedro ReisAnne GranetDorin MlakiMaria Pilar Lopez-DeberLong DoNishant SinghalEliezer MasliahMatthew L PearnAndrea PfeiferAndreas MuhsWilliam C MobleyIn Down syndrome (DS) or trisomy of chromosome 21, the β-amyloid (Aβ) peptide product of the amyloid precursor protein (APP) is present in excess. Evidence points to increased APP gene dose and Aβ as playing a critical role in cognitive difficulties experienced by people with DS. Particularly, Aβ is linked to the late-life emergence of dementia as associated with neuropathological markers of Alzheimer's disease (AD). At present, no treatment targets Aβ-related pathogenesis in people with DS. Herein we used a vaccine containing the Aβ 1-15 peptide embedded into liposomes together with the adjuvant monophosphoryl lipid A (MPLA). Ts65Dn mice, a model of DS, were immunized with the anti-Aβ vaccine at 5 months of age and were examined for cognitive measures at 8 months of age. The status of basal forebrain cholinergic neurons and brain levels of APP and its proteolytic products were measured. Immunization of Ts65Dn mice resulted in robust anti-Aβ IgG titers, demonstrating the ability of the vaccine to break self-tolerance. The vaccine-induced antibodies reacted with Aβ without detectable binding to either APP or its C-terminal fragments. Vaccination of Ts65Dn mice resulted in a modest, but non-significant reduction in brain Aβ levels relative to vehicle-treated Ts65Dn mice, resulting in similar levels of Aβ as diploid (2N) mice. Importantly, vaccinated Ts65Dn mice showed resolution of memory deficits in the novel object recognition and contextual fear conditioning tests, as well as reduction of cholinergic neuron atrophy. No treatment adverse effects were observed; vaccine did not result in inflammation, cellular infiltration, or hemorrhage. These data are the first to show that an anti-Aβ immunotherapeutic approach may act to target Aβ-related pathology in a mouse model of DS.https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0152471&type=printable
spellingShingle Pavel V Belichenko
Rime Madani
Lorianne Rey-Bellet
Maria Pihlgren
Ann Becker
Adeline Plassard
Stephanie Vuillermot
Valérie Giriens
Rachel L Nosheny
Alexander M Kleschevnikov
Janice S Valletta
Sara K S Bengtsson
Gordon R Linke
Michael T Maloney
David T Hickman
Pedro Reis
Anne Granet
Dorin Mlaki
Maria Pilar Lopez-Deber
Long Do
Nishant Singhal
Eliezer Masliah
Matthew L Pearn
Andrea Pfeifer
Andreas Muhs
William C Mobley
An Anti-β-Amyloid Vaccine for Treating Cognitive Deficits in a Mouse Model of Down Syndrome.
PLoS ONE
title An Anti-β-Amyloid Vaccine for Treating Cognitive Deficits in a Mouse Model of Down Syndrome.
title_full An Anti-β-Amyloid Vaccine for Treating Cognitive Deficits in a Mouse Model of Down Syndrome.
title_fullStr An Anti-β-Amyloid Vaccine for Treating Cognitive Deficits in a Mouse Model of Down Syndrome.
title_full_unstemmed An Anti-β-Amyloid Vaccine for Treating Cognitive Deficits in a Mouse Model of Down Syndrome.
title_short An Anti-β-Amyloid Vaccine for Treating Cognitive Deficits in a Mouse Model of Down Syndrome.
title_sort anti β amyloid vaccine for treating cognitive deficits in a mouse model of down syndrome
url https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0152471&type=printable
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